Lessons for the UK on implementation and evaluation of breastfeeding support: evidence syntheses and stakeholder engagement

Article history

The research reported in this issue of the journal was funded by the HSDR programme or one of its preceding programmes as award number NIHR130995. The contractual start date was in February 2021. The draft manuscript began editorial review in March 2023 and was accepted for publication in November 2023. The authors have been wholly responsible for all data collection, analysis and interpretation, and for writing up their work. The HSDR editors and production house have tried to ensure the accuracy of the authors’ manuscript and would like to thank the reviewers for their constructive comments on the draft document. However, they do not accept liability for damages or losses arising from material published in this article.

Copyright statement

Copyright © 2024 Gavine et al. This work was produced by Gavine et al. under the terms of a commissioning contract issued by the Secretary of State for Health and Social Care. This is an Open Access publication distributed under the terms of the Creative Commons Attribution CC BY 4.0 licence, which permits unrestricted use, distribution, reproduction and adaptation in any medium and for any purpose provided that it is properly attributed. See: https://creativecommons.org/licenses/by/4.0/. For attribution the title, original author(s), the publication source – NIHR Journals Library, and the DOI of the publication must be cited.

2024 Gavine et al.

Importance of breastfeeding

Breastfeeding has a significantly positive impact on multiple health outcomes across the lifespan. For children, this includes fewer deaths and hospital admissions for infectious diseases1–4 and reduced incidence of obesity, diabetes mellitus and dental disease. 5–7 Breastfeeding has been linked to improved educational and behavioural outcomes. 8–10 For women, breastfeeding is associated with a lower risk of cardiovascular disease, breast and ovarian cancer and diabetes mellitus. 11–13 The impact of breastfeeding on health outcomes applies across settings and population groups, including in high-income countries (HICs) such as the UK. Globally, the scaling up of breastfeeding to near-universal level could prevent 823,000 deaths of children under 5 years old and 20,000 annual deaths of women from breast cancer. 14 To optimise population health, global and UK infant recommendations are that infants should be breastfed (or receive breastmilk) exclusively for about 6 months and that this should continue as part of a mixed diet until 2 years or beyond. 15,16

Increased breastfeeding has the potential to reduce healthcare costs. 17,18 In addition to the important effects on health for women and children, breastfeeding has wider health system and societal impacts, including cost savings for the NHS and environmental benefits. The cost to the global economy of not breastfeeding has been estimated at £242B, and, in the UK, estimates are that £23.6M in additional treatment costs could be saved each year by increased breastfeeding. 17 A further cost to the NHS is the increasing number of prescriptions for specialist formula to treat cow’s milk protein allergy. 19 The environmental impact of not breastfeeding (i.e. feeding with infant formula) is significant, for example from plastics and resources used by the dairy industry. 20,21 Therefore, significant health, societal and environmental gains are to be had from increasing breastfeeding duration and exclusivity.

UK breastfeeding patterns

The UK has low breastfeeding rates. Following the cessation of the quinquennial UK-wide Infant Feeding Surveys, comprehensive, robust data on breastfeeding rates are lacking. For England, the most recent data (2020/21 data), reported by NHS trusts, showed a 72% initiation rate and 49% prevalence of breastfeeding at 6–8 weeks. 22 The comparative figures were 60% initiation and 45% prevalence at 6–8 weeks for Wales (2016 data)23 and 65% initiation and 43% prevalence at 6–8 weeks for Scotland (2018/19 data). 24 In Northern Ireland (2020 data), the initiation rate was 62% and prevalence at 6 weeks was 40%. 25 Rates of exclusive breastfeeding are much lower in all four countries. Throughout the UK, there is a marked social gradient in breastfeeding rates whereby women from socioeconomically deprived groups, those with lower education levels and adolescent women are least likely to breastfeed. 26 For example, in Scotland (2018/19 data),24 breastfeeding prevalence at 6–8 weeks was 62% in the wealthiest quintile compared with 28% in the most deprived quintile. The differences were starker by mother’s age, with breastfeeding prevalence at 6–8 weeks of 58% among mothers aged 40 years and 13% among mothers aged under 20 years. 24 In the UK, women from non-white ethnic groups had higher rates of breastfeeding initiation, prevalence and duration than white women; rates of exclusive breastfeeding after 1 week were similar. 26 Women and babies from the most deprived backgrounds and younger mothers have most to gain from the health benefits conferred by breastfeeding. It has also been reported that around 80% of women in the UK stop breastfeeding before they intended, causing distress26 and potentially leading to poorer mental health. 27,28

Comparing breastfeeding rates between the four countries of the UK, and with countries internationally, is fraught with difficulty, as data are collected in different ways, at different time points and for different years. Nevertheless, rates of breastfeeding in the UK are consistently reported to be lower than those in other European countries. For example, in 2015, a survey of European countries found that breastfeeding initiation rates ranged from 80% in the Netherlands to 98% in Norway and that breastfeeding prevalence at 2 months ranged from 64% in the Netherlands to 89% in Norway (both outcomes were reported by 6 of 11 countries). 29 The exception is Ireland, which has similar rates to the UK with a breastfeeding initiation rate of 64%30 and breastfeeding prevalence at 3 months of 35%. 29

Breastfeeding support

In the UK, formal breastfeeding support, comprising practical, informational, emotional and social support may be provided by healthcare practitioners, voluntary organisations and peer supporters. Women may also receive informal breastfeeding support from families and friends. However, many women report feeling unsupported by healthcare providers and their social networks, especially in the early weeks following birth. 31 This was exacerbated by the impact of COVID-19 on breastfeeding support services, which were already being reduced. 32,33

There is evidence that women living in deprived areas face multiple barriers to breastfeeding and accessing appropriate breastfeeding support. Common barriers include pain, the perception that they do not produce sufficient milk to meet their baby’s needs,34 embarrassment about breastfeeding in public and negative societal attitudes to breastfeeding. 34,35 While these barriers affect all women, they can be particularly challenging in settings where family and friends lack knowledge and experience of breastfeeding. 35 Women from disadvantaged backgrounds may value particularly the experiential knowledge and skills adapted to local contexts provided by peer support. 36 However, survey data suggested that coverage of breastfeeding peer support across the UK was variable and not accessed by socially disadvantaged women. 33 Additional barriers for women from minority ethnic groups, for example Bangladeshi women, include diverse cultural influences of their heritage and their areas of residence in the UK37 and cultural stereotypes held by healthcare providers. 38 There is strong global evidence that, for healthy women and babies, breastfeeding support is effective at increasing partial and exclusive breastfeeding. 39–42 However, these reviews combine evidence from high-, middle- and low-income countries, with most of the HIC evidence coming from the USA. Interventions tested in trials are heterogeneous and generally undertheorised. The extent to which global evidence is transferable to the UK setting is unclear. Previous evidence from UK-based trials is limited and has not demonstrated efficacy of interventions. 43–45 Feasibility studies in the UK show that peer support interventions are acceptable46,47 but effectiveness has not been established.

Women with multimorbidities

The prevalence of maternal chronic conditions is rising,48 which is in part due to increasing maternal age and the improved management of long-term conditions (LTCs). 49 For instance, UK data show that 2.3% of women have been diagnosed with diabetes either prior to or during pregnancy,50 0.5% have a diagnosis of inflammatory bowel disease,51 0.5–1.0% have a diagnosis of epilepsy,52 18.4% have a postnatal diagnosis of anxiety53 and 11.4% have a postnatal diagnosis of depression. 53 Moreover, the rates of gestational diabetes in pregnant women in the UK range from 1.2% to 24.2% depending on maternal characteristics and diagnostic method,54 and this increases the risk of developing type 2 diabetes 10-fold. 55

The prevalence of multiple long-term conditions (MLTC) in the UK is also rising, particularly among working-age adults. 56 Within a general adult population, the onset of MLTC happens 10–15 years earlier in those living in the most deprived areas than in those living in more affluent areas. 57 The MuM-PreDiCT study sought to identify the prevalence of multimorbidities specifically during pregnancy and reported that between 19.8% and 46.2% of pregnant women experience two or more LTCs. 58 LTCs were defined as conditions that had a significant impact on patients, and the specific 79 conditions included in the study were determined in consultation with stakeholders. 59 Unlike in the general adult population, it is not currently clear if the prevalence of MLTC is higher among women from areas of high socioeconomic deprivation. The MuM-PreDiCT study did not find higher odds of multimorbidities in women from areas of high socioeconomic deprivation or in any specific ethnic group. 58 Post hoc analysis explored whether this was being impacted by the health conditions used to define multimorbidity, as some conditions, such as irritable bowel syndrome, anxiety and polycystic ovarian syndrome, were higher in more affluent areas. 58 When a shortened list of conditions was used, socioeconomic deprivation was associated with multimorbidities after adjusting for maternal age and gravidity [adjusted odds ratio (aOR) 1.30, 95% confidence interval (CI) 1.08 to 1.57]. 58 However, this was no longer significant once body mass index (BMI) and smoking status were also adjusted for (aOR 1.05, 95% CI 0.87 to 1.27). MLTC were more common in mothers aged 45–49 years (aOR 1.8, 95% CI 1.0 to 3.20), and this remained significant when adjusted for other characteristics.

Living with MLTC can have a significant impact on mental well-being and can make engaging in other activities difficult. 60 Within the context of maternal health, experiencing a LTC during pregnancy is associated with mental health conditions in the postpartum period such as post-traumatic stress. 61 Mothers with LTCs are also more likely to experience other adverse determinants of health, such as intimate partner violence, smoking, living in poverty and a lack of educational qualifications. 62

There is some evidence for the management of single conditions during pregnancy and the postnatal period, for example diabetes,63 epilepsy,64 and depression,65 that is focused on the treatment modalities for that single condition. However, there is a complete lack of evidence on MLTC in mothers. Postnatal care, in particular, has been universally described as poor due to a lack of follow-up care and help for women to care for their babies. 66 Breastfeeding could present a challenge to women with MLTC, as is evidenced by significantly lower breastfeeding rates among women with single LTCs. 62,67 For instance, a study comparing UK women with lifelong limiting conditions found that breastfeeding rates at 3 months were lower in this group than among women without any conditions (25.6% vs. 33.4%);62 however, rates of initiation were similar. Data from Canada showed that although women with chronic diseases had similar odds of initiating breastfeeding, they were more likely to cease breastfeeding early than women in the general population (aOR 2.48, 95% CI 1.49 to 4.12). 68 Data from other countries also suggest that breastfeeding rates are lower among women with a range of specific conditions such as insulin-dependent diabetes (aOR 0.49, 95% CI 0.27 to 0.89), epilepsy (aOR 0.42, 95% CI 0.26 to 0.68)69 and rheumatoid arthritis (any breastfeeding at 3 months in women with rheumatoid arthritis = 26% vs. 46% of general population). There is currently a complete lack of evidence on breastfeeding rates in women with MLTC. 70

There are several reasons why women with LTCs may have additional difficulties breastfeeding, including a physiological delay in milk release to 72 hours after birth, an increased risk of early separation from the infant as a result of caesarean section and/or requirement for the infant to be placed in neonatal intensive care unit facilities, fatigue, and poor and inconsistent advice about the safety of medications. 68 Anecdotal evidence from the Breastfeeding Network has also identified a lack of joined-up care as a barrier to breastfeeding. As breastfeeding can confer significant health benefits to both mother and infant,14 there is a need for breastfeeding support interventions to provide effective support to all women that is tailored to their individual needs. 71

Economic impact

Breastfeeding in itself is considered a cost-effective intervention. 17,72 Increased breastfeeding has the potential to reduce healthcare costs. 17,18 In addition to the important effects on the health of women and children, breastfeeding has wider health system and societal impacts, including cost savings for the NHS and environmental benefits. The cost to the global economy of not breastfeeding has been estimated at US$570B (£396B) each year, with estimates indicating that 0.75% of gross national income in HICs is lost from not breastfeeding. 73 With a UK gross national income of £2505B in 2022,74 this equates to a value to the UK economy of £18.8B. For the UK health system, estimates are that £23.6M of additional treatment costs each year could be saved by increased breastfeeding. 17 This cost to the NHS is considered a conservative estimate, as a limited number of maternal and child-related illnesses were included in the analysis. A further cost to the NHS is the increasing number of prescriptions for specialist formula to treat cow’s milk protein allergy. 19 For example, an 800g tin of specialised formula (Aptamil Pepti^® 1 powder, Nutricia, Trowbridge, UK) prescribed for cow’s milk allergy, which would feed a baby under 6 months old for 1 week, costs the NHS £19.72 at 2023 prices. 75 The environmental impact of not breastfeeding (i.e. feeding with infant formula) is significant. For example, plastics and resources used by the dairy industry have a cost in carbon dioxide emissions equivalent to 50,000–77,500 cars on the road each year and a water footprint of 4700 l/kg. 20,21 Therefore, significant health, societal and environmental gains are to be had from increasing breastfeeding duration and exclusivity. In choosing a breastfeeding support intervention to implement into a health system, policy-makers need to understand not only the evidence of effect and contextual factors that should be considered but also the evidence of cost-effectiveness. With pressure on NHS resources, service managers need to ensure that any investment yields a positive return both in the short term, with increased breastfeeding, and in the long term, with reduced health service resource use and subsequent cost savings.

Why this research is needed

There is a need to find out what works to support women in the UK to meet their infant-feeding goals, to breastfeed for longer, and to increase rates of exclusive breastfeeding. This involves understanding the characteristics and components of breastfeeding support interventions that are likely to be effective and cost-effective in the UK, as well as how to implement and evaluate such interventions. This is particularly the case for populations where breastfeeding rates are low, including young mothers, women of low socioeconomic status, those from marginalised groups, and those with multimorbidities. Although this has been a policy aspiration in the UK for several decades, there is a gap in evidence regarding effective interventions. At a time when the NHS is struggling to meet demand, and life expectancy is stalling, cost-effective public health interventions targeted to disadvantaged communities are vital.

Aim and objectives

The aim was to synthesise global and UK evidence to co-create with stakeholders a framework to guide the implementation and evaluation of cost-effective breastfeeding support interventions in the NHS.

Study design

The study comprised evidence syntheses and economic evaluations with embedded stakeholder engagement, including patient and public involvement (PPI). We used principles of co-creation to ensure that study outputs were relevant to the NHS context. The main study comprised four interlinked work packages with a cross-cutting strand of stakeholder engagement and PPI, as shown in Figure 1. The main study took place over 2 years and the additional work took place over 9 months.

FIGURE 1.

Study flow diagram.

The methods for each evidence synthesis are described in the relevant chapters. In this chapter we present our approach to stakeholder engagement and PPI.

Stakeholder and parent engagement: main study

To ensure joint ownership,76 our approach was ‘active involvement’, defined as ‘the contribution of any person who would be a knowledge user but whose primary role is not research’, throughout the process of evidence synthesis, including planning, production and dissemination. 77 Involvement and co-creation were essential to enhance the quality and relevance of the evidence syntheses. 78,79 Stakeholders and parents were involved in three ways: a co-investigator (PB) from a breastfeeding support organisation represented service user views; the stakeholder working group, parents’ panel and focus group discussions ensured that the experiences of breastfeeding women and service providers were represented in key decisions; and attendees at four workshops co-created the study outputs. Here we describe the participants, activities and outcomes of the stakeholder working group, parents’ panel and focus group discussions. See Chapter 9 for details of the workshops.

Stakeholder and parent engagement (multiple long-term conditions)

Role of stakeholder engagement

The main purpose of the stakeholder working groups, the parents’ panel and the focus group discussion was to adapt the international evidence, that is, the findings of the reviews, to ensure relevance to the UK context and the NHS, and to coproduce the toolkit. The stakeholder engagement therefore influenced the interpretation and adaptation to the UK setting of the review findings rather than their methods. The exception to this was in influencing the decision on outcome time points and variables for the meta-regression for review 1.

Introduction

This chapter contains a summary of the methods and results section from the updated Cochrane review on breastfeeding support for healthy term women with healthy term babies. 83 The full review including table of characteristics, forest plots and risk of bias assessments is published in the Cochrane Library. 83 Parts of this chapter have been reproduced with permission from Wiley. Copyright © 2022 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd.

Objectives

1. To describe types of breastfeeding support for healthy breastfeeding women with healthy term babies.

2. To examine the effectiveness of different types of breastfeeding support interventions focusing on breastfeeding support provided on its own or breastfeeding support in combination with a wider maternal and child health intervention.

3. To examine the effectiveness of the following intervention characteristics on breastfeeding support:

   1. type of support (e.g. face to face, telephone, digital technologies, group or individual support, proactive or reactive)

   2. intensity of support (i.e. number of postnatal contacts)

   3. person delivering the intervention (e.g. healthcare professional, lay person)

   4. to examine whether the impact of support varied between high‐ and low‐ and middle‐income countries.

Methods

Results

A total of 590 trial reports were assessed for inclusion in this update (see Gavine et al. 83 for full details). This included 560 studies from the updated search, 16 trial reports that were awaiting classification in the previous version of the review, 8 studies that were ongoing in the last version of the review and 6 previously excluded studies that were reassessed due to the change in inclusion criteria. Of these, 72 met the inclusion criteria.

All studies (100 previously included and 72 newly identified studies) were assessed against Cochrane’s criteria for trustworthiness. Of the 100 previously included studies, we requested further information for 38, and of the new studies identified in this update, we required clarification for 43. In total, we received satisfactory responses for 27 studies. In total, 54 studies were reclassified to ‘awaiting classification’. The remaining studies were included, and this updated review includes 116 trials, of which 103 contribute data to the analyses.

In total, 249 studies were excluded with reasons (this comprises 139 reports from the updated search and 110 reports from previous versions of the review). The majority of studies (n = 136) were excluded as the intervention was not relevant to the review, for example interventions that were focused on education and/or promotion only and did not offer any support, interventions that were focused on other aspects of postnatal care, and antenatal-only interventions. We excluded any study that was not a RCT (n = 53). A further 49 studies were excluded because they did not focus on healthy mothers (e.g. coexisting medical conditions requiring additional care) or babies (e.g. preterm, low birthweight). Eleven studies were excluded because the comparator was not either standard care or an alternative non‐breastfeeding intervention. Finally, four studies were excluded as they were not research papers. For full details, see Gavine et al. 83 for characteristics of the excluded studies.

