Cue-based versus scheduled feeding for preterm infants transitioning from tube to oral feeding: the Cubs mixed-methods feasibility study

Article history

The research reported in this issue of the journal was funded by the HTA programme as project number 16/144/05. The contractual start date was in May 2020. The draft report began editorial review in December 2020 and was accepted for publication in July 2021. The authors have been wholly responsible for all data collection, analysis and interpretation, and for writing up their work. The HTA editors and publisher have tried to ensure the accuracy of the authors’ report 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 report.

Disclaimers

This report contains transcripts of interviews conducted in the course of the research and contains language that may offend some readers.

Copyright statement

Copyright © 2021 McFadden et al. This work was produced by McFadden 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 adaption 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.

2021 McFadden et al.

Background and context

The frequency of feeding and volume of milk intake of healthy term-born infants is generally dictated by the infant’s appetite. Term infants exhibit feeding and satiation cues and adjust their volume of intake to compensate for differences in the nutrient density of various milks. 1 In contrast, enteral feeds for preterm infants are usually given as prescribed volumes at scheduled intervals. 2 However, there is some evidence that preterm infants can self-regulate their intake. 3 Furthermore, while feeding cues may be more difficult to detect in preterm infants, they may be sufficiently evident for a parent or caregiver to recognise and respond to,4 thereby supporting safer and more successful feeding experiences. Caregivers and parents can use infants’ physiological and behavioural channels of communication to inform their feeding decisions and actions. This may also set the scene for future feeding practices and success, as well as parent–infant interaction. Although studies have shown that cue-based (also known as responsive) feeding is feasible for preterm infants, the adoption of cue-based feeding has been constrained by the ‘schedule- and volume-driven culture’ in many neonatal units (NNUs). 5

Alternatives to scheduled interval feeding regimens that aim to respond to infant feed cues have been described. 6 These are relevant to the transition from gastric tube feeding to oral feeding,7 when preterm infants are developing periods of sustained alert activity and a suck–swallow–breathe pattern8 sufficient for oral feeding to commence. 1

Cue-based feeding is a co-regulated approach. 6 The enteral feeding process starts when the caregiver recognises infant cues that indicate readiness to feed and ends when the infant demonstrates satiation. The infant, therefore, determines the timing, duration and volume of intake. At each stage during transition to oral feeding, through understanding and interpretation of their cues, infants are supported in such a way that they can achieve all they are capable of with regards to oral feeding. Cue-based feeding occurs alongside supplementary tube feeding with the understanding that, developmentally, many preterm infants are not yet ready to fully sustain themselves by oral feeding. In modifications of cue-based feeding, caregivers may pre-set a maximum permitted duration of inactivity or sleep between feeds or a maximum volume of intake or modify feeding plans to consider the reduced endurance levels of preterm infants.

The transition to oral feeding is a critical developmental stage for preterm infants. Cue-based feeding may be considered a part of an integrated approach to providing ‘developmental care’ when infants are seen as individuals and caregivers are guided by the needs and behaviours of the infant. 9

Cue-based feeding may also increase rest between feeds promoting infant-led sleep and wake patterns. Potentially, infant-led feeding patterns will facilitate development of organised behaviour states leading to earlier establishment of oral feeding and thereby shortening hospital stay for preterm infants. 1

Reducing length of hospital stay has a direct effect on hospital costs and may also decrease cot occupancy in NNUs, thus reducing the need for inter-hospital transfer of women and infants. 10 Compared with a scheduled approach, cue-based feeding may support infant stability during oral feeds as infants’ cues will be responded to, resulting in fewer episodes of physiological instability, which could cause significant harm (e.g. aspiration, desaturation and bradycardia events). 11 As feeding an infant is a primary activity over the first year of life and a major preoccupation of parents, it is anticipated that there may be other benefits for the family and caregivers of cue-based feeding, principally allowing parents to feel more directly involved with their infant’s care and better understand their infant’s communication, together with increasing parental confidence and ability to recognise and respond to their infant’s needs during their hospital stay and beyond. Enhanced parental satisfaction is a key quality indicator in measuring the effectiveness of family-integrated care in neonatal services. 12