Risk-of-bias assessments

We considered that the overall risk of bias of trials included in the review was mixed. Blinding of participants and personnel is not feasible in such interventions, and, as studies utilised self‐report breastfeeding data, there is also a risk of bias in outcome assessment.

For full details of the risk-of-bias assessments, see Gavine et al. 83 A summary of the judgements is detailed in Figure 2.

FIGURE 2.

Risk-of-bias graph: review authors’ judgements about each risk-of-bias item presented as percentages across all included studies (Gavine et al. 83).

Chapter summary

The update of this Cochrane review on breastfeeding support for healthy term women identified 116 trials, of which 103 contribute data to the analyses. More than 98,816 mother–infant pairs were included. When ‘breastfeeding only’ support is offered to women, the duration and particularly the exclusivity of breastfeeding is likely to be increased. Support may also be more effective in reducing the number of women stopping breastfeeding at 3–4 months than at later time points. For ‘breastfeeding plus’ interventions the evidence is less certain.

There does not appear to be a difference in who provides the support (i.e. professional or non‐professional) or how it is provided (face to face, telephone, digital technologies or combinations). Indeed, various kinds of support may be needed in different geographical locations to meet the needs of the people within that locality.

Introduction

The Cochrane review update undertaken in our review 1 confirmed that there is ample evidence to know that breastfeeding women need support to be available and to be provided, and that such support is likely to make a difference. Such an evidence base also suggests that one key research question for the future is to identify how such support can best be provided consistently across countries and settings.

Therefore, there is now a need to improve the evidence base around scaling up issues for breastfeeding support interventions, which will require a greater emphasis on implementation and quality improvement approaches rather than effectiveness studies. To enable further advances in this area, it will be fundamental to identify and synthesise available qualitative and process evaluation data on existing interventions. The overall aim of this review was to conduct a theoretically informed mixed-methods synthesis of process evaluations of breastfeeding support interventions identified as effective in review 1.

Objectives

1. To identify qualitative and quantitative data from process evaluation studies linked to breastfeeding support interventions identified as effective in review 1.

2. To synthesise the views and experiences of those involved in receiving or delivering breastfeeding support interventions identified as effective in review 1.

3. To identify the contextual factors (barriers/facilitators) affecting the implementation of breastfeeding support interventions identified as effective in review 1.

Methods

The protocol for this systematic review is registered on PROSPERO (CRD42021229769).

Results

The searches identified 2894 records, which were assessed against the inclusion criteria. Title and abstract screening resulted in 243 records considered eligible or inconclusive. Full-text articles were then retrieved and assessed for eligibility. Two records could not be retrieved. Of the 241 records screened at full text, 225 were excluded. The main reason for exclusion was studies not being linked to an intervention identified as effective in review 1 (n = 84), followed by standalone studies that were not linked to any intervention (n = 51) and studies not involving implementation research and/or process evaluation data (e.g. pre-implementation or intervention development studies) from eligible interventions (n = 50). Other reasons for exclusion were studies linked to either interventions (n = 26) or populations (n = 6) not eligible for inclusion in review 1, and systematic reviews (n = 4) and other publication types not reporting primary research findings (n = 4). The remaining 16 studies were included in the final synthesis (Figure 3). The 16 studies are linked to 10 RCTs of effective interventions from review 1.

FIGURE 3.

Preferred Reporting Items for Systematic Reviews and Meta-Analyses flow diagram.

Chapter summary

This review comprised 16 studies linked to 10 interventions identified as effective in review 1, which reported the views and experiences of those delivering or receiving breastfeeding support. The quality of the included studies was mixed, but all study findings were judged to be at least fairly well supported by the data.

The synthesis resulted in three overarching themes, theoretically informed by the CFIR: (1) assessing the needs of those delivering and receiving breastfeeding support interventions; (2) assessing the context and optimising delivery and engagement with breastfeeding support interventions; and (3) reflecting and evaluating the success of implementing and providing breastfeeding support.

Included studies identified several implementation challenges relating to the needs, preferences and priorities of those delivering and receiving breastfeeding support interventions. Breastfeeding supporter training was a commonly reported implementation strategy, which also enabled implementation teams to identify and address breastfeeding supporters’ needs. Included studies reported a range of contextual factors (e.g. alignment with local policies) affecting the implementation and delivery of breastfeeding support interventions as well as a range of tailoring strategies (e.g. community involvement, use of lay language, responsive support content/information) to address contextual factors. Reports about implementation success focused on issues relating to key implementation outcomes such as satisfaction, fidelity and usefulness.

Overview

Previous chapters have identified which support interventions were effective in terms of stopping the drop-off of women breastfeeding, and what contextual factors need to be considered when implementing interventions into healthcare settings in the UK. This chapter builds on this evidence by exploring how well breastfeeding support interventions work in relation to how much they cost health services. A systematic review of economic evidence was conducted to appraise and synthesise what was already known about the cost-effectiveness of breastfeeding support interventions for healthy mothers with healthy babies. This was followed by a model-based economic evaluation, which was informed by the systematic reviews of effect and of cost-effectiveness. The health economic component of the evidence syntheses was designed and interpreted with input and advice from the stakeholder engagement groups, workshops and the study steering committee.

Systematic review of economic evidence

The aim of this review of economic evidence was to gain an understanding of whether breastfeeding support interventions for healthy mothers with healthy babies were considered value for money. The overarching review question was: What are the incremental costs and cost-effectiveness of breastfeeding support interventions in comparison with standard care, no intervention, or an alternative intervention for healthy mothers with healthy babies in the UK? The review objectives were to:

1. identify and synthesise the evidence base for incremental costs and cost-effectiveness of breastfeeding support interventions

2. assess the applicability of the evidence to a UK setting

3. identify limitations and uncertainties in the applicable economic evaluations

4. examine the level of consistency between applicable economic evaluations.

Methods

Results

Study selection

Following engagement with stakeholders, as reported in Chapter 2, agreement was reached that all breastfeeding support interventions identified as effective were deemed suitable and transferable to a UK setting. Justification for this was based on the consideration that if an intervention was effective and resources were available, then implementation should be supported to adapt services to deliver the intervention. For this review of economic evidence, consideration also needed to be given to whether the system and context of the setting were similar to those of the UK. Subsequently, while no consideration was given to country setting for inclusion, only those studies conducted in Organisation for Economic Cooperation and Development (OECD) settings were assessed for applicability, and only those judged to be directly or partially applicable were assessed for limitations. 111

Figure 4 presents the PRISMA flow diagram for the study selection process. Following the removal of duplicate records, 5699 records were screened at the title and abstract stage. Of these, 5491 were excluded and the full text of 208 records was sought. Nine records could not be retrieved: three were ongoing studies still in the recruitment phase of the aligned RCT, three had no relevant data available and three were awaiting classification with no response from the corresponding authors. Of the 199 records screened for eligibility, 162 were excluded. The main reason for exclusion was the wrong design (n = 116), as on full-text review many studies did not report an economic evaluation. Further reasons for exclusion were the wrong intervention (n = 28), wrong population (n = 15) and wrong outcomes (n = 1). The systematic search, identification and screening process resulted in 39 studies eligible for inclusion.

FIGURE 4.

The PRISMA flow diagram for review of economic evidence for breastfeeding support interventions for healthy mothers with healthy babies.

Chapter summary

Thirty-nine studies were identified that conducted a partial or full economic evaluation of a breastfeeding support intervention for healthy women compared with a control. Nine of these studies were judged to be applicable or partially applicable to the UK setting. Of these, four assessed the cost-effectiveness of ‘breastfeeding only’ support and five assessed the cost-effectiveness of ‘breastfeeding plus’ support.

For 'breastfeeding only’ support, there was limited evidence that interventions were cost-effective. One model-based cost–utility analysis estimated that a hypothetical intervention providing six contacts with a health professional or lay person, starting in the antenatal period and continuing in the early postnatal period, was considered unlikely to be cost-effective in terms of cost per QALY gained (£56,075 per QALY gained). There was limited evidence for the incremental cost per additional woman breastfeeding (any or exclusive), with estimates from cost-effectiveness analyses ranging from £67 at 5–12 days to £2446 at 6 months postpartum. Without WTP thresholds, whether the interventions are cost-effective is unclear. Evidence for breastfeeding plus support was reported in two studies that modelled cost-effectiveness in terms of cost per QALY gained. Both studies identified the interventions as not value for money.

We judged that there was uncertainty in the findings of cost-effectiveness due to the limited number of studies and the lack of good-quality evidence. Limitations of the evaluations centred on a short time horizon, with seven out of nine studies not extrapolating beyond the time frame of the underlying effectiveness study, and a limit to the scope of costs and benefits measured. Five of the nine studies costed the intervention only and did not record the health service resource use of the mother or infant. These limitations suggest uncertainty in the findings. There appeared to be consistency between studies evaluating ‘breastfeeding only’ support in terms of cost per additional woman breastfeeding (any or exclusive). There was less consistency observed between studies assessing ‘breastfeeding plus’ interventions. These inconsistencies may be due to the different time horizons, differing scope of costs and benefits measured and valued and different outcomes of cost-effectiveness estimated, which make comparison difficult.

Introduction

Women with LTCs face additional challenges in breastfeeding. The Cochrane review on breastfeeding support for healthy women with healthy term babies by its nature excludes women with LTCs. By receiving additional study funding, we were able to conduct an additional piece of work that looked at the effectiveness of breastfeeding support for women with LTCs.

Aim and objectives

The aim of this systematic review was to identify the effectiveness of breastfeeding support interventions in women with LTCs.

The objectives were to:

1. identify breastfeeding support interventions that have been designed for women with LTCs

2. describe the characteristics of breastfeeding support interventions –

   1. provider

   2. intensity of support

   3. type of support (e.g. face to face, telephone, digital technologies, group or individual support, proactive or reactive)

   4. additional intervention components (i.e. wider child and maternal healthcare)

   5. timing of support (antenatal, postnatal)

3. determine the effectiveness of breastfeeding support interventions for women with LTCs.

Methods

This systematic review followed the methods for systematic reviews of interventions outlined in the Cochrane Handbook. 87 The protocol is registered on PROSPERO (CRD42022337239).

Study selection

We imported all records identified via electronic databases into Covidence, a web-based collaboration software platform that streamlines the production of systematic and other literature reviews. 84 The title and abstract of each record were double-screened by two reviewers (AG, LH, SS, AMcF, FL, PB or FXV). If the two reviewers disagreed, consensus was reached via discussion by AG and LH. The same process was followed for full-text screening. The results of this selection process are shown in a PRISMA flow chart (see Figure 5).

FIGURE 5.

The PRISMA flow diagram illustrating study selection.

Results

Strengths and limitations

We followed systematic review methods outlined in the Cochrane Handbook;87 however, there is the potential that bias was introduced into the review. First, because of resource constraints we were able to include only studies published in English, so there is a risk of language bias. Second, although we attempted to identify all published and unpublished trials on breastfeeding support for women with LTCs, it is possible that not all existing trials were identified. Funnel plot analyses suggested some possible asymmetry; however, interpretation is limited by the small number of studies. Third, we were unable to adjust for clustering in one of the studies; however, a sensitivity analysis from which that study was removed did not change the effect estimate. Fourth, there was considerable variability in how breastfeeding initiation was measured (e.g. within 24 hours vs. ever), so a post hoc decision was made to exclude this outcome from the meta-analysis. Finally, there is heterogeneity between the studies, which may be a result of differences between interventions and population characteristics, in particular the LTCs.

Chapter summary

Twenty-two studies were identified that examined the effectiveness of breastfeeding support for women with single LTCs. Of these, 20 contributed data to the review. No studies were identified that included women specifically with MLTC.

The most common conditions were overweight and obesity, with nine studies focused on this. A further three studies were for women with GDM. Five studies included women with HIV. Two studies were for women with substance misuse problems and only one was for women with anxiety and depression. Interventions varied in terms of whether they only provided breastfeeding support or if they also provided support for the LTC. The majority of studies had an antenatal component.

We performed meta-analysis for all the primary and additional breastfeeding outcomes. There was little to no difference between intervention and controls for any of these. We judged these outcomes to be low and moderate certainty. When we used a sensitivity analysis to exclude interventions for women with HIV, there was no meaningful change in the effect estimates. We considered the overall risk of bias in the included trials to be mixed. Sensitivity analyses excluding studies rated as being at high or unclear risk of bias for allocation concealment and attrition also did not alter the effect estimates.

Fifteen studies measured secondary non-breastfeeding outcomes, which included infant weight, infant health, maternal weight and health behaviours, satisfaction with care and satisfaction with feeding method. Because of the heterogeneity in outcomes, meta-analysis was not possible and so results were reported narratively. There was little evidence of any beneficial intervention effect on any of the secondary outcomes measured.

To conclude, this review identified that the breastfeeding support interventions for women with LTCs probably had little to no effect on breastfeeding outcomes. There is, therefore, a need for further research to develop breastfeeding support interventions for women with LTCs.

Introduction

As part of our additional funding for MLTC, we sought to complement the evidence on effectiveness from review 4 (see Chapter 6) by undertaking a mixed-methods review looking at what is known about the views and experiences of breastfeeding support in women with LTCs.

Objectives

1. To identify and synthesise the views and experiences of those involved in delivering and receiving breastfeeding support for women with LTCs.

2. To identify the contextual factors (barriers/facilitators) affecting the implementation of breastfeeding support for women with LTCs.

Methods

The protocol for this systematic review is registered on PROSPERO (CRD42022374509).

Results

The searches identified 5058 records, which were assessed against the inclusion criteria. Title and abstract screening resulted in 119 records considered eligible or inconclusive. Full-text articles were then retrieved and assessed for eligibility. Three records could not be retrieved. Of the 116 records screened at full text, 92 were excluded. The main reason for exclusion was not reporting views and experiences of breastfeeding support (e.g. views and experiences of breastfeeding) (n = 37), followed by involving study designs not eligible for inclusion in this review (e.g. effectiveness studies) (n = 22). Other reasons for exclusion were abstract-only records (e.g. conference proceedings) (n = 19), focusing on ineligible populations (n = 8), not reporting on views and experiences (n = 5), and language of publication not being English (n = 1). The remaining 24 studies were included in the final synthesis (see Figure 6).

FIGURE 6.

The PRISMA flow diagram.

Chapter summary

This review comprised 24 studies reporting primary research on the views and experiences of breastfeeding women with LTCs and/or support providers.

The health conditions covered were HIV-positive, obesity and overweight, substance use, diabetes in pregnancy, disabilities and a rare genetic disorder. The overall quality of included studies was mixed, with some studies rated as weak and/or with poor reporting.

Four key themes were identified: (1) additional breastfeeding support needs for women with LTCs; (2) availability of breastfeeding support for mothers with LTCs; (3) the role and practice of breastfeeding support for mothers with LTCs; and (4) suggested strategies to improve breastfeeding support for mothers with LTCs.

Included studies highlighted a range of additional support needs for women with LTCs, such as issues relating to treatments or medical interventions for women’s/infant’s health conditions, misconceptions, misinformation or emotional distress. Studies reported variable or insufficient availability of breastfeeding support for mothers with LTCs, particularly when support was needed across multiple healthcare settings beyond maternity care. The data suggest complex breastfeeding journeys involving a wide range of interactions, individuals, settings and factors that could impact women’s experiences.

Overview

The previous two chapters reported a systematic review identifying (1) which interventions were effective in providing breastfeeding support for women with single LTCs, and (2) the barriers to and facilitators of breastfeeding support to women with LTCs. This chapter builds on this evidence by assessing how well breastfeeding support interventions for women with LTCs work in relation to how much they cost health services. As evidence was expected to be limited, a systematic review of economic evidence was planned to appraise and synthesise what is known about the cost-effectiveness of breastfeeding support interventions for mothers with LTCs.

Aim and objectives

The aim of this review of economic evidence was to gain an understanding of whether breastfeeding support interventions for mothers with LTCs were considered value for money. The overarching review question was: What are the incremental costs and cost-effectiveness of breastfeeding support interventions in comparison to standard care, no intervention, or an alternative intervention for mothers with LTCs? The review objectives were to:

1. identify and synthesise the evidence base for incremental costs and cost-effectiveness of breastfeeding support interventions

2. assess the applicability of the evidence to a UK setting

3. identify limitations of and uncertainties in the applicable economic evaluations

4. examine the level of consistency between applicable economic evaluations.

Methods

Results

Study selection

Figure 7 presents the PRISMA flow diagram for the study selection process. Following the removal of duplicate records, 5732 records were screened at the title and abstract stage. Of these, 5713 were excluded and the full text of 19 records were sought. One record, which was an ongoing study, could not be retrieved (Jacobson, 2020). Of the 18 records screened at full text, 13 were excluded. The main reason for exclusion was the wrong study design (n = 7), followed by wrong population (n = 4) and wrong intervention (n = 2). The systematic search, identification and screening process resulted in five studies eligible for inclusion.

FIGURE 7.

The PRISMA 2020 flow diagram for review of economic evidence for breastfeeding support interventions for women with LTCs.

Strengths and limitations

To the best of our knowledge, this is the only systematic review of economic evidence on breastfeeding support interventions for women with LTCs, and it has identified a lack of evidence on incremental cost and incremental cost-effectiveness that is applicable to a UK setting.

We followed methods recommended for identifying, assessing and reviewing economic evidence;87,110,112,220 however, there is a potential for bias. While we attempted to identify all published and unpublished economic evaluations on breastfeeding support for women with LTCs, it is possible that not all studies were identified.

Chapter summary

Five studies were identified that examined the incremental cost and/or cost-effectiveness of breastfeeding support interventions for women with LTCs compared with a control or provided a cost–outcome description. The conditions assessed in the studies were HIV, obesity, prenatal opioid use and medically high risk (maternal hypertension and diabetes prior to birth). Interventions provided only breastfeeding support or also provided support for the LTC or provided care across the continuum. Each of the interventions assessed in the full economic evaluations was deemed cost-effective for the base case. On appraisal, none of the studies was judged to be applicable to the system and context of the UK.

Aims

The final stage of the research aimed to develop and refine a toolkit for the implementation and evaluation of effective breastfeeding interventions relevant to the UK, based on all evidence and stakeholder input from the previous work. An additional aim was to elicit stakeholders’ preferences in terms of WTP for a breastfeeding support intervention. This stage of the research included both the main study and additional work for women with LTCs. See Appendix 8 for the draft toolkit.

Methods

The co-creation of the toolkit built on the findings of the evidence syntheses and stakeholder engagement, as described in the previous chapters, as follows:

• effective breastfeeding support interventions for healthy women with healthy term babies from the updated Cochrane review83

• barriers to and enablers of implementing breastfeeding support derived from synthesising process evaluations of effective interventions (see Chapter 4)

• barriers to implementation and strategies to overcome them derived from the main study stakeholder engagement (see Chapter 2)

• key challenges for women with multimorbidities when accessing support for breastfeeding and for healthcare providers in offering support derived from the MLTC stakeholder engagement (see Chapter 2).

The next stage of developing the toolkit involved a wider group of stakeholders via co-creation workshops. The workshop activities revolved around a prototype breastfeeding support intervention drawn from elements of interventions from the Cochrane review. 83 The interventions that informed the prototype were selected as they were effective in reducing the number of women stopping breastfeeding and were judged to be at low risk of bias using allocation concealment as a proxy for this. This prototype is a composite of the characteristics of these seven interventions and together they provide a range of the ways breastfeeding support could effectively be implemented. 103,221–226

Prototype intervention

The breastfeeding support intervention will be delivered one-to-one by infant-feeding advisors. It consists of one 30-minute antenatal appointment, one 30-minute postnatal visit in hospital, one 30-minute home visit within 48 hours of discharge and regular telephone calls. The antenatal session will focus on rapport building, education and identifying any concerns regarding breastfeeding. The hospital and discharge visits will involve checking latch, helping with positioning and observing a feed if requested by the mother. Infant-feeding advisors will also provide encouragement and reassurance during visits. Women will be given the chance to ask questions and raise any concerns.

Following the initial three contacts, support will be provided remotely unless a face-to-face visit is required. For the first 4 weeks there will be a weekly proactive telephone call, and beyond that support will be provided monthly until 3 months or when breastfeeding ceases. Women can also contact infant-feeding advisors as needed via telephone or SMS during this 3-month period and beyond as new issues arise.

The infant-feeding advisor will also signpost women to the local breastfeeding peer support group, which provides support via WhatsApp and weekly face-to-face support groups and/or one-to-one peer support service. Infant-feeding advisors will receive training on the intervention delivery.

Workshops

Four 1-day workshops were held in November 2022 in Belfast, Birmingham, Cardiff and Edinburgh, representing the four nations of the UK. We aimed to include up to 30 participants in each workshop representing four key groups: (1) service users and their representatives, including third-sector advocacy organisations and lay/peer supporters; (2) health services including frontline practitioners (e.g. midwives, health visitors, doctors, lactation consultants, support workers), and service managers and commissioners; (3) national and local policy-makers, including government bodies, and public health and social care organisations; and (4) academic researchers. Invitations were disseminated via the research team’s networks, members of the stakeholder working groups and third-sector organisations with a focus on participants who represent or work with communities where breastfeeding rates are low to maintain the focus on inequalities.

Workshop activities

Following an overview of the main study and the additional work for women with LTCs, and an explanation of the prototype intervention, participants worked in small groups of six to eight people on four activities. Each group comprised participants from the four main groups of attendees as described above, and a member of the research team to facilitate and document key discussion points. Next was a description of each of the activities along with a summary of the key findings based on a synthesis from all four workshops. Throughout all the activities, participants were asked to focus on women from communities with low breastfeeding rates.

Activity 1: adaptation of the prototype intervention for women with multimorbidities

Participants were presented with a hypothetical case study of a woman with several LTCs (fibromyalgia, Crohn’s disease and anxiety), which was drawn from the experiences of members of the MLTC parents’ panel. Participants were asked to discuss what, if any, adaptations to the prototype intervention would be needed to meet the needs of breastfeeding women with multimorbidities.

The consensus across the workshops was that the intervention needed significant modifications. The greatest consensus was on three modifications: (1) the antenatal appointment should be longer than 30 minutes; (2) continuity, with the same person delivering the intervention antenatally and postnatally so that women do not have to repeat their stories; and (3) infant-feeding advisors should be included in joint obstetric and medical clinics.

Other modifications mentioned frequently were:

• The person delivering the intervention should have expertise in medications and breastfeeding, as well as in breastfeeding support.

• Antenatal appointments of 90 minutes would be more realistic, or several shorter appointments could be helpful.

• Starting discussions early in pregnancy could be beneficial to take account of the higher risk of preterm birth in women with multimorbidities and to give practitioners more time to find accurate information.

• Women require a medication review in early pregnancy, and this should involve a pharmacist who is knowledgeable about medications and breastfeeding.

• Women should be able to see all of their healthcare providers (e.g. midwife, obstetrician, physician, pharmacist) during one appointment to minimise women’s time, effort and costs. Ideally, the appointment would include key members of the women’s support network (e.g. partner, family).

• The antenatal appointment should focus on practical tips for managing varying levels of fatigue and pain, such as how to find comfortable feeding positions. Content should also be flexible to the women’s needs, adaptable to changing circumstances and consistent across different healthcare providers.

• 30-minute postnatal appointments are too short.

• For the 3-month follow-up support, women should have the option of telephone or face-to-face contacts, and 24-hour telephone support should be available.

• Peer support could be offered antenatally, and group antenatal peer support could help normalise breastfeeding for women with LTCs. Women could be offered the choice of one-to-one or group peer support.

• Third-sector organisations could help with the provision of breastfeeding and emotional support.

• To be sustainable, peer supporters should be paid.

• Training is needed to increase knowledge of breastfeeding and multimorbidities in the multidisciplinary team, including GPs. Supporting women with multimorbidities to breastfeed should be included in routine breastfeeding training updates.

• Services should be co-ordinated, with the infant-feeding advisor as the key point of contact for the multidisciplinary team.

Activity 2: identified barriers to implementation of prototype intervention for healthy women and women with multimorbidities

Participants were asked within their groups to discuss and list barriers to implementing and, for parents, accessing the prototype intervention in their settings. Open discussion was encouraged; however, facilitators were provided with a prompt sheet comprising the domains and constructs adapted from the CFIR227 to stimulate consideration of all aspects of implementation and accessibility. The lists of barriers were collated and, along with the 18 barriers identified by the stakeholder working group and parents’ panel (see Chapter 2), mapped to the updated CFIR. 92 There was a high degree of overlap within and across the workshops and between the workshops and stakeholder and parents’ panel discussions. We present the main themes under each domain of the CFIR,92 while acknowledging that there is overlap between constructs within a domain and between domains. Constructs of the CFIR are denoted in italics in the following text.

Innovation domain: Barriers relating to the innovation, defined as the ‘thing being implemented’,92 mapped mainly to the constructs of adaptability, complexity, design and cost. The most frequently mentioned barrier referred to adaptability in that the schedule and length of appointments lacked flexibility and would need to be tailored to individual women’s needs and circumstances. The next most frequently mentioned barrier was that the design of the intervention did not include the women’s partner and/or other family members who could be important sources of breastfeeding support. Further frequently mentioned concerns with the intervention design were lack of continuity across the intervention and lack of intensity in the first 2 weeks postnatally. Barriers related to cost highlighted concerns that costs to the service could be high and may not represent value for money or be sustainable. Regarding complexity, the intervention was perceived to be multifaceted and to necessitate multiple appointments that may not be convenient for women. The stakeholder working group identified that the intervention may not be perceived to offer relative advantage compared with existing or alternative approaches to breastfeeding support.

Outer setting domain: Barriers related to local attitudes to breastfeeding were discussed by all groups across the four workshops and were one of the most frequently mentioned of all barriers across all domains. Typically, barriers were phrased as ‘negative societal attitudes to breastfeeding’ or the existence of a ‘bottle-feeding culture’. These were said to result in family or peer pressure for women to formula feed based on unhelpful beliefs. Linked to this were external societal pressures, including the impact of social media influencers and formula company marketing. Lack of political priority and/or strategy for breastfeeding and failure to monitor and enforce the International Code of Marketing of Breastmilk Substitutes were common themes that mapped to policies and laws. A further frequently mentioned barrier related to the challenges of developing partnerships and connections between health services and other sectors such as third-sector organisations or local authorities. Outer setting barriers are also related to local conditions, for example lack of good transport and/or childcare, digital poverty and the current cost-of-living crisis. Lack of financing for breastfeeding along with funding targets was mentioned, but it was often unclear when this referred to the outer (funding from external entities) or the inner setting (funding to implement and deliver the innovation). Finally, the impact of COVID-19-related restrictions, particularly on group settings, was seen to be a barrier reflecting critical incidents.

Inner setting domain: There were twice as many barriers identified under the inner setting domain as under any of the other domains. The most frequent themes are linked to work infrastructure, culture and available resources. Workforce challenges such as staff shortages, high turnover of staff and lack of time/protected time were the most frequently mentioned. Other barriers related to work infrastructure included a lack of the right skill mix and overdependency on one or a small group of individuals. Overlapping with work infrastructure were barriers relating to relational connections and communications, for example poor communication and working practices across the multidisciplinary team, fragmented services and challenges to embracing peer support within the health settings. The last of these included peer support not being valued and a reliance on unpaid volunteers. Regarding culture, a very frequent theme (linked to human equality-centredness and recipient-centredness) was barriers relating to the lack of accessibility of services to diverse populations, including lack of language support, sensitivity to women’s backgrounds and stereotyping, as well as the cost of the intervention (e.g. travel costs) to women who have little resource. Also linked to culture were issues of learning-centredness, such as lack of visibility of data to staff (e.g. breastfeeding rates), lack of data sharing and lack of sharing of good practice. Regarding available resources, the most frequently mentioned barriers were lack of funding and lack of space, such as appropriate venues to deliver the intervention with consideration of space for women to breastfeed and accessible locations for groups to meet. Other themes were lack of compatibility of the innovation with existing policies and guidelines or with the practice of early postnatal discharge. Workshop participants and the stakeholder working group identified that the innovation overlapped with current provision and may not fit with existing workflows or system values.

Individuals domain: The most common theme in this domain is mapped to the capability (knowledge, skills, interpersonal competence) of innovation deliverers, resulting in conflicting information for breastfeeding women. The main concern was a lack of experience and training of many of the staff who would be delivering the intervention. This included a lack of access to high-quality education. A frequently mentioned barrier was some staff’s negative attitudes to breastfeeding, which could impact on their interactions with women. Second to the capability of staff was that some staff lacked motivation either because they did not value breastfeeding or due to professional fatigue. Lack of confidence of staff to implement the innovation was identified as a barrier by the stakeholder working group and at two workshops. The second most common theme of barriers related to the buy-in, understanding and valuation (capability and motivation) of the innovation by high- and mid-level leaders, that is key strategic decision-makers and those whose remit is to operationalise strategic decisions, without whose support the implementation would be unlikely to succeed. The stakeholder working group identified a lack of champions and skilled implementation leads as a further barrier. A final theme under individuals related to innovation recipients with barriers to opportunity such as lack of time and lack of knowledge of or access to services.

Implementation process domain: At the workshops, fewer barriers were linked to this domain than to the other domains. The only barriers mentioned by more than one group related to engaging, for example staff lack of engagement or resistance to change, and planning in the lack of management oversight to ensure that the innovation is being implemented as intended. The stakeholder working group identified concerns regarding the lack of feedback to staff to evaluate the quality of the intervention (reflecting and evaluating), the need to assess accurately the needs of parents and families (assessing needs) and poor communication of the goals, policies and procedures related to the innovation (planning).

Activity 3: prioritised strategies to overcome implementation barriers

In this activity, participants were presented with the 34 implementation strategies adapted from the Expert Recommendations for Implementing Change framework,82 derived from the stakeholder and parent consensus-building exercise (see Chapter 2). The task was to select the most relevant strategies to overcome each of the barriers identified in activity 2. Participants selected multiple strategies for each barrier, and each strategy multiple times. Given that so many barriers were related to the inner setting domain and were therefore context driven, we here present those strategies that were most frequently selected, giving examples of the barriers that they might address. Participants were also invited to add any additional strategies they thought were missing from the those provided. A full list of strategies and the number of times each was selected, along with additional strategies suggested by participants, can be found in Report Supplementary Material 1, Table 9 presents the five strategies chosen most frequently, along with examples of the barriers these were selected to overcome.

TABLE 9 - Most frequently selected strategies with examples of barriers

Strategy	Number of times selected	Examples of barriers
Deliver realistic, evidence-based information in multiple formats on how to deliver the breastfeeding support intervention and why it is important	84	Lack of staff training, knowledge and skills Lack of consistency of information Lack of continuity of care Challenges to accessing the intervention for women and families Lack of buy-in from senior managers
Assign a key practitioner to raise awareness about the intervention to ensure a consistent message	75	Challenges to working with sectors outside the health system Poor communication across the multidisciplinary team Lack of joined-up vision and working
New or existing funding for breastfeeding support should be a general health investment for local councils and the government, and not just the NHS	72	Lack of funding within the health system Cost of the service to the NHS Lack of relationship between the health system and the community Lack of sustainability Cost of the intervention to women Reliance on non-paid peer supporters
Create an infant-feeding team in every NHS organisation to lead the intervention, working collaboratively with multidisciplinary practitioners and lay supporters	72	Lack of availability of good-quality training Time and capacity issues Professional boundaries – especially working with peer supporters Lack of confidence of those delivering the intervention Lack of integration across the continuum (antenatal/postnatal) and across the multidisciplinary team
Revise roles as needed to support the intervention; for example, integrate peer supporters with NHS infant-feeding teams, and consider upskilling maternity staff to specialist lactation training levels	70	Barriers to integrating peer support with health services, including lack of valuing peer support Lack of right skill mix Lack of knowledge and skills of staff delivering the intervention Infant-feeding specialists overloaded

While Table 9 shows the most frequently selected strategies across all four workshops, there were differences between the workshops. For example, the two most frequently selected strategies at the Edinburgh workshop did not feature in the top five strategies across the workshop. They were:

1. start with pilots (in Baby-Friendly Initiative and non-Baby-Friendly Initiative accredited settings) to refine implementation and resources required as a means of phasing in the intervention and change in a sustainable way (#6 in the overall strategy ranking)

2. use new survey and routine data to assess the impact and monitor the quality of the breastfeeding support intervention (#12 in the overall strategy ranking).

The second most frequently selected strategy at the Cardiff workshop was:

• involve parents, peer supporters and charities in adapting the intervention for the local area and to encourage uptake (#10 in the overall strategy ranking).

The differences between the workshops can most likely be explained by a combination of the different balance of participants at each workshop (with more parents and third-sector representatives at the Edinburgh workshop) and the different policy contexts of the four nations.

Activity 4: considerations for evaluating breastfeeding support interventions

Participants discussed how the prototype intervention could be evaluated and were prompted to consider outcomes that are important to parents, the timing of breastfeeding outcome data collection, the important data related to processes, and how to assess the impact on health inequalities.

Important outcomes for parents were suggested to be meeting their feeding goals and expectations, whether the support and information was helpful, and how confident or empowered a woman felt after the intervention.

With regard to the timing of breastfeeding outcomes data collection, the most frequently mentioned was to collect data on ‘any’ and ‘exclusive’ breastfeeding at the following time points:

• first feed within 1 hour after birth

• discharge from hospital

• 6–8 weeks

• 6 months.

Other suggestions with high consensus were 10–12 days (to coincide with discharge from routine midwifery care), 3–4 months and 1 year. Other comments on collecting breastfeeding outcome related to definitions of any and exclusive and whether these needed to be subdivided further.

Other outcomes felt to be important included health outcomes, for example the number of infants admitted to hospital and the reasons for stopping breastfeeding.

Process data

The most frequently mentioned were the views and experiences of those receiving and delivering the intervention (including women, healthcare practitioners and peer supporters), women’s satisfaction, and intervention fidelity (did women receive all components of the intervention). There was discussion that data could be collected early to capture those who cease to engage with the intervention and to gain feedback from those who declined the intervention. Many methods for collecting data were suggested, including digital options such as WhatsApp, and there was high consensus that participants in studies should be offered options for follow-up, for example between online, telephone, e-mail, post or a phone app.

Impact on inequalities

Discussions about evaluating the impact on health inequalities centred around gathering background information such as maternal characteristics (age, ethnicity, socioeconomic status) and making sure that the intervention and evaluation are inclusive, for example by addressing language barriers.

Activity 5: willingness to pay for a breastfeeding support intervention

To evaluate stakeholders’ preferences for a breastfeeding support intervention, participants in the workshop were presented with a stated preference discrete choice experiment (DCE). A DCE is a method of eliciting preferences for a given product or service by presenting a series of scenarios to individuals; each scenario presents two or more alternatives that differ in the attributes of the product/service, and the individual chooses their preferred alternative. 228 The theoretical underpinnings of the experiment are derived from (1) random utility maximisation,229 where it is assumed that individuals’ choice behaviours are made to maximise their satisfaction while allowing for unobserved sources of utility; and (2) Lancaster’s economic theory of value, which posits that an individual’s utility for a whole product or service can be separated into utilities for each component or attribute of that service. 230 If a change thus occurs in one of the attributes of the service, the individual may choose an alternative product if they deem it of greater value, while acting to minimise cost. 231 DCEs have been used increasingly over the last 20 years in health-related research and are useful for informing health policy, providing preferences for clinical outcomes of a service, as well as the process and cost attributes. 232 The aim of the experiment presented during the workshops was to estimate the value of a breastfeeding support intervention to participants, as well as the relative importance of each attribute and attribute level of the intervention.