Potential adverse effects of cue-based feeding for preterm infants are recognised. These mainly relate to whether or not such a regimen can guarantee metabolic stability, particularly normoglycaemia, in this vulnerable group. Even at the point of discharge home from hospital, some preterm infants are known to be susceptible to hypoglycaemia if a scheduled enteral feed is omitted or delayed. 13 Concern exists that repeated or prolonged episodes of hypoglycaemia may impair longer-term growth and development. 14 There may be more acute problems relating to gastrointestinal immaturity, such as feeding intolerance and a higher risk of aspiration of gastric contents into the lungs, as well as concerns that allowing unrestrained volumes of enteral intake may increase the risk of gastro-oesophageal reflux or feed intolerance. However, there is a lack of evidence of these potential adverse effects and, indeed, some problems may be exacerbated by not responding to the infant’s cues. 11 Despite these concerns, and despite a lack of evidence of benefit, cue-based feeding is established in some NNUs in other countries (e.g. Sweden15,16 and the USA17,18) and is increasingly being used in NNUs in the UK. 19 Cue-based feeding for preterm infants is now recommended as a method to increase the duration of breastfeeding in the United Nations Children’s Fund (UNICEF) Baby Friendly Initiative (BFI) ‘Ten steps to successful breastfeeding’. 20

Overall, the evidence to support cue-based feeding is limited. A recent Cochrane review1 concluded that there was low-quality evidence that cue-based feeding compared with scheduled feeding leads to earlier transition to full oral feeding. The review authors noted that this evidence should be treated with caution owing to several methodological weaknesses. 1 Furthermore, there is a lack of strong or consistent evidence of the effect of cue-based feeding compared with scheduled feeding on important outcomes for preterm infants or their families. 1 Therefore, there is a need for rigorous evaluation of cue-based feeding for preterm infants within the NHS setting, based on the most up-to-date and complete evidence, and considering stakeholders’ (including parents’) views. The first step towards this is to assess if such an intervention trial is justifiable and feasible.

Research objectives

The overall aim of this study was to develop a manualised intervention and to assess whether or not it is feasible to conduct a clinical and cost-effectiveness study of cue-based compared with scheduled feeding for preterm infants in NNUs.

The study had eight research objectives:

1. to describe the characteristics, components, theoretical basis and outcomes of approaches to feeding preterm infants transitioning from tube to oral feeding, including by feeding type (breast milk, donor breast milk, formula, combined) and method (breastfeeding, bottle feeding)

2. to identify operational policies, barriers and facilitators, and staff and parents’ education needs in NNUs implementing cue-based feeding

3. to co-produce an evidence-informed, adaptable, manualised intervention, including staff and parent educational support for feeding preterm infants at the transition from tube to oral feeding in response to feeding cues and signs of infant stability

4. to appraise the willingness of parents and staff to implement and sustain the intervention

5. to assess the associated costs of implementing cue-based feeding in NNUs

6. to determine the feasibility and acceptability of conducting a future randomised controlled trial (RCT), including views on important outcomes of parents, staff and service commissioners

7. to scope existing data-recording systems and potential short- and long-term outcome measures (e.g. feeding outcomes, length of time to transition to full oral feeding, length of stay in NNUs, adverse events, infant growth, parent–infant attachment and well-being, and parent and staff satisfaction)

8. to determine key stakeholders’ views based on the evidence from our study (objectives 1–7) of whether or not a RCT of this approach is feasible and what the components of a future study would look like.

Overview of the study and structure of the report

The study comprised four work packages (WPs) based on the Medical Research Council principles for developing and evaluating complex interventions21 and process evaluations. 22 The study flow chart is shown in Figure 1.

FIGURE 1.

Study flow chart.

Pre submission of the funding application

We involved parents in preparing the application for funding to conduct the study. Through two third-sector charitable organisations [Bliss (London, UK) and TAMBA (Twins and Multiple Births Association) (now Twins Trust, Aldershot, UK)] which support parents and infants in NNUs, we discussed the study design with five women with recent experience of the transition from tube to oral feeding with a preterm infant in a NNU. The women had experienced scheduled feeding moving to cue-based feeding near discharge from the NNU. These discussions highlighted the importance of education for parents so that they understand the rationale for cue-based feeding and can identify their infant’s feeding cues. We learned that staff training is needed so that information and support for parents is consistent while also tailored to individual need. The women highlighted the need for the intervention to support breastfeeding as well as formula feeding and suggested important outcomes were time to discharge from the NNU and breastfeeding rates. The research team included a co-investigator from the neonatal charity Bliss, who contributed to the design of the study (in particular, the parent involvement component and suggestions to incorporate parents’ recording of outcome data in the intervention).