Guidance on constructing the experimental design for DCEs was followed. 233,234 Careful consideration was given to the selection of attributes and suitable levels to be presented within the DCE. While DCEs present participants with hypothetical scenarios to choose from, it is important that the scenarios reflect practice and are recognisable to participants to ensure that the exercise is capable of deriving preferences. 228 The attributes (n = 7) and attribute levels (range 3–5) that were used to create the alternative choices presented in each scenario are outlined in Table 10.

TABLE 10 - Attributes and levels used to elicit preferences for an additional breastfeeding support intervention

Attributes	Attribute levels
	1	2	3	4	5
Number of contacts	≤3	4–8	≥9	–	–
Provider	Peer supporter	Breastfeeding counsellor	Health professional	Lactation consultant	Combined provision
Mode of support	Telephone	Face to face	Online	Hybrid	–
Approach to support	Reactive	Proactive	Blended	–	–
Reduction in drop-off for any breastfeeding at 6 weeks	1%	5%	10%	15%	–
Reduction in drop-off for exclusive breastfeeding at 6 weeks	No reduction	1%	5%	10%	–
Additional cost per woman	£25	£50	£100	£150	–

The attributes and levels were informed by the findings from the systematic reviews reported in Chapters 3 and 5, the findings from the stakeholder engagement, which comprised online discussions, the modified Delphi study and face-to-face focus groups, and the resulting prototype intervention. The intervention components included process attributes of the number of contacts between service users and service providers, provider of the intervention, mode of support and approach to support. The clinical outcome attributes were the percentage reduction in drop-off for any, or exclusive, breastfeeding at 6 weeks. Only one of the two clinical outcome attributes was presented in any given experiment to each participant. Finally, the cost attribute indicated the additional cost to the NHS per woman supported.

A fractional factorial design was then used to create the experiment to limit participant fatigue and the length of time required to complete it. An orthogonal main effects plan, using Statistical Package for Social Sciences version 27 (IBM Corporation, Armonk, NY, USA) software, generated profiles for the alternatives and 12 choice sets. Participants were presented with an unlabelled DCE with two alternative intervention options (A and B), which differed in their attribute levels, along with a third alternative of choosing neither intervention. This third alternative provided an unconditional choice set where participants could opt out if they preferred. Figure 8 illustrates an example scenario designed to enable participants to trade across attributes and, thus, identify the relative value of each attribute and level for stakeholders.

FIGURE 8.

Example scenario presented to workshop participants.

An interview-based format was used to administer the experiment to workshop participants, allowing the facilitator to answer any queries and clarify any issues. Before the activity commenced, the DCE was explained, and participants were introduced to each attribute and associated levels. They were informed that the breastfeeding support intervention was additional to current service provision and that several outcomes of effect may occur as a result of the additional support, such as a change in maternal satisfaction with care or a change in breastfeeding initiation rates. However, for the purposes of the exercise they were asked to consider a reduction in drop-off for breastfeeding (any or exclusive) at 6 weeks, which reflected the outcome of effect in the Cochrane review that had recently been updated as part of the study. 83

Data from the experiment were entered into Microsoft Excel. Data entry was carried out using a multiple-line format, whereby data are divided into a number of blocks. Effects coding was used for the levels of the process attributes, while the clinical outcomes and cost attributes were maintained. Each block represented a participant’s choice set and each row within that block corresponded to an alternative within the choice set, effectively clustering the data to allow for multiple observations from respondents to the experiment. The choice outcome was the variable that signified the decision made for each scenario and, as such, was the dependent variable within the model. The discrete choice analysis was undertaken using a random utility model and conducted in R (The R Foundation for Statistical Computing, Vienna, Austria) using guidance provided by Croissant. 235 Modelling the choice sets of participants produced choice probability estimates and an indirect utility function for choosing an alternative, an attribute and an attribute level. Estimated marginal rates of substitution enabled the interpretation of participants’ WTP for each attribute and attribute level.

Results

A total of 87 workshop participants completed the DCE in November 2022. Table 11 presents the results from the discrete choice modelling.

TABLE 11 - Results of the modelling preferences derived from the DCE

*p < 0.05; **p<0.01.

Reference levels for effects-coded attributes were calculated as the negative sum of the estimated attribute levels.

Attribute and attribute level	Βeta coefficient	Standard error
Number of contacts
≥9	0.03	0.142
4–8	0.35**	0.122
≤ 3a	−0.38
Provider
Combined provision	0.46**	0.172
Lactation consultant	0.03	0.165
Breastfeeding counsellor	0.18	0.202
Peer supporter	−0.45*	0.196
Healthcare professionala	−0.22
Mode of support
Hybrid	−0.30	0.187
Online	0.11	0.187
Telephone	−0.29*	0.135
Face to facea	0.48
Approach to support
Blended	−0.15	0.148
Proactive	0.16	0.117
Reactivea	−0.01
Reduction in drop-off for any breastfeeding at 6 weeks	0.26**	0.018
Reduction in drop-off for exclusive breastfeeding at 6 weeks	0.52**	0.070
Additional cost per woman	−0.02**	0.002
Neither intervention	−1.16**	0.186
Log-likelihood	−748.03
Number of iterations	5

With regard to the estimated beta coefficients, preference formation was as expected a priori and resonated with the findings from the stakeholder engagement activities and the resulting prototype intervention. Stakeholders exhibited statistically significant preference for four to eight contacts over three or fewer (β = 0.35, SE 0.122, p < 0.01) and provision from a range of providers over healthcare professional alone (β = 0.46, SE 0.172, p < 0.01), and valued face-to-face support over telephone support (β = −0.29, SE 0.135, p < 0.05). Although there was a positive value for a proactive approach to support over reactive support, this was not statistically significant (β = 0.16, SE 0.117, p > 0.05), suggesting that stakeholders did not consider the different approaches to support (reactive, proactive, hybrid) in their decision-making process. Both clinical outcome attributes of reducing the number of women stopping any breastfeeding (β = 0.26, SE 0.018, p < 0.01) or exclusive breastfeeding (β = 0.52, SE 0.070, p < 0.01) at 6 weeks postpartum were statistically significant, suggesting that the greater the percentage reduction in drop-off, the greater the value to participants. For the additional cost per woman, participants valued a lower cost intervention over a higher cost (β = −0.02, SE 0.002, p < 0.01), upholding underlying assumptions of individuals acting to minimise cost. 231 The overall preference by stakeholders for introducing an additional breastfeeding support intervention into practice was reiterated by the lack of preference for the status-quo alternative, which displayed a negative beta coefficient (β = −1.16, SE 0.186, p < 0.01).

In terms of WTP for additional breastfeeding support, estimated marginal rates of substitution indicated that participants were willing to pay £67.40 per woman for additional breastfeeding support, regardless of how it was delivered or whether it was effective in reducing the number of women stopping breastfeeding at 6 weeks postpartum. Table 12 presents the WTP for each clinical outcome attribute and each process attribute level valued by participants, which was represented by a statistically significant, positive beta coefficient in the model.

TABLE 12 - Participants’ marginal rates of substitution between cost of additional breastfeeding support and intervention attributes

Reference level.

Attribute valued	Marginal WTP/woman
4–8 contacts	£20.29
Combined provision	£26.83
Face-to-face supporta	£27.89
For each 1% reduction in drop-off for any breastfeeding at 6 weeks	£14.90
For each 1% reduction in drop-off for exclusive breastfeeding at 6 weeks	£30.03

As an example, the estimated WTP by stakeholders was £89.91 per woman for a breastfeeding support intervention that realised a 1% reduction in drop-off for any breastfeeding at 6 weeks postpartum, and £105.04 per woman for an intervention that realised a 1% reduction in drop-off for exclusive breastfeeding at 6 weeks postpartum. The WTP thresholds would increase to £149.51 and £225.16 if the interventions realised a 5% reduction in drop-off of any or exclusive breastfeeding at 6 weeks postpartum, respectively.

Summary of findings

The aim of this study was to synthesise global and UK evidence to co-create with stakeholders a framework to guide the implementation and evaluation of cost-effective breastfeeding support interventions in the NHS. The original focus of the study was on women without LTCs; however, we broadened the scope to include women with MLTC when additional funding was awarded. Given the anticipated paucity in evidence for women with MLTC, our review work considered women with single LTCs.

In total, we conducted six systematic reviews:

• two systematic reviews and meta-analyses examining the effectiveness of breastfeeding support for healthy women and women with LTCs

• a theoretically informed mixed-methods synthesis of process evaluations of UK-relevant breastfeeding support intervention

• a mixed-methods synthesis of barriers to and facilitators of breastfeeding support in women with LTCs

• two economic evaluations of breastfeeding support for healthy women and women with LTCs.

This study also contained embedded stakeholder engagement in the form of stakeholder working groups, parents’ panels, focus group discussions with women from socially disadvantaged groups and four workshops held across the UK.

The first work package was an update of the Cochrane review on breastfeeding support for healthy women with healthy term infants. 83 We included 116 studies in the review and ‘breastfeeding only’ interventions, which included breastfeeding support only (n = 86), and ‘breastfeeding plus’ interventions (n = 30), which included other aspects of maternal and child health such as vaccinations, well-baby clinics, intrapartum care and contraceptive services. We found moderate‐certainty evidence that ‘breastfeeding only’ interventions probably led to a small reduction in the risk of women stopping exclusive breastfeeding at 6 months, 4–6 weeks, 2 months and 3–4 months, and stopping any breastfeeding at 6 months, 4–6 weeks and 3–4 months. Effect estimates ranged from RR 0.93 (95% CI 0.89 to 0.97) for stopping any breastfeeding at 6 months to RR 0.81 (95% CI 0.77 to 0.95) for stopping exclusive breastfeeding at 3–4 months. Effect estimates were generally greater for exclusive breastfeeding than for any breastfeeding.

For ‘breastfeeding plus’ the evidence was less consistent. Support probably reduced the number of women stopping any breastfeeding or exclusive breastfeeding at 6 months. The evidence suggests that ‘breastfeeding plus’ support probably results in little to no difference in any of the other outcomes. Effect estimates ranged from RR 0.94 (95% CI 0.91 to 0.97) for stopping any breastfeeding at 6 months to RR 0.73 (95% CI 0.57 to 0.95) for stopping exclusive breastfeeding at 4–6 weeks. Again, effect estimates were generally greater for exclusive breastfeeding than for any breastfeeding. It is not clear why ‘breastfeeding plus’ interventions tended to have less of an impact on the number of women stopping breastfeeding. The proportion of interventions categorised as low, medium and high intensity was broadly similar for ‘breastfeeding only’ and ‘breastfeeding plus’. However, it is feasible that the time spent providing breastfeeding support was less in the ‘breastfeeding plus’ interventions as other aspects of maternal and infant care were also included. There was a lack of information in the intervention characteristics to explore this issue fully. Moreover, although interventions were categorised into ‘breastfeeding only’ and ‘breastfeeding plus’, there was still substantial heterogeneity in interventions.

Meta‐regression was conducted to further explore heterogeneity. This suggested that moderate-intensity (four to eight visits) compared with low-intensity (three or less), support may be beneficial for reducing the number of women stopping exclusive breastfeeding at 4–6 weeks or 6 months. Additionally, women in LMICs were less likely to have stopped exclusive breastfeeding at 6 months, and this may be explained by the higher background breastfeeding rates at 6 months in LMICs. 14 However, beyond this the meta-regression did not explain the high levels of heterogeneity. As we did not want to increase the likelihood of false positives from the meta-regression, we limited the number of variables to four,88 and these were determined in conjunction with stakeholders. There is, therefore, a possibility that variables not included in the meta-regression may be contributing to the high levels of heterogeneity. For example, just under half of the included studies focused on populations described as high poverty, deprivation and poor health outcomes, and it is possible that this may explain some of the heterogeneity as breastfeeding rates typically are lower in groups of high levels of deprivation in HICs. 14

The second work package comprised a mixed-methods synthesis of process evaluations of effective breastfeeding support interventions identified in work package 1. We included 16 studies linked to 10 effective interventions. The identified 18 factors affecting implementation of interventions, and data-driven analytical themes were mapped to a theoretical implementation framework resulting in three overarching, theoretically informed, analytical themes: (1) assessing the needs of those delivering and receiving breastfeeding support interventions; (2) assessing the context and optimising delivery and engagement with breastfeeding support interventions; and (3) reflecting and evaluating the success of implementing and providing breastfeeding support. Included studies identified implementation challenges relating to the needs, preferences and priorities of intervention providers and recipients. Overall, breastfeeding women perceived support as positive, important and needed. Breastfeeding supporter training was a commonly reported implementation strategy that enabled implementation teams to address breastfeeding supporters’ needs. Studies reported contextual factors (e.g. alignment with local policies) affecting the implementation and delivery of breastfeeding support interventions as well as tailoring strategies (e.g. community involvement, use of lay language, responsive support content/information) to address contextual factors. Reports about implementation success focused on key implementation outcomes such as satisfaction, fidelity or usefulness.

The third work package comprised a review of economic evidence of both trial- and model-based evaluations of the incremental cost and incremental cost-effectiveness of breastfeeding support interventions. Of the 39 studies identified, nine were deemed directly or partially applicable to the UK system. Evidence of cost-effectiveness using the UK-recommended incremental cost per QALY gained was limited and inconsistent. For breastfeeding-only support, one study provided evidence that the estimated ICER for the intervention was not cost-effective, at £56,075 per QALY gained. This ICER held in deterministic and probabilistic sensitivity analyses. However, there were notable limitations to the model, with the exclusion of costs (cost savings) and benefits to infants beyond 1 year of age and clinical conditions that were excluded, such as obesity. A lack of good-quality epidemiological and cost data warranted the exclusion but highlights the uncertainty in the findings and the need for more robust evidence to inform future economic evaluations. There was evidence for the incremental cost per additional woman breastfeeding (any or exclusive) with ICERs ranging from £67 to £112 from 2 weeks up to 8 weeks postpartum and from £2446 to £4226 up to 6 months postpartum. However, we judged the findings to be uncertain because of the limited number of studies and the lack of good-quality evidence. None of these studies extrapolated data beyond the time horizon of the associated trial, and potential costs (cost savings) from health service use were not estimated and valued. Without WTP thresholds, whether or not the findings were cost-effective was unclear. Evidence for ‘breastfeeding plus’ support suggested that this was not cost-effective in terms of cost per QALY gained, with similar inconsistencies in results. The scope of costs and outcomes reported and the time horizon for many of the studies was limited. What is missing from the evidence is a high-quality trial-based economic evaluation that then models costs and outcomes beyond the trial period. If breastfeeding in itself is considered a cost-effective intervention, then the provision of additional effective support to populations or subgroups of women with lower rates of breastfeeding initiation is likely to be worth the investment. Engagement with stakeholders during the workshops elicited a positive value for a breastfeeding support intervention, with a WTP of £89.91/£105.04 per woman for a 1% reduction in drop-off for any/exclusive breastfeeding at 6 weeks postpartum. If policy- and decision-makers are willing to pay this cost to realise this outcome, then such a breastfeeding support intervention, delivering four to eight face-to-face contacts with women by a combination of providers, would be considered value for money.

The first work package of the additional funding aimed to identify effective interventions that provide breastfeeding support for women with LTCs. We identified 22 studies that met the inclusion criteria, all of which were for women with single LTCs. A range of conditions were identified: overweight and obesity (nine studies), HIV (five studies), gestational diabetes (two studies), substance misuse (two studies) and depression (one study). Interventions varied in terms of whether they provided only breastfeeding support or if they also provided support for the LTC. No studies were identified for women with MLTC. In contrast to the Cochrane review of breastfeeding support,83 most studies had an antenatal component. The importance of antenatal support, particularly having a flexible feeding plan, was raised by the stakeholder working group and parents’ panel for women with MLTC. Effect estimates for the primary breastfeeding outcomes were generally small and crossed the 95% CI, which suggests that included interventions probably had little to no impact on the number of women stopping breastfeeding. Effect estimates ranged from RR 0.83 (95% CI 0.67 to 1.01) for stopping any breastfeeding at 6 months to RR 0.95 (95% CI 0.89 to 1.00) for stopping exclusive breastfeeding at 6 months. Findings for the additional breastfeeding outcomes were similar. Due to the small number of studies, meta-regression to explore the impact of the nature of the LTC on breastfeeding rates was not possible. Sensitivity analysis did not find a difference in findings when studies with women with HIV were excluded. Similarly, due to the small number of studies, meta-regression was not possible to explore possible causes of heterogeneity, such as nature of condition or socioeconomic deprivation. Moreover, only a few studies had beneficial intervention effects for at least one outcome. 184,185,187 It is, therefore, not possible to make any conclusions about support being more or less effective for specific conditions. Similarly, a narrative synthesis showed little to no beneficial effect on maternal and infant health outcomes.

The second work package for women with MLTC comprised a mixed-methods synthesis of experiences of breastfeeding support for women with LTCs. The 24 included studies covered health conditions including HIV, obesity and overweight, substance use, diabetes in pregnancy, disabilities and a rare genetic disorder. Key findings were that women with LTCs have additional breastfeeding support needs, but that breastfeeding support can be difficult to access. Women and healthcare providers reported challenges including the overshadowing of breastfeeding support by condition-related support and supporters lacking in knowledge and skills. Suggested strategies to improve breastfeeding support for mothers with LTCs included acknowledging the influence of partners, families and friends and training healthcare providers to improve their understanding of the specific breastfeeding support needs of women with LTCs.

The third work package for women with MLTC conducted a review of economic evidence for breastfeeding support interventions for women with LTCs. Five evaluations were identified that assessed cost and effect for women with a small range of health conditions: HIV, obesity, prenatal opioid use, and women considered medically high risk (maternal hypertension and diabetes prior to birth). There was a lack of evidence of cost-effectiveness from full economic evaluations, with limited scope in the costs and benefits valued. One cost-effectiveness analysis study reported a cost of US$88 per increased month of exclusive breastfeeding to support women living with HIV to breastfeed, while a cost–utility analysis study reported that promoting breastfeeding was less costly and more effective, in terms of disability-adjusted life-years averted, for women living with HIV in rural areas than the current scenario. A third cost–utility analysis study reported less cost and more effect, in terms of QALYs gained, for breastfeeding support using rooming-in after childbirth for women with prenatal opioid use. However, none of the studies met the applicability criteria for the UK system, making it likely that these conclusions of cost-effectiveness would change if tested in a UK setting.

The final phase of the project involved developing and refining a toolkit for implementing and evaluating effective breastfeeding interventions relevant to the UK, based on synthesising the findings of the reviews and stakeholder and parent engagement along with the views of a broader group of stakeholders who attended workshops. The toolkit presents an example intervention based on high-quality evidence on effective breastfeeding support interventions. The intervention comprises structured, proactive antenatal and postnatal components, combines professional and peer support, and offers face-to-face and telephone follow-up. The toolkit proposes the most important considerations when adapting this evidence-based intervention for local services, which are acceptability to the local population, the quality of the primary evidence, and the sustainability of the intervention. Regarding tailoring the intervention for women with LTCs, the most important modifications to be considered are more time for antenatal breastfeeding support, continuity of support, and including infant-feeding specialists in combined obstetric and medical clinics.

The toolkit highlights barriers that may be encountered when implementing breastfeeding support interventions considering the intervention itself, the broader societal setting, the context of local services, the roles and capabilities of those implementing and receiving the intervention and, finally, the process of implementation (see Appendix 8). The toolkit proposes a range of strategies that can be used to address barriers, the most important of which are providing information on how to deliver the intervention and why it is important, assigning roles such as a key practitioner to raise awareness, having an infant-feeding team to lead implementation, integrating peer support with NHS services, and leveraging investment from local councils and government as well as the NHS.

Finally, the toolkit proposes considerations for evaluating the intervention, including whether women meet their infant-feeding goals and expectations and whether the support is helpful. The suggested times to measure breastfeeding outcomes are first feed, discharge from hospital, 6–8 weeks and 6 months. Other important outcomes to consider are infant admissions to hospital and the reason for stopping breastfeeding. Process data to be considered include views and experiences of the intervention deliverers and recipients, women’s satisfaction and intervention fidelity. To assess impact on inequalities, data should be collected on women’s characteristics and the intervention and evaluation should be inclusive, that is accessible to all women.

Agreements and disagreements with other reviews

First, in terms of the Cochrane review, both ‘breastfeeding only’ and ‘breastfeeding plus’ tended to have a greater impact on exclusive breastfeeding. One explanation for this comes from a realist review that suggested that more highly motivated mothers may benefit more from breastfeeding support. 236 In addition, effect estimates tended to be greater at earlier time points, which may be a consequence of support being primarily targeted at the first 1–2 months. At later time points, wider issues around returning to employment influence breastfeeding rates237 and may not be considered in the interventions included. The results of this meta-analysis are similar to effect estimates reported in a review looking at breastfeeding counselling interventions. 238 Other systematic reviews looking at support interventions have shown greater effect estimates;239 however, these reviews identified a much smaller number of studies due to limitations in search strategies and selection processes. In addition, previous systematic reviews have found greater effect estimates for multicomponent breastfeeding support (i.e. providing different aspects of breastfeeding support in a combination of settings such as BFHI). 40,240,241 We did initially aim to categorise the interventions based on breastfeeding support components; however, given the large number and heterogeneity of interventions we were unable to do this in any meaningful way. Interestingly, our review suggested slightly higher effect estimates than did a review looking at breastfeeding support that was provided on a remote basis only. 242 However, in our review, meta-regression did not identify any clear differences between support provided remotely and that provided face to face, but the power to detect any differences was limited. Reviews of alternative methods to increase breastfeeding rates have identified a relatively small number of studies and no clear intervention effects, for example incentives243 or workplace-based strategies. 244

To the best of our knowledge, this is the first systematic review to focus on implementation research linked to breastfeeding support interventions for healthy women with healthy term babies that have shown effectiveness in RCTs. However, some existing reviews have looked more widely at the views and experiences of those delivering and receiving breastfeeding support interventions and have reported findings that are well aligned with our review. These include the importance of key intervention strategies that women perceive as supportive, such as those that rely on the provision of both practical/technical expertise245–247 and emotional support/encouragement245,246,248 and are person centred and socioculturally specific,246,247,249 as well as key implementation issues such as the importance of contextual factors. 249,250 In terms of the review on effectiveness of breastfeeding support for women with LTCs, our findings are consistent with a Cochrane review of support targeted at women with overweight and obesity for which only a few small-scale studies were identified. 251 Meta-analysis likewise identified small effect estimates and imprecision. A further systematic review that included any intervention (e.g. support, breast pumps, education) designed to increase breastfeeding initiation and continuation in women with overweight/obesity also did not appear to show any impact on improving breastfeeding rates. 252 To the best of our knowledge, there are no existing reviews of breastfeeding support for any other form of LTC.

Our mixed-methods synthesis of experiences of breastfeeding support for women with LTCs is consistent with other review findings. This includes that overweight/obese women find breastfeeding challenging. 253,254 Similar to our review, Chang et al. 253 concluded that healthcare professionals require education to enable them to provide tailored, non-judgemental breastfeeding support. Cummins et al. 255 made similar recommendations based on their systematic review of in-hospital support for women with GDM. Tanganhito et al. 256 emphasised the influence of family and friends and professional support for women with postnatal depression.

In terms of the review of economic evidence, our findings resonate with one previous economic evidence review. 257 This review was conducted to inform NICE guidance on postnatal care and comprised seven studies. The authors judged the existing evidence to be inconclusive. While their inclusion criteria had a wider remit of breastfeeding education, advice and support interventions, which included financial incentives, their findings were consistent with the findings in the current review for ‘breastfeeding only’ support. The review highlighted similar limitations and inconsistencies between studies, such as the limited time horizon, the different economic outcomes estimated and the different scope of costs and benefits measured and valued, which have an impact on the strength of any conclusions.

Strengths and limitations

This study has several key strengths. First, a criticism of systematic reviews is a lack of uptake of review findings into policy and practice;258 however, the mixed-methods reviews and stakeholder engagement have enabled to us to understand how interventions could be effectively implemented in practice. To address this, we included two mixed-methods syntheses that aimed to explore how such support could be implemented in the NHS for all women. We believe that this is the first comprehensive synthesis of evidence of effectiveness of breastfeeding support and of barriers to and strategies for implementing breastfeeding support for women with and women without LTCs. Furthermore, our work has been underpinned by implementation frameworks providing theoretically informed recommendations in the form of a toolkit. Finally, and perhaps most importantly, it had extensive PPI and stakeholder involvement that ensured a co-created output grounded in the realities of women’s experiences of breastfeeding, particularly those from socially disadvantaged groups, and NHS context and practice. Hopefully, this gives a sense of ownership to those involved in the project. The toolkit should be relevant and adaptable to the four UK nations. Second, the two effectiveness reviews and meta-analyses followed Cochrane methodology to ensure rigour. Third, the update of Cochrane review of breastfeeding support83 included the use of a new trustworthiness checklist, which helps ensure that the findings of this review are not based on fraudulent data. 86

However, there are several limitations that should be considered. With all systematic reviews there is the potential for bias to be introduced. First, although we did involve two reviewers in all review processes (e.g. study selection, data extraction, critical appraisal, synthesis, GRADE), these judgements are subjective. Second, except for the Cochrane review, studies not published in English were excluded, so there is a risk of language bias. Third, although we attempted to identify all available evidence meeting our inclusion criteria, it is possible that we did not identify all studies, and the Cochrane review in particular showed some evidence of funnel plot asymmetry, which may be suggestive of publication bias. Fourth, issues in reporting meant that there was often insufficient information about intervention characteristics (e.g. person providing the intervention, number of contacts, theoretical basis, definitions of exclusive breastfeeding, nature of standard care). Fifth, the systematic reviews on effectiveness identified a lack of digitally provided interventions. As the COVID-19 pandemic has led to an increase in remotely provided maternity care,242 this evidence is perhaps limited in a post-COVID world. Sixth, our syntheses were limited by the mixed quality and lack of published process evaluations linked to effective interventions, as well as the relative dearth and poor quality of studies of experiences of breastfeeding support for women with LTCs. The latter body of evidence covers a very limited range of conditions, with many being studies of HIV-positive women in LMICs and of obese/overweight women in HICs. We did not find any studies of experiences of breastfeeding for women with mental health conditions. Seventh, there was a lack of evidence from the UK. This is representative of a long-standing problem whereby UK trials have failed to demonstrate benefits for breastfeeding outcomes, possibly due to the interventions tested and the way they were delivered rather than the trial design. 44 Eighth, the search for the Cochrane review on breastfeeding support was conducted in May 2021 and will not have included any studies that look at digital support post COVID-19. Ninth, a post hoc decision was made to exclude breastfeeding initiation from the review on effectiveness of support for women with LTCs. Studies that included this as an outcome used considerably different definitions (e.g. within 1 hour vs. ever), which gave rise to some nonsensical findings, such as in the same study more women breastfeeding at 4–8 weeks than had initiated it. Finally, there is unexplained heterogeneity in both the Cochrane review and the review on breastfeeding support for women with LTCs. In both these reviews, just under half of the studies were targeted at populations at risk of poorer outcomes (e.g. high levels of socioeconomic deprivation, ethnicity, young motherhood). As these factors influence breastfeeding rates,14 it is possible that the impact of support is different in these populations. However, for the Cochrane review this was not included as a variable in our meta-regression and for the LTC review there were insufficient studies to investigate this. In addition, the review for women with LTCs has additional heterogeneity due to the different conditions included.

Strengths and limitations of patient and public involvement and stakeholder involvement

We used the GRIPP2 (Guidance for Reporting Involvement of Patients and Public) checklist to inform our account of PPI in the study. 259 There was significant involvement of stakeholders and PPI in this project. In addition to the research team’s reflections, we sought the views of the main study stakeholder working groups and parents’ panels on their engagement.

First, our research team included a PPI co-applicant (PB) who was involved at all stages from the initial design to writing the final report and disseminating the findings. This ensured the PPI voice in all team meetings, providing valuable advice and feedback and influencing decisions. Furthermore, PB participated in the systematic reviews, including study selection, data extraction, quality appraisal and interpreting the results, and is a co-author of the Cochrane review. 260

Stakeholder engagement and PPI were identified as a cross-cutting theme in the study protocol, co-led by co-applicants PB and JM, ensuring that it was a standing agenda item in all team discussions and study steering committee meetings. A key responsibility of the full-time project manager was co-ordinating the stakeholder working group and PPI meetings ensuring sufficient administration time was dedicated to it.

A considerable strength was the range of individuals involved in the stakeholder working groups and parents’ panels. Members of the parents’ panels had a wide range of breastfeeding experiences, and experiences of breastfeeding with a range of comorbidities. We also included two fathers in the main study parents’ panel. Members of the stakeholder working groups represented the main health professions involved in breastfeeding support as well as the key national breastfeeding support third-sector organisations, and a national policy-maker. This work was enhanced by conducting focus group discussions in an area of high deprivation and ethnic diversity to ensure that we gained perspectives from communities that have low breastfeeding rates and to complement the parents’ panels. A further strength was that 87 people attended the co-creation workshops, covering extensive geographies, NHS and third-sector organisations, and parents.

All parents’ panels and stakeholder working group meetings were held virtually, by necessity at the outset of the project, which removed geographical barriers from inclusion. We worked hard to keep participants engaged in the work, as can be seen from the level of engagement across the 2-year study. Focus group participants were offered a choice of face-to-face or virtual meetings, and we ran both modes at each of the three time points. Holding the workshops face to face was a huge advantage, and participants provided very positive feedback about the activities and the benefits of working with others on such an important topic. For many, it was their first experience of a face-to-face event since the COVID-19 restrictions were lifted.

All those involved have been remunerated for attending meetings, as well as for expenses incurred travelling to the workshops. NHS organisations were reimbursed for releasing staff to attend meetings and workshops.

We were transparent at every stage of the study about how the PPI and stakeholder involvement influenced the study, including in co-creation of the toolkit. Feedback from the main study parents’ panel and stakeholder working group was that they felt proud to be involved and enjoyed seeing how the project evolved, that the meetings were very inclusive, and that the communication from the team both during and between meetings was very informative and clear.

There were several limitations to this component of the study. We acknowledge that recruiting parents via a third-sector organisation could have resulted in participants who were mainly from middle-class backgrounds. We feel that we mitigated this by conducting the focus group discussions. However, we did not collect sociodemographic data from participants in the parents’ panels and focus groups. We did not recruit any women to the parents’ panels and focus group discussions who had exclusively formula-fed their babies, and this could be considered a limitation. However, this was because the focus of our work was supporting women who had chosen to breastfeed in continuing longer and increasing exclusivity. Nevertheless, our parents’ panels and focus group discussion participants included several women who had combined formula feeding and breastfeeding, and those who had breastfed initially but had switched to formula feeding because of the challenges they had faced. We believe that this brought a wide range of views to our work. We had originally intended to conduct the initial meeting of the main study stakeholder working group face to face, but this was not possible due to COVID-19 restrictions. A face-to-face meeting may have helped build rapport and allowed for informal conversations. We were aware that some participants accessed meetings on their mobile phones and tried to plan activities accordingly, but it was still challenging for some. Finding a convenient time for meetings was difficult, and although we offered evening times for the parents’ panels, this option was not taken up. Nevertheless, some parents were disappointed that they could not attend all meetings. Several members of the stakeholder working group changed roles during the study, and offered replacements, but this inevitably resulted in some lost continuity. The university processes for reimbursement were bureaucratic and time-consuming for the participants and the project manager. Although our workshops had good attendance, many more people registered than attended. The workshops were held in November 2022, a time of high levels of winter illnesses (COVID-19 and flu), travel disruption and high demand in the NHS, all of which affected attendance. Some members of the parents’ panels were disappointed that they could not attend a workshop because of distance and full-time employment. One suggestion from the parents’ panel was to have a combined meeting with the parents’ panel and the stakeholder working group. We held the meetings separately to ensure that the parents’ voices were heard but will consider at least one combined meeting in future projects.

Implications for practice

Considering the importance of breastfeeding for public health and the existence of high-quality, moderate-certainty evidence of what works to support healthy women to breastfeed, the key challenge is overcoming the barriers to implementing breastfeeding support interventions. Decision-makers and frontline practitioners can use the toolkit to inform implementation efforts and to overcome barriers specific to their settings. Further co-development work is ongoing with an extended set of stakeholders to refine the draft toolkit and produce a user-friendly output that will support NHS and third-sector organisations to implement evidence-based breastfeeding support for women in the UK. Key to success will be addressing the system barriers and enhancing the skills, knowledge and confidence of practitioners. To reduce inequalities, interventions must be adapted to be accessible to all women, for example by ensuring that venues are accessible at a low cost and that language and cultural barriers are considered. Breastfeeding peer support is lacking across much of the UK. 33 Addressing barriers to integrating peer support with health service support is needed, as suggested by Trickey et al. 236 This requires action by health service strategic and operational decision-makers to adequately resource and value peer support as integral to effective breastfeeding support.

While less research evidence is available on how to provide effective breastfeeding support for women with LTCs, our stakeholder engagement and PPI work highlighted additional support needs and proposed possible strategies for achieving this. Health services could consider implementing proposals to integrate an infant-feeding specialist with the multidisciplinary team to give infant feeding a higher profile in obstetric and medical care.

The lack of knowledge, skills and confidence of those providing breastfeeding support is a frequent theme in research on breastfeeding support. Our stakeholder work suggested that training to UNICEF UK Baby-Friendly Initiative standards261 should be a minimum level for those providing care to mothers and infants. However, our workshop participants also proposed enhancing the training of those delivering breastfeeding support to lactation consultant level. Any upskilling strategies should incorporate the needs of women with LTCs.

The toolkit can be used by those leading breastfeeding support services to guide implementation efforts. This will probably necessitate rethinking existing roles and skill mix and involve finding ways to work with other sectors such as third-sector and community organisations. According to our work, a key to effective implementation is providing feedback to staff through data sharing.

The societal and commercial influences on women’s breastfeeding experiences are well recognised. 17,262 Although this needs a whole-system approach beyond the scope of our work, one strategy emphasised by our project is to involve partners and wider families in breastfeeding support interventions, as found in Bengough et al. 249 Regarding reducing inequalities in breastfeeding, the current economic climate and cost-of-living crisis is likely to exacerbate inequalities and necessitate the consideration of minimising costs to breastfeeding women such as ensuring that venues are accessible and helping with travel costs. Digital poverty must also be considered if the breastfeeding service has a digital component. Exploring the needs and preferences of the local population and working with a wide range of third-sector organisations and local government could address this.

Suggested future research

Crucially, this study found only a small number of studies on breastfeeding support for women with LTCs and a lack of evidence on cost-effectiveness in this group, compared with the large number of studies looking at support for healthy women. Moreover, both reviews identified that effect estimates were generally small. There is therefore a need to develop support interventions that are effective for all women. While further inspection of the Cochrane review findings did identify specific interventions that had larger effects and could form the basis of a NHS intervention, many of the barriers to breastfeeding for women with LTCs identified by our parents’ panel and stakeholder working group, and the mixed-methods synthesis, would not be considered in these interventions. In particular, there is a greater need for antenatal support and development of a feeding plan, consistent communication between healthcare professionals regarding medication safety, and the consideration of breastfeeding as a physical activity. There is therefore a need to develop and test an intervention for women with MLTC that takes account of these aspects. In particular, this work identified a very small number of studies for women with mental health conditions. In addition, while many of our included studies did focus on women with overweight/obesity and GDM, the interventions were generally not effective. Given the prevalence and co-occurrence of these conditions, and the fact they are more likely to affect women from groups least likely to breastfeed, we would suggest these as priority areas.

Both systematic reviews on the effectiveness of breastfeeding support identified a lack of digitally provided interventions. As the COVID-19 pandemic has led to an increase in remotely provided maternity care,242 there is also a need to consider how digital technologies could be utilised. However, both our work with stakeholders and existing research263 suggest that remotely provided support cannot be a replacement for face-to-face support and thus it should be provided alongside face-to-face breastfeeding support.

More research is needed on the experiences of receiving and providing breastfeeding support among women with LTCs and those with multimorbidities.

Evidence for the effectiveness of breastfeeding feeding support interventions in the UK is lacking, and the toolkit can be used to guide evaluation design. This could be via implementation or effectiveness studies or by using quality improvement methodology. Studies could be based on the prototype intervention developed for this study (tailored to local contexts), as described in the draft toolkit, and could test different implementation strategies for effectiveness. Further evidence of value for money in a UK setting is also needed.

Future economic evaluations would need to address the current limitations in the evidence in terms of the short time horizon and limited scope of health service resource use measured and valued. A cost–utility analysis could be conducted alongside an effectiveness study, combining trial-based and model-based evidence with long-term follow-up of mother–child dyads to collect data on resource use and health-related quality of life, and modelling costs and benefits over the lifetime. A societal perspective should also be considered in conjunction with the provider (NHS) perspective to gain a better understanding of the opportunity cost of providing support to women to breastfeed.

Equality, diversity and inclusion

We addressed equality, diversity and inclusion in the following ways:

• Our work focuses on support for breastfeeding women; women of childbearing age and pregnant women are recognised as underserved groups.

• The stakeholder working groups included healthcare practitioners serving ethnically diverse and disadvantaged populations, and rural localities across the UK.

• The parents’ panel for the main study included a Gypsy/Traveller mother (one the most socially marginalised groups in the UK) and two fathers (men are rarely included in breastfeeding research).

• The parents’ panel for women with MLTC included women with multiple physical and mental health conditions and are a group who face additional challenges in accessing breastfeeding support and are often excluded from breastfeeding research.

• For the main study, we ensured the voices of women from ethnically diverse and socioeconomically deprived populations were included through conducting focus group discussions in West Yorkshire to supplement the views of the parents’ panel.

• We ensured all communication was accessible for participants.

• We offered evening meetings for the parents’ panels.

• We paid parents and third-sector organisation representatives for their involvement to value their contributions.

• In our workshops, we focused activities on the needs of populations with low breastfeeding rates.

• The co-applicant team involved a range of levels of experience, included male and female researchers, and a PPI representative.

• Our approach to the work was inclusive and everyone had the opportunity to contribute all aspects resulting in co-authorship of the report and development of knowledge and skills in evidence synthesis methods.

• We also included a wide range of early career researchers including doctoral students in the conduct of the reviews to develop skills and have co-authorship of the resulting publications including the Cochrane review.

Regarding limitations, the research team (co-investigators) was not ethnically diverse, and we will consider this in future research. We acknowledge that recruiting parents to the parents’ panels via a variety of Facebook breastfeeding support groups, including those run by third-sector organisations, somewhat restricted those who engaged with us in terms of diversity of backgrounds. In future work, we will consider different strategies to optimise diversity.

Conclusions

‘Breastfeeding only’ support probably leads to a small reduction in the number of women stopping any and exclusive breastfeeding. ‘Breastfeeding plus’ support and breastfeeding support for women with LTCs probably leads to little or no reduction in the number of women stopping breastfeeding for most outcomes. As the work with stakeholders and mixed-methods review identified that women with LTCs face additional challenges when breastfeeding, more research is needed to develop effective support. In addition, evidence for the effectiveness and cost-effectiveness of breastfeeding feeding support interventions in the UK is lacking.

Acknowledgements

We would like to thank all members of our stakeholder working groups, parents’ panels, focus group participants and workshop attendees for their time and insights.

We also thank members of the study steering committee for their invaluable advice and encouragement.

Thanks to the following people for their contribution to the project:

• Karen Allum

• Sharon Carstairs

• Sushila Chowdhry

• Shadrach Dare

• Wendy Jones

• Jayne Samples

• Jonathan West

• Flavia Ximenes Vasconcelos.

Contributions of authors

Anna Gavine (https://orcid.org/0000-0003-3750-2445) (Lecturer) led the systematic reviews on effectiveness of breastfeeding support for healthy women [update of Cochrane review (see Chapter 3)] and breastfeeding support for women with long-term conditions (see Chapter 6); participation in tasks related to mixed-methods review (see Chapter 7); design and running of stakeholder working groups, parents’ panels and workshops.

Albert Farre (https://orcid.org/0000-0001-8970-6146) (Lecturer) led the mixed-methods systematic reviews for healthy women (see Chapter 4) and women with long-term conditions (see Chapter 7); design, running and analysis of stakeholder working groups, parents’ panels and workshops; participation in tasks related to update of Cochrane review (see Chapter 3).

Fiona Lynn (https://orcid.org/000-0002-0216-643X) (Senior Lecturer) led the systematic reviews on cost-effectiveness (see Chapters 5 and 8); design and running of stakeholder working groups, parents’ panels and workshops; participation in tasks related to update of Cochrane review (see Chapter 3).

Shona Shinwell (https://orcid.org/0000-0001-9369-9698) (Project Manager) participation in tasks related to update of Cochrane review (see Chapter 3) and mixed-methods systematic review (see Chapter 4); design and running of stakeholder working groups, parents’ panels and workshops.

Phyllis Buchanan (https://orcid.org/0000-0002-1436-4396) (PPI Breastfeeding Network) participation in tasks related to update of Cochrane review (see Chapter 3) and effectiveness of breastfeeding support for women with LTCs (see Chapter 6); design, running and analysis of stakeholder working groups, parents’ panels, focus groups and workshops.

Joyce Marshall (https://orcid.org/0000-0002-2784-1817) (Lecturer) design, running and analysis of focus groups, stakeholder working groups, parents’ panels and workshops; participation in tasks related to update of Cochrane review (see Chapter 3).

Sara Cumming (https://orcid.org/0000-0003-0714-128x) (Clinical Academic Fellow) participation in tasks related to update of Cochrane review (see Chapter 3), mixed-methods reviews (see Chapters 4 and 7) and effectiveness of breastfeeding support for women with LTCs (see Chapter 6); design and analysis of stakeholder working groups, parents’ panels and workshops.

Louise Wallace (https://orcid.org/0000-0003-3770-0580) (Professor) study design and running of workshops.

Angie Wade (https://orcid.org/0000-0002-3944-8908) (Professor) design and analysis of update of Cochrane review (see Chapter 3); statistical advice for Chapter 6.

Elayne Ahern (https://orcid.org/0000-0001-9230-6776) (Lecturer) contributed to tasks related to the cost-effectiveness reviews (see Chapters 5 and 8).

Laura Hay (https://orcid.org/0000-0002-3259-9463) (Project Manager) design of and contribution to systematic review on effectiveness of breastfeeding support for women with LTCs (see Chapter 6); design and running of stakeholder working groups, parents’ panels and workshops.

Marianne Cranwell (https://orcid.org/0000-0003-0605-3923) (Research Assistant) analysis of stakeholder working group and parents’ panels; contribution of tasks related to mixed-methods review (see Chapter 7).

Alison McFadden (https://orcid.org/0000-0002-5164-2025) (Professor) led stakeholder and parent engagement work packages; participated in tasks related to update of Cochrane review (see Chapter 3), systematic review on effectiveness of breastfeeding support for women with LTCs (see Chapter 6), mixed-methods reviews (see Chapters 4 and 7); project oversight.

All authors were involved in drafting and/or commenting on the report.

Disclosure of interests

Full disclosure of interests: Completed ICMJE forms for all authors, including all related interests, are available in the toolkit on the NIHR Journals Library report publication page at https://doi.org/10.3310/DGTP5702.

Primary conflicts of interest: Louise Wallace was a panel member of the following: DH National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research (HSDR) Development panel (2012–13); NIHR HSDR Researcher Led panel (2013–15); SDO Service Evaluations panel (2009–11); and NIHR Health Services & Research Development panel (Seacole) (October 2020–22). She received no payments for these other than expenses. Louise Wallace was also a paid scientific advisor for the NIHR HSDR Remit and Competitive Committee (2015–19).

Data-sharing statement

Further anonymised data are available on request from the corresponding author.

Ethics statement

The stakeholder engagement and PPI components of the study were approved by the University of Dundee School of Health Sciences research ethics committee (UOD‐SHS‐2021‐010) on 24 June 2021.

Information governance statement

The University of Dundee is committed to handling all personal information in line with the UK Data Protection Act (2018) and the General Data Protection Regulation (EU GDPR) 2016/679. Under the Data Protection legislation, the University of Dundee is the Data Controller, and you can find out more about how we handle personal data, including how to exercise your individual rights and the contact details for our Data Protection Officer here https://www.dundee.ac.uk/corporate-information/data-protection-policy

Department of Health and Social Care disclaimer

This publication presents independent research commissioned by the National Institute for Health and Care Research (NIHR). The views and opinions expressed by authors in this publication are those of the authors and do not necessarily reflect those of the NHS, the NIHR, MRC, NIHR Coordinating Centre, the HSDR programme or the Department of Health and Social Care.

This monograph was published based on current knowledge at the time and date of publication. NIHR is committed to being inclusive and will continually monitor best practice and guidance in relation to terminology and language to ensure that we remain relevant to our stakeholders.

Publication

Gavine A, Shinwell SC, Buchanan P, Farre A, Wade A, Lynn F, et al. Support for healthy breastfeeding mothers with healthy term babies. Cochrane Database Syst Rev 2022;10:CD001141.

Disclaimers

This article presents independent research funded by the National Institute for Health and Care Research (NIHR). The views and opinions expressed by authors in this publication are those of the authors and do not necessarily reflect those of the NHS, the NIHR, the HSDR programme or the Department of Health and Social Care. If there are verbatim quotations included in this publication the views and opinions expressed by the interviewees are those of the interviewees and do not necessarily reflect those of the authors, those of the NHS, the NIHR, the HSDR programme or the Department of Health and Social Care.

MEDLINE search strategy for main study mixed-methods systematic review (see Chapter 4)

MEDLINE search strategy for main study economic evidence review (see Chapter 5)

Ovid MEDLINE^® ALL 1946 to 17 August 2022

n = 2911, searched on 2 February 2022

1. exp Breast Feeding/

2. breastfeed*.mp.

3. breastfed.mp.

4. breast-feed*.mp.

5. breast-fed.mp.

6. breast feed*.mp.

7. breast fed.mp.

8. infant feed*.mp.

9. exp Milk, Human/

10. Lactation/

11. lactat*.mp.

12. support.mp.

13. Social Support/

14. advice.mp.

15. advis*.mp.

16. help*.mp.

17. supportive adj2 relationship.mp

18. counsel*.mp.

19. educat*.mp.

20. consult*.mp.

21. Health Promotion/

22. Health Education/

23. Economics/

24. exp “Costs and cost analysis”/

25. “Cost allocation”/

26. Cost-benefit analysis/

27. “Cost control”/

28. “Cost savings”/

29. “Cost of illness”/

30. “Cost sharing”/

31. “deductibles and coinsurance”/

32. Medical savings accounts/

33. Health care costs/

34. Direct service costs/

35. Drug costs/

36. Employer health costs/

37. Hospital costs/

38. Health expenditures/

39. Capital expenditures/

40. Value of life/

41. exp economics, hospital/

42. exp economics, medical/

43. Economics, nursing/

44. Economics, pharmaceutical/

45. exp “fees and charges”/

46. exp budgets/

47. (low adj cost).mp.

48. (high adj cost).mp.

49. (health?care adj cost$).mp.

50. (fiscal or funding or financial or finance).tw.

51. (cost adj estimate$).mp.

52. (cost adj variable).mp.

53. (unit adj cost$).mp.

54. (economic$ or pharmacoeconomic$ or price$ or pricing).tw.

55. or/1-11

56. or/12-22

57. or/23-54

58. 55 and 56 and 57

59. exp animals/ not humans.sh.

60. 58 not 59

MEDLINE search strategy for LTCs effectiveness review (see Chapter 6)

Ovid MEDLINE^® ALL 1946 to 17 August 2022

n = 1144, searched on 18 August 2022

1. exp Breast Feeding/ 42,543

2. (breastfeed* or breast-feed* or breast feed*).ab. 38,704

3. (breastfed or breast-fed or breast fed).ab. 12,845

4. lactation.ab. 35,254

5. infant feed*.ab. 4936

6. exp Lactation/ 46,504

7. exp Breast Milk Expression/ 385

8. exp Milk, Human/ 21,849

9. 1 or 2 or 3 or 4 or 5 or 6 or 7 or 8 129,346

10. ((support* or help or assist* or class* or workshop* or champion* or promot*) adj5 (breastfeed* or breast feed* or breastfed or breast fed or lactation or infant feed*)).ab. 7340

11. exp Social Support/ 78,157

12. anticipatory guidance.mp. 1527

13. exp Counseling/ 47,862

14. counsel*.mp. 152,131

15. exp Directive Counseling/ 4838

16. exp Health Promotion/ 83,692

17. exp Health Education/ 259,211

18. peer support.mp. 6270

19. professional support.mp. 2017

20. 10 or 11 or 12 or 13 or 14 or 15 or 16 or 17 or 18 or 19 478,908

21. exp Chronic Disease/ 598,162

22. chronic disease*.mp. 339,068

23. chronic illness*.mp. 18,795

24. chronic condition*.mp. 23,438

25. (long term condition* or long-term condition*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 2409

26. exp Comorbidity/ 124,635

27. (comorbid* or co-morbid*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 306,350

28. exp Multimorbidity/ 2332

29. (multimorbid* or multi-morbid*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 8950

30. (multidiseas* or multi-diseas*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 265

31. cancer.mp. or exp Neoplasms/ 4,276,838

32. atrial fibrillation.mp. or exp Atrial Fibrillation/ 98,446

33. cardiomyopathy.mp. or exp Cardiomyopathies/ 141,244

34. heart failure.mp. or exp Heart Failure/ 240,900

35. exp Hypercholesterolemia/or exp Hyperlipidemias/ 69,579

36. (hypercholesterol?emia or hyperlipid?emia).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 75,870

37. exp Hypertension/or hypertension.mp. 541,419

38. exp Myocardial Ischemia/ 461,609

39. (isch?emic heart disease or myocardial infarction).mp. [mptitle, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 296,886

40. peripheral vascular disease.mp. or exp Peripheral Vascular Diseases/ 64,594

41. exp Stroke/or stroke.mp. 371,135

42. exp Ischemic Attack, Transient/ 21,493

43. transient isch?emic attack.mp. 12,427

44. congenital heart disease.mp. or exp Heart Defects, Congenital/ 176,482

45. valvular heart disease.mp. or exp Heart Valve Diseases/ 135,662

46. rheumatic heart disease.mp. or exp Rheumatic Heart Disease/ 14,973

47. exp Heart Diseases/ 1,235,397

48. (heart disease or cardiac disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 211,888

49. alopecia areata.mp. or exp Alopecia Areata/ 5330

50. vitiligo.mp. or exp Vitiligo/ 8844

51. exp Eczema/or eczema.mp. 23,857

52. psoriasis.mp. or exp Psoriasis/ 59,218

53. acne.mp. or exp Acne Vulgaris/ 20,890

54. hidradenitis suppurativa.mp. or exp Hidradenitis Suppurativa/ 3821

55. lichen planus.mp. or exp Lichen Planus/ 11,081

56. rosacea.mp. or exp Rosacea/ 4650

57. seborrheic dermatitis.mp. or exp Dermatitis, Seborrheic/ 3404

58. allergic rhinitis.mp. or exp Rhinitis, Allergic/ 31,279

59. allergic conjunctivitis.mp. or exp Conjunctivitis, Allergic/ 4460

60. exp Hearing Loss/ 75,766

61. (hearing loss or deaf*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 110,959

62. exp Addison Disease/ or addison* disease.mp. 5996

63. exp Adrenocortical Adenoma/ or adren* adenoma.mp. 3139

64. exp Pheochromocytoma/ 16,444

65. ph?eochromocytoma.mp. 23,761

66. exp Cushing Syndrome/ or cushing* syndrome.mp. 15,868

67. exp Diabetes Mellitus, Type 2/ or exp Diabetes, Gestational/ or exp Diabetes Mellitus/ or exp Diabetes Mellitus, Type 1/ 485,761

68. (diabetes or diabetic$1).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 796,593

69. exp Parathyroid Diseases/ or (parathyroid dis* or hyperparathyroid* or hypoparathyroid*).mp. 46,457

70. exp Thyroid Diseases/ or thyroid dis*.mp. 165,219

71. (hyperthyroid* or hypothyroid* or thyroiditis or graves disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 100,159

72. exp Pituitary Diseases/ or pituitary dis*.mp. 64,970

73. exp Endocrine System Diseases/ 1,088,901

74. exp Vision Disorders/ 77,172

75. (visual* impair* or blindness).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 58,699

76. exp Cataract/or cataract.mp. 71,192

77. exp Diabetic Retinopathy/ 28,274

78. (diabetic retinopathy or diabetic eye dis*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 38,358

79. glaucoma.mp. or exp Glaucoma/ 78,243

80. scleritis.mp. or exp Scleritis/ 2363

81. episcleritis.mp. 619

82. exp Uveitis/or uveitis.mp. 40,674

83. retinal detachment.mp. or exp Retinal Detachment/ 28,643

84. exp Eye Diseases/ 619,560

85. alcoholic liver disease.mp. or exp Liver Diseases, Alcoholic/ 18,468

86. autoimmune hepatitis.mp. or exp Hepatitis, Autoimmune/ 7214

87. sclerosing cholangitis.mp. or exp Cholangitis, Sclerosing/ 7796

88. primary biliary cirrhosis.mp. or exp Liver Cirrhosis, Biliary/ 10,926

89. chronic hepatitis.mp. or exp Hepatitis, Chronic/ 73,061

90. exp Hepatitis B, Chronic/ or hepatitis B.mp. 108,026

91. exp Hepatitis C, Chronic/ or hepatitis C.mp. 98,802

92. liver cirrhosis.mp. or exp Liver Cirrhosis/ 111,811

93. non-alcoholic fatty liver disease.mp. or exp Non-alcoholic Fatty Liver Disease/ 26,127

94. exp Liver Diseases/ 608,267

95. chronic pancreatitis.mp. or exp Pancreatitis, Chronic/ 16,961

96. c?eliac disease.mp. or exp Celiac Disease/ 26,258

97. food allergy.mp. or exp Food Hypersensitivity/ 25,939

98. cholelithiasis.mp. or exp Cholelithiasis/ 40,132

99. gallstones.mp. 20,398

100. inflammatory bowel disease.mp. or exp Inflammatory Bowel Diseases/ 112,247

101. exp Crohn Disease/ or crohn* disease.mp. 62,081

102. ulcerative colitis.mp. or exp Colitis, Ulcerative/ 55,469

103. proctitis.mp. or exp Proctitis/ 4698

104. irritable bowel syndrome.mp. or exp Irritable Bowel Syndrome/ 16,561

105. lactose intolerance.mp. or exp Lactose Intolerance/ 3642

106. peptic ulcer.mp. or exp Peptic Ulcer/ 88,760

107. chronic pelvic inflammatory dis*.mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 135

108. exp Pelvic Inflammatory Disease/ 11,366

109. dysmenorrhea.mp. or exp Dysmenorrhea/ 7265

110. endometriosis.mp. or exp Endometriosis/ 31,546

111. infertility.mp. or exp Infertility/ 103,527

112. assisted reproduction.mp. or exp Reproductive Techniques, Assisted/ 80,694

113. (in vitro fertili#ation or IVF).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 40,047

114. leiomyoma.mp. or exp Leiomyoma/ 25,329

115. fibroids.mp. 5638

116. exp Menopause/ or menopause.mp. 74,996

117. menorrhagia.mp. or exp Menorrhagia/ 6281

118. exp Urinary Incontinence/ or exp Pelvic Floor Disorders/ or exp Pelvic Organ Prolapse/ or exp Fecal Incontinence/ 54,690

119. (pelvic floor dysfunction or pelvic floor disorder*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 2942

120. (urinary incontinence or f?ecal incontinence).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 54,435

121. pelvic organ prolapse.mp. 7750

122. exp Polycystic Ovary Syndrome/ or polycystic ovar* syndrome.mp. 21,740

123. recurrent miscarriage.mp. or exp Abortion, Habitual/ 9651

124. exp Blood Coagulation Disorders/ or coagulation disorder.mp. 103,883

125. h?emophilia.mp. 28,903

126. exp Anemia, Sickle Cell/or sickle cell.mp. 31,815

127. thalass?emia.mp. or exp Thalassemia/ 30,118

128. thrombophilia.mp. or exp Thrombophilia/ 30,397

129. pernicious an?emia.mp. or exp Anemia, Pernicious/ 7005

130. exp Thrombocytopenia/ or primary thrombocytopenia.mp. 52,195

131. venous thromboembolism.mp. or exp Venous Thromboembolism/ 28,924

132. deep ve* thrombosis.mp. 31,151

133. pulmonary embolism.mp. or exp Pulmonary Embolism/ 59,166

134. HIV.mp. or exp HIV Infections/ 431,237

135. AIDS.mp. or exp Acquired Immunodeficiency Syndrome/ 231,823

136. exp Immunocompromised Host/ 27,296

137. exp Immunosuppression Therapy/ or exp Immunosuppressive Agents/ 386,330

138. immunosuppress*.mp. 247,166

139. exp Transplantation/ 558,640

140. transplant*.mp. 821,998

141. exp Alcohol-Related Disorders/ 119,529

142. (alcohol misuse or alcohol abuse or alcohol dependence or alcoholism).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 102,278

143. exp Substance-Related Disorders/ 303,321

144. (substance misuse or substance abuse or substance dependence).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 61,758

145. exp Anxiety/ or exp Anxiety Disorders/ 179,995

146. anxiety.mp. 287,258

147. panic disorder.mp. or exp Panic Disorder/ 11,686

148. phobic disorder.mp. or exp Phobic Disorders/ 12,239

149. phobia.mp. 9375

150. exp Stress Disorders, Post-Traumatic/ 39,160

151. (post traumatic stress disorder or post-traumatic stress disorder or PTSD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 36,261

152. exp Mood Disorders/ 132,929

153. exp Depression/ or exp Depression, Postpartum/ 148,840

154. (depression or depressive disorder or mood disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 504,128

155. dementia.mp. or exp Dementia/ 245,230

156. eating disorder.mp. or exp “Feeding and Eating Disorders”/ 38,341

157. (anorexia nervosa or bulimia nervosa).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 21,280

158. bipolar disorder.mp. or exp Bipolar Disorder/ 54,684

159. exp “schizophrenia spectrum and other psychotic disorders”/ 159,357

160. (schizophrenia or psychosis or psychotic disorder or schizoaffective disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 182,203

161. dissociative disorder.mp. or exp Dissociative Disorders/ 4820

162. obsessive compulsive disorder.mp. or exp Obsessive-Compulsive Disorder/ 21,228

163. personality disorder.mp. or exp Personality Disorders/ 49,154

164. self harm.mp. or exp Self-Injurious Behavior/ 83,262

165. exp Mental Disorders/ 1,386,917

166. (mental disorder or psychiatric disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 21,591

167. (serious mental illness or severe mental illness).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 9002

168. exp Attention Deficit Disorder with Hyperactivity/ 33,335

169. (attention deficit hyperactivity disorder or ADHD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 36,926

170. exp Autism Spectrum Disorder/ 39,314

171. (autism or autistic).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 64,227

172. cerebral palsy.mp. or exp Cerebral Palsy/ 30,646

173. intellectual disabilit*.mp. or exp Intellectual Disability/ 113,325

174. exp Down Syndrome/ or down* syndrome.mp. 32,403

175. exp Brain Injuries/ or acquired brain injury.mp. 79,525

176. exp Headache Disorders/ 38,752

177. (cluster headache or tension headache or chronic headache or migraine).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 47,340

178. exp Epilepsy/ 122,463

179. (epilepsy or epileptic).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 167,049

180. idiopathic intracranial hypertension.mp. or exp Pseudotumor Cerebri/ 5202

181. multiple sclerosis.mp. or exp Multiple Sclerosis/ 94,760

182. peripheral neuropathy.mp. or exp Peripheral Nervous System Diseases/ 171,846

183. exp Parkinson Disease/ or parkinson* disease.mp. 126,506

184. exp Neurodegenerative Diseases/ 351,658

185. (huntington* disease or huntington* chorea).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 20,268

186. motor neurone disease.mp. 1091

187. exp Sleep Wake Disorders/ 104,157

188. (sleep disorder or narcolepsy or obstructive sleep apn?ea).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 41,903

189. spina bifida.mp. or exp Spinal Dysraphism/ 12,050

190. exp Fatigue Syndrome, Chronic/ 6142

191. (chronic fatigue syndrome or myalgic encephalomyelitis).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 6471

192. fibromyalgia.mp. or exp Fibromyalgia/ 13,310

193. exp Chronic Pain/ 20,443

194. exp Complex Regional Pain Syndromes/ 5884

195. exp Myofascial Pain Syndromes/ 6762

196. (chronic pain or pain syndrome).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 66,066

197. exp Back Pain/ or chronic back pain.mp. 44,337

198. osteoarthritis.mp. or exp Osteoarthritis/ 104,568

199. exp Osteoporosis/ 60,974

200. (osteoporosis or osteopenia).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 101,668

201. scoliosis.mp. or exp Scoliosis/ 28,225

202. exp Spinal Diseases/ 136,461

203. exp Fractures, Compression/ 2943

204. (compression fracture or collapsed vertebra*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 2462

205. intervertebral disc degeneration.mp. or exp Intervertebral Disc Degeneration/ 8378

206. sciatica.mp. or exp Sciatica/ 7190

207. spinal stenosis.mp. or exp Spinal Stenosis/ 9544

208. spondylosis.mp. or exp Spondylosis/ 11,155

209. spondylolisthesis.mp. or exp Spondylolisthesis/ 7445

210. amputation.mp. or exp Amputation/ 52,172

211. amputee.mp. 2992

212. paralysis.mp. or exp Paralysis/ 116,571

213. exp Hemiplegia/ or hemiplegia.mp. 16,538

214. exp Paraplegia/ or paraplegia.mp. 22,118

215. quadriplegia.mp. or exp Quadriplegia/ 10,093

216. exp Disabled Persons/ 71,645

217. (disabled or disabilit*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 362,858

218. chronic kidney disease.mp. or exp Renal Insufficiency, Chronic/ 162,815

219. h?emodialysis.mp. or exp Renal Dialysis/ 146,472

220. exp Urinary Calculi/ 37,823

221. (urinary tract stone* or kidney stone*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 7369

222. exp Asthma/ or asthma.mp. 191,929

223. exp Lung Diseases/ or chronic lung disease.mp. 1,147,038

224. bronchiectasis.mp. or exp Bronchiectasis/ 15,082

225. exp Pulmonary Disease, Chronic Obstructive/ 64,208

226. (chronic obstructive pulmonary disease or COPD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 75,276

227. cystic fibrosis.mp. or exp Cystic Fibrosis/ 55,910

228. interstitial lung disease.mp. or exp Lung Diseases, Interstitial/ 87,153

229. pulmonary fibrosis.mp. 34,100

230. pulmonary hypertension.mp. or exp Hypertension, Pulmonary/ 56,630

231. exp Sarcoidosis/ or sarcoidosis.mp. 33,019

232. exp Tuberculosis/ or tuberculosis.mp. 272,422

233. exp Ehlers-Danlos Syndrome/ or ehlers-danlos.mp. 4661

234. rheumatoid arthritis.mp. or exp Arthritis, Rheumatoid/ 160,931

235. exp Sjogren’s Syndrome/ 14,126

236. (sjogren* syndrome or sjogren* disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 19,617

237. exp Raynaud Disease/ or raynaud*.mp.  10,156

238. systemic sclerosis.mp. or exp Scleroderma, Systemic/ 26,894

239. scleroderma.mp. 29,230

240. primary systemic vasculitis.mp. or exp Systemic Vasculitis/ 18,109

241. marfan* syndrome.mp. or exp Marfan Syndrome/ 8617

242. spondyloarthritis.mp. or exp Spondylarthritis/ 31,153

243. psoriatic arthritis.mp. or exp Arthritis, Psoriatic/ 12,337

244. ankylosing spondylitis.mp. or exp Spondylitis, Ankylosing/ 21,066

245. systemic lupus erythematosus.mp. or exp Lupus Erythematosus, Systemic/ 79,628

246. autoimmune disease.mp. or exp Autoimmune Diseases/ 540,341

247. frailty.mp. or exp Frailty/ 22,139

248. exp COVID-19/ or long covid.mp. 181,547

249. post COVID syndrome.mp. 213

250. exp Obesity/ 247,988

251. (obese or obesity).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 411,960

252. polypharmacy.mp. or exp Polypharmacy/ 12,571

253. turner* syndrome.mp. or exp Turner Syndrome/ 9685

254. 21 or 22 or 23 or 24 or 25 or 26 or 27 or 28 or 29 or 30 or 31 or 32 or 33 or 34 or 35 or 36 or 37 or 38 or 39 or 40 or 41 or 42 or 43 or 44 or 45 or 46 or 47 or 48 or 49 or 50 or 51 or 52 or 53 or 54 or 55 or 56 or 57 or 58 or 59 or 60 or 61 or 62 or 63 or 64 or 65 or 66 or 67 or 68 or 69 or 70 or 71 or 72 or 73 or 74 or 75 or 76 or 77 or 78 or 79 or 80 or 81 or 82 or 83 or 84 or 85 or 86 or 87 or 88 or 89 or 90 or 91 or 92 or 93 or 94 or 95 or 96 or 97 or 98 or 99 or 100 or 101 or 102 or 103 or 104 or 105 or 106 or 107 or 108 or 109 or 110 or 111 or 112 or 113 or 114 or 115 or 116 or 117 or 118 or 119 or 120 or 121 or 122 or 123 or 124 or 125 or 126 or 127 or 128 or 129 or 130 or 131 or 132 or 133 or 134 or 135 or 136 or 137 or 138 or 139 or 140 or 141 or 142 or 143 or 144 or 145 or 146 or 147 or 148 or 149 or 150 or 151 or 152 or 153 or 154 or 155 or 156 or 157 or 158 or 159 or 160 or 161 or 162 or 163 or 164 or 165 or 166 or 167 or 168 or 169 or 170 or 171 or 172 or 173 or 174 or 175 or 176 or 177 or 178 or 179 or 180 or 181 or 182 or 183 or 184 or 185 or 186 or 187 or 188 or 189 or 190 or 191 or 192 or 193 or 194 or 195 or 196 or 197 or 198 or 199 or 200 or 201 or 202 or 203 or 204 or 205 or 206 or 207 or 208 or 209 or 210 or 211 or 212 or 213 or 214 or 215 or 216 or 217 or 218 or 219 or 220 or 221 or 222 or 223 or 224 or 225 or 226 or 227 or 228 or 229 or 230 or 231 or 232 or 233 or 234 or 235 or 236 or 237 or 238 or 239 or 240 or 241 or 242 or 243 or 244 or 245 or 246 or 247 or 248 or 249 or 250 or 251 or 252 or 253 14,314,773

255. randomized controlled trial.pt. 575,118

256. controlled clinical trial.pt. 94,989

257. randomi#ed.ab. 684,071

258. placebo.ab. 230,860

259. drug therapy.fs. 2,521,208

260. randomly.ab. 389,335

261. trial.ab. 612,686

262. groups.ab. 2,394,830

263. 255 or 256 or 257 or 258 or 259 or 260 or 261 or 262 5,455,224

264. exp animals/ not humans.sh. 5,037,553

265. 263 not 264 4,753,806

266. 9 and 20 and 254 and 265 1144

MEDLINE search strategy for LTCs mixed-methods systematic review (see Chapter 7)

Ovid MEDLINE^® ALL 1946 to 23 November 2022

n = 2187, searched on 24 November 2022

1. exp Breast Feeding/ 42,954

2. (breastfeed* or breast-feed* or breast feed*).ab. 39,367

3. (breastfed or breast-fed or breast fed).ab. 13,012

4. lactation.ab. 35,775

5. infant feed*.ab. 4996

6. exp Lactation/ 46,965

7. exp Breast Milk Expression/ 385

8. exp Milk, Human/ 22,053

9. 1 or 2 or 3 or 4 or 5 or 6 or 7 or 8 130,890

10. ((support* or help or assist* or class* or workshop* or champion* or promot*) adj5 (breastfeed* or breast feed* or breastfed or breast fed or lactation or infant feed*)).ab. 7474

11. exp Social Support/ 78,607

12. anticipatory guidance.mp. 1545

13. exp Counseling/ 48,142

14. counsel*.mp. 154,556

15. exp Directive Counseling/ 4882

16. exp Health Promotion/ 84,136

17. exp Health Education/ 260,144

18. peer support.mp. 6522

19. professional support.mp. 2070

20. 10 or 11 or 12 or 13 or 14 or 15 or 16 or 17 or 18 or 19 483,065

21. exp Chronic Disease/ 602,943

22. chronic disease*.mp. 342,009

23. chronic illness*.mp. 19,105

24. chronic condition*.mp. 24,060

25. (long term condition* or long-term condition*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 2474

26. exp Comorbidity/ 125,267

27. (comorbid* or co-morbid*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 312,919

28. exp Multimorbidity/ 2454

29. (multimorbid* or multi-morbid*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 9346

30. (multidiseas* or multi-diseas*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 278

31. cancer.mp. or exp Neoplasms/ 4,329,926

32. atrial fibrillation.mp. or exp Atrial Fibrillation/ 100,464

33. cardiomyopathy.mp. or exp Cardiomyopathies/ 143,152

34. heart failure.mp. or exp Heart Failure/ 245,181

35. exp Hypercholesterolemia/ or exp Hyperlipidemias/ 69,913

36. (hypercholesterol?emia or hyperlipid?emia).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 76,793

37. exp Hypertension/or hypertension.mp. 547,959

38. exp Myocardial Ischemia/ 464,415

39. (isch?emic heart disease or myocardial infarction).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 299,852

40. peripheral vascular disease.mp. or exp Peripheral Vascular Diseases/ 65,062

41. exp Stroke/ or stroke.mp. 378,102

42. exp Ischemic Attack, Transient/ 21,618

43. transient isch?emic attack.mp. 12,655

44. congenital heart disease.mp. or exp Heart Defects, Congenital/ 177,949

45. valvular heart disease.mp. or exp Heart Valve Diseases/ 136,981

46. rheumatic heart disease.mp. or exp Rheumatic Heart Disease/ 15,031

47. exp Heart Diseases/ 1,245,391

48. (heart disease or cardiac disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 214,566

49. alopecia areata.mp. or exp Alopecia Areata/ 5456

50. vitiligo.mp. or exp Vitiligo/ 8980

51. exp Eczema/ or eczema.mp. 24,134

52. psoriasis.mp. or exp Psoriasis/ 60,070

53. acne.mp. or exp Acne Vulgaris/ 21,180

54. hidradenitis suppurativa.mp. or exp Hidradenitis Suppurativa/ 3951

55. lichen planus.mp. or exp Lichen Planus/ 11,203

56. rosacea.mp. or exp Rosacea/ 4729

57. seborrheic dermatitis.mp. or exp Dermatitis, Seborrheic/ 3431

58. allergic rhinitis.mp. or exp Rhinitis, Allergic/ 31,617

59. allergic conjunctivitis.mp. or exp Conjunctivitis, Allergic/ 4493

60. exp Hearing Loss/ 76,292

61. (hearing loss or deaf*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 112,157

62. exp Addison Disease/ or addison* disease.mp. 6034

63. exp Adrenocortical Adenoma/ or adren* adenoma.mp. 3177

64. 64exp Pheochromocytoma/ 16,529

65. ph?eochromocytoma.mp. 23,936

66. exp Cushing Syndrome/ or cushing* syndrome.mp. 15,980

67. exp Diabetes Mellitus, Type 2/ or exp Diabetes, Gestational/ or exp Diabetes Mellitus/ or exp Diabetes Mellitus, Type 1/ 492,036

68. (diabetes or diabetic$1).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 809,517

69. exp Parathyroid Diseases/ or (parathyroid dis* or hyperparathyroid* or hypoparathyroid*).mp. 46,799

70. exp Thyroid Diseases/ or thyroid dis*.mp. 166,479

71. (hyperthyroid* or hypothyroid* or thyroiditis or graves disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 100,991

72. exp Pituitary Diseases/ or pituitary dis*.mp. 65,344

73. exp Endocrine System Diseases/ 1,099,839

74. exp Vision Disorders/ 77,726

75. (visual* impair* or blindness).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 59,580

76. exp Cataract/ or cataract.mp. 71,946

77. exp Diabetic Retinopathy/ 28,637

78. (diabetic retinopathy or diabetic eye dis*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 39,000

79. glaucoma.mp. or exp Glaucoma/ 79,199

80. scleritis.mp. or exp Scleritis/ 2393

81. episcleritis.mp. 624

82. exp Uveitis/ or uveitis.mp. 41,131

83. retinal detachment.mp. or exp Retinal Detachment/ 28,930

84. exp Eye Diseases/ 624,904

85. alcoholic liver disease.mp. or exp Liver Diseases, Alcoholic/ 18,612

86. autoimmune hepatitis.mp. or exp Hepatitis, Autoimmune/ 7336

87. sclerosing cholangitis.mp. or exp Cholangitis, Sclerosing/ 7900

88. primary biliary cirrhosis.mp. or exp Liver Cirrhosis, Biliary/ 10,999

89. chronic hepatitis.mp. or exp Hepatitis, Chronic/ 73,739

90. exp Hepatitis B, Chronic/ or hepatitis B.mp. 109,029

91. exp Hepatitis C, Chronic/ or hepatitis C.mp. 99,589

92. liver cirrhosis.mp. or exp Liver Cirrhosis/ 113,060

93. non-alcoholic fatty liver disease.mp. or exp Non-alcoholic Fatty Liver Disease/ 27,321

94. exp Liver Diseases/ 613,804

95. chronic pancreatitis.mp. or exp Pancreatitis, Chronic/ 17,127

96. c?eliac disease.mp. or exp Celiac Disease/ 26,440

97. food allergy.mp. or exp Food Hypersensitivity/ 26,232

98. cholelithiasis.mp. or exp Cholelithiasis/ 40,302

99. gallstones.mp. 20,545

100. inflammatory bowel disease.mp. or exp Inflammatory Bowel Diseases/ 114,088

101. exp Crohn Disease/ or crohn* disease.mp. 62,863

102. ulcerative colitis.mp. or exp Colitis, Ulcerative/ 56,412

103. proctitis.mp. or exp Proctitis/ 4741

104. irritable bowel syndrome.mp. or exp Irritable Bowel Syndrome/ 16,860

105. lactose intolerance.mp. or exp Lactose Intolerance/ 3661

106. peptic ulcer.mp. or exp Peptic Ulcer/ 88,964

107. chronic pelvic inflammatory dis*.mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 135

108. exp Pelvic Inflammatory Disease/ 11,423

109. dysmenorrhea.mp. or exp Dysmenorrhea/ 7392

110. endometriosis.mp. or exp Endometriosis/ 31,997

111. infertility.mp. or exp Infertility/ 104,859

112. assisted reproduction.mp. or exp Reproductive Techniques, Assisted/ 81,518

113. (in vitro fertili#ation or IVF).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 40,612

114. leiomyoma.mp. or exp Leiomyoma/ 25,551

115. fibroids.mp. 5762

116. exp Menopause/ or menopause.mp. 75,624

117. menorrhagia.mp. or exp Menorrhagia/ 6342

118. exp Urinary Incontinence/ or exp Pelvic Floor Disorders/ or exp Pelvic Organ Prolapse/ or exp Fecal Incontinence/ 55,156

119. (pelvic floor dysfunction or pelvic floor disorder*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 3030

120. (urinary incontinence or f?ecal incontinence).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 54,896

121. pelvic organ prolapse.mp. 7917

122. exp Polycystic Ovary Syndrome/ or polycystic ovar* syndrome.mp. 22,165

123. recurrent miscarriage.mp. or exp Abortion, Habitual/ 9737

124. exp Blood Coagulation Disorders/ or coagulation disorder.mp. 104,538

125. h?emophilia.mp. 29,136

126. exp Anemia, Sickle Cell/ or sickle cell.mp. 32,225

127. thalass?emia.mp. or exp Thalassemia/ 30,381

128. thrombophilia.mp. or exp Thrombophilia/ 30,589

129. pernicious an?emia.mp. or exp Anemia, Pernicious/ 7018

130. exp Thrombocytopenia/ or primary thrombocytopenia.mp. 52,629

131. venous thromboembolism.mp. or exp Venous Thromboembolism/ 29,575

132. deep ve* thrombosis.mp. 31,577

133. pulmonary embolism.mp. or exp Pulmonary Embolism/ 59,857

134. HIV.mp. or exp HIV Infections/ 434,875

135. AIDS.mp. or exp Acquired Immunodeficiency Syndrome/ 233,377

136. exp Immunocompromised Host/ 27,383

137. exp Immunosuppression Therapy/ or exp Immunosuppressive Agents/ 388,928

138. immunosuppress*.mp. 250,351

139. exp Transplantation/ 562,203

140. transplant*.mp. 829,484

141. exp Alcohol-Related Disorders/ 120,079

142. (alcohol misuse or alcohol abuse or alcohol dependence or alcoholism).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 102,885

143. exp Substance-Related Disorders/ 305,479

144. (substance misuse or substance abuse or substance dependence).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 62,218

145. exp Anxiety/ or exp Anxiety Disorders/ 182,212

146. anxiety.mp. 293,708

147. panic disorder.mp. or exp Panic Disorder/ 11,740

148. phobic disorder.mp. or exp Phobic Disorders/ 12,318

149. phobia.mp. 9483

150. exp Stress Disorders, Post-Traumatic/ 39,810

151. (post traumatic stress disorder or post-traumatic stress disorder or PTSD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 37,152

152. exp Mood Disorders/ 133,816

153. exp Depression/ or exp Depression, Postpartum/ 151,065

154. (depression or depressive disorder or mood disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 512,446

155. dementia.mp. or exp Dementia/ 249,440

156. eating disorder.mp. or exp “Feeding and Eating Disorders”/ 38,866

157. (anorexia nervosa or bulimia nervosa).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 21,488

158. bipolar disorder.mp. or exp Bipolar Disorder/ 55,305

159. exp “schizophrenia spectrum and other psychotic disorders”/ 160,377

160. (schizophrenia or psychosis or psychotic disorder or schizoaffective disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 184,089

161. dissociative disorder.mp. or exp Dissociative Disorders/ 4851

162. obsessive compulsive disorder.mp. or exp Obsessive-Compulsive Disorder/ 21,507

163. personality disorder.mp. or exp Personality Disorders/ 49,545

164. self harm.mp. or exp Self-Injurious Behavior/ 84,338

165. exp Mental Disorders/ 1,400,314

166. (mental disorder or psychiatric disorder).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 22,024

167. (serious mental illness or severe mental illness).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 9183

168. exp Attention Deficit Disorder with Hyperactivity/ 33,703

169. (attention deficit hyperactivity disorder or ADHD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 37,625

170. exp Autism Spectrum Disorder/ 40,143

171. (autism or autistic).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 65,738

172. cerebral palsy.mp. or exp Cerebral Palsy/ 31,036

173. intellectual disabilit*.mp. or exp Intellectual Disability/ 114,299

174. exp Down Syndrome/ or down* syndrome.mp. 32,674

175. exp Brain Injuries/ or acquired brain injury.mp. 80,562

176. exp Headache Disorders/ 39,158

177. (cluster headache or tension headache or chronic headache or migraine).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 47,919

178. exp Epilepsy/ 123,445

179. (epilepsy or epileptic).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 168,992

180. idiopathic intracranial hypertension.mp. or exp Pseudotumor Cerebri/ 5277

181. multiple sclerosis.mp. or exp Multiple Sclerosis/ 96,126

182. peripheral neuropathy.mp. or exp Peripheral Nervous System Diseases/ 173,401

183. exp Parkinson Disease/ or parkinson* disease.mp. 128,663

184. exp Neurodegenerative Diseases/ 356,554

185. (huntington* disease or huntington* chorea).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 20,536

186. motor neurone disease.mp. 1097

187. exp Sleep Wake Disorders/ 105,644

188. (sleep disorder or narcolepsy or obstructive sleep apn?ea).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 42,731

189. spina bifida.mp. or exp Spinal Dysraphism/ 12,140

190. exp Fatigue Syndrome, Chronic/ 6267

191. (chronic fatigue syndrome or myalgic encephalomyelitis).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 6552

192. fibromyalgia.mp. or exp Fibromyalgia/ 13,499

193. exp Chronic Pain/ 21,039

194. exp Complex Regional Pain Syndromes/ 5902

195. exp Myofascial Pain Syndromes/ 6785

196. (chronic pain or pain syndrome).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 67,401

197. exp Back Pain/ or chronic back pain.mp. 44,796

198. osteoarthritis.mp. or exp Osteoarthritis/ 106,483

199. exp Osteoporosis/ 61,530

200. (osteoporosis or osteopenia).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 102,970

201. scoliosis.mp. or exp Scoliosis/ 28,584

202. exp Spinal Diseases/ 137,686

203. exp Fractures, Compression/ 3022

204. (compression fracture or collapsed vertebra*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 2523

205. intervertebral disc degeneration.mp. or exp Intervertebral Disc Degeneration/ 8566

206. sciatica.mp. or exp Sciatica/ 7238

207. spinal stenosis.mp. or exp Spinal Stenosis/ 9721

208. spondylosis.mp. or exp Spondylosis/ 11,295

209. spondylolisthesis.mp. or exp Spondylolisthesis/ 7552

210. amputation.mp. or exp Amputation/ 52,820

211. amputee.mp. 3034

212. paralysis.mp. or exp Paralysis/ 117,463

213. exp Hemiplegia/ or hemiplegia.mp. 16,665

214. exp Paraplegia/ or paraplegia.mp. 22,286

215. quadriplegia.mp. or exp Quadriplegia/ 10,148

216. exp Disabled Persons/ 72,228

217. (disabled or disabilit*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 368,071

218. chronic kidney disease.mp. or exp Renal Insufficiency, Chronic/ 165,323

219. h?emodialysis.mp. or exp Renal Dialysis/ 147,868

220. exp Urinary Calculi/ 38,075

221. (urinary tract stone* or kidney stone*).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 7526

222. exp Asthma/ or asthma.mp. 193,826

223. exp Lung Diseases/ or chronic lung disease.mp. 1,172,810

224. bronchiectasis.mp. or exp Bronchiectasis/ 15,266

225. exp Pulmonary Disease, Chronic Obstructive/ 65,000

226. (chronic obstructive pulmonary disease or COPD).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 76,565

227. cystic fibrosis.mp. or exp Cystic Fibrosis/ 56,480

228. interstitial lung disease.mp. or exp Lung Diseases, Interstitial/ 88,055

229. pulmonary fibrosis.mp. 34,669

230. pulmonary hypertension.mp. or exp Hypertension, Pulmonary/ 57,397

231. exp Sarcoidosis/ or sarcoidosis.mp. 33,257

232. exp Tuberculosis/ or tuberculosis.mp. 274,528

233. exp Ehlers-Danlos Syndrome/ or ehlers-danlos.mp. 4735

234. rheumatoid arthritis.mp. or exp Arthritis, Rheumatoid/ 162,479

235. exp Sjogren’s Syndrome/ 14,277

236. (sjogren* syndrome or sjogren* disease).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 19,871

237. exp Raynaud Disease/ or raynaud*.mp. 10,222

238. systemic sclerosis.mp. or exp Scleroderma, Systemic/ 27,173

239. scleroderma.mp. 29,499

240. primary systemic vasculitis.mp. or exp Systemic Vasculitis/ 18,247

241. marfan* syndrome.mp. or exp Marfan Syndrome/ 8688

242. spondyloarthritis.mp. or exp Spondylarthritis/ 31,559

243. psoriatic arthritis.mp. or exp Arthritis, Psoriatic/ 12,555

244. ankylosing spondylitis.mp. or exp Spondylitis, Ankylosing/ 21,291

245. systemic lupus erythematosus.mp. or exp Lupus Erythematosus, Systemic/ 80,550

246. autoimmune disease.mp. or exp Autoimmune Diseases/ 545,468

247. frailty.mp. or exp Frailty/ 23,160

248. exp COVID-19/ or long covid.mp. 199,512

249. post COVID syndrome.mp. 288

250. exp Obesity/ 251,605

251. (obese or obesity).mp. [mp=title, book title, abstract, original title, name of substance word, subject heading word, floating sub-heading word, keyword heading word, organism supplementary concept word, protocol supplementary concept word, rare disease supplementary concept word, unique identifier, synonyms] 419,142

252. polypharmacy.mp. or exp Polypharmacy/ 12,907

253. turner* syndrome.mp. or exp Turner Syndrome/ 9757

254. 21 or 22 or 23 or 24 or 25 or 26 or 27 or 28 or 29 or 30 or 31 or 32 or 33 or 34 or 35 or 36 or 37 or 38 or 39 or 40 or 41 or 42 or 43 or 44 or 45 or 46 or 47 or 48 or 49 or 50 or 51 or 52 or 53 or 54 or 55 or 56 or 57 or 58 or 59 or 60 or 61 or 62 or 63 or 64 or 65 or 66 or 67 or 68 or 69 or 70 or 71 or 72 or 73 or 74 or 75 or 76 or 77 or 78 or 79 or 80 or 81 or 82 or 83 or 84 or 85 or 86 or 87 or 88 or 89 or 90 or 91 or 92 or 93 or 94 or 95 or 96 or 97 or 98 or 99 or 100 or 101 or 102 or 103 or 104 or 105 or 106 or 107 or 108 or 109 or 110 or 111 or 112 or 113 or 114 or 115 or 116 or 117 or 118 or 119 or 120 or 121 or 122 or 123 or 124 or 125 or 126 or 127 or 128 or 129 or 130 or 131 or 132 or 133 or 134 or 135 or 136 or 137 or 138 or 139 or 140 or 141 or 142 or 143 or 144 or 145 or 146 or 147 or 148 or 149 or 150 or 151 or 152 or 153 or 154 or 155 or 156 or 157 or 158 or 159 or 160 or 161 or 162 or 163 or 164 or 165 or 166 or 167 or 168 or 169 or 170 or 171 or 172 or 173 or 174 or 175 or 176 or 177 or 178 or 179 or 180 or 181 or 182 or 183 or 184 or 185 or 186 or 187 or 188 or 189 or 190 or 191 or 192 or 193 or 194 or 195 or 196 or 197 or 198 or 199 or 200 or 201 or 202 or 203 or 204 or 205 or 206 or 207 or 208 or 209 or 210 or 211 or 212 or 213 or 214 or 215 or 216 or 217 or 218 or 219 or 220 or 221 or 222 or 223 or 224 or 225 or 226 or 227 or 228 or 229 or 230 or 231 or 232 or 233 or 234 or 235 or 236 or 237 or 238 or 239 or 240 or 241 or 242 or 243 or 244 or 245 or 246 or 247 or 248 or 249 or 250 or 251 or 252 or 253 14,494,082

255. (questionnaire* or survey* or interview* or focus group* or case stud* or observ* or ethnograph* or hermeneutic* or narrative* or phenomenolog* or grounded theory or process evaluation or implementation study or implementation research or view* or experienc* or opinion* or attitude* or perception* or perceive* or belie* or feel* or know* or understand* or barrier* or facilitator* or enabler* or obstacle*).tw. 9,977,739

256. exp Qualitative Research/ or (qualitative or mixed method*).tw. 332,065

257. 255 or 256 10,059,034

258. 9 and 20 and 254 and 257 2187

MEDLINE search strategy for LTCs economic evidence review (see Chapter 8)

Ovid MEDLINE^® ALL 1946 to 17 August 2022

n = 3077, searched on 17 August 2022

1. exp Breast Feeding/

2. breastfeed*.mp.

3. breastfed.mp.

4. breast-feed*.mp.

5. breast-fed.mp.

6. breast feed*.mp.

7. breast fed.mp.

8. infant feed*.mp.

9. exp Milk, Human/

10. Lactation/

11. lactat*.mp.

12. support.mp.

13. Social Support/

14. advice.mp.

15. advis*.mp.

16. help*.mp.

17. supportive adj2 relationship.mp

18. counsel*.mp.

19. educat*.mp.

20. consult*.mp.

21. Health Promotion/

22. Health Education/

23. Economics/

24. exp “Costs and cost analysis”/

25. “Cost allocation”/

26. Cost-benefit analysis/

27. “Cost control”/

28. “Cost savings”/

29. “Cost of illness”/

30. “Cost sharing”/

31. “deductibles and coinsurance”/

32. Medical savings accounts/

33. Health care costs/

34. Direct service costs/

35. Drug costs/

36. Employer health costs/

37. Hospital costs/

38. Health expenditures/

39. Capital expenditures/

40. Value of life/

41. exp economics, hospital/

42. exp economics, medical/

43. Economics, nursing/

44. Economics, pharmaceutical/

45. exp “fees and charges”/

46. exp budgets/

47. (low adj cost).mp.

48. (high adj cost).mp.

49. (health?care adj cost$).mp.

50. (fiscal or funding or financial or finance).tw.

51. (cost adj estimate$).mp.

52. (cost adj variable).mp.

53. (unit adj cost$).mp.

54. (economic$ or pharmacoeconomic$ or price$ or pricing).tw.

55. or/1-11

56. or/12-22

57. or/23-54

58. 55 and 56 and 57

59. exp animals/ not humans.sh.

60. 58 not 59