Parent involvement

Bliss was critical to facilitating parent involvement in the Cubs study. As well as having a member of Bliss on the research team, Bliss hosted the Parents’ Panel and the Bliss research team member attended all the research team meetings, Stakeholder Advisory Group meetings, and the workshops in WP 2 and WP 4.

Through an advertisement on the Bliss website, we recruited six parents with relevant experience of having an infant in a NNU to form a Parents’ Panel. Over the course of the study, three teleconferences were held with the Parents’ Panel. Activities of the Parents’ Panel included reviewing the participant information sheets prior to submission for ethics approval, co-designing the ‘Our Feeding Journey’ document that was an integral part of the intervention, and reviewing the intervention components including the posters and film. The Parents’ Panel reviewed and made many suggestions for the script of the film. The Parents’ Panel also suggested solutions to some of the challenges encountered during WP 3, for example offering alternatives to the telephone follow-up. Members of the Parents’ Panel attended the workshops in WPs 2 and 4.

Two members of the Parents’ Panel were also members of the Stakeholder Advisory Group and the Study Steering Committee. They were involved in all Stakeholder Advisory Group meetings including the final one at which they contributed to discussion of the findings, and options for optimising the intervention and research.

Stakeholder involvement

Other than parents, the main stakeholders for our work were health-care practitioners working in NNUs and researchers with an interest in neonatal care and/or intervention development and feasibility studies. We involved these stakeholders through our Stakeholder Advisory Group and the two study workshops (WPs 2 and 4). The Stakeholder Advisory Group comprised two neonatal care practitioners (one from England and one from Scotland), a representative from UNICEF UK BFI National Neonatal Network, an associate professor from Sweden with an interest in neonatal care and three researchers (a health economist, a statistician and an expert in using the ADePT framework, all from Scotland). The group met four times during the study and provided advice on the intervention and the research, including suggesting solutions to overcome challenges (e.g. slow recruitment). Wider stakeholder engagement was also achieved through the consensus-building workshops in WPs 2 and 4. The methods and findings of these workshops are reported in Chapters 7 and 11.

The parental and wider stakeholder involvement was critical to the study and had many benefits, as can be seen throughout this report. Parental engagement was better at the beginning of the study; for example, five parents attended the first workshop but only one parent attended the final workshop. However, engagement with the Parents’ Panel remained consistent.

Review aims and questions

The aims of the systematic review were to synthesise existing evidence on the components, characteristics, theoretical basis and associated BCTs23 of interventions, infant and parent outcomes and any economic evaluations.

The specific review questions were as follows.

In studies examining the effectiveness of approaches to feeding preterm infants transitioning from tube to oral feeding:

• What are the study characteristics (participants, interventions, comparisons, context)?

• What are the specific components of the interventions?

• What, if any, is the theoretical basis of any intervention being tested?

• What specific outcomes have been tested and what is the magnitude and direction of any effect?

We examined the nature of the evidence regarding the feeding type adopted (i.e. mother’s breast milk, donor breast milk, formula, any combination of these) and by feeding method (breastfeeding, bottle feeding, other methods such as cup feeding).

Review methods

The review protocol was registered on PROSPERO (registration number CRD42018097317).

Results

As a result of the search strategy, we screened 1028 studies and subsequently included a total of 25 unique studies reported in 30 publications (Figure 2 reports the numbers of records at each stage in the screening process).

FIGURE 2.

The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) flow diagram of study selection.

Analysis of policies and guidelines

The aim of the guideline and policy review was to inform the development of the Cubs intervention by considering current cue-based feeding practices within NNUs worldwide.

Summary

The systematic review included 25 studies, of which 10 were RCTs, nine were quality improvement projects and six were observational studies. The quality of the studies was low, with all but one having a high risk of bias. Two key components of all the interventions were a cue-based feeding protocol and training for staff and parents. Only two interventions reported a theoretical basis. Across all studies, 22 BCTs were identified, of which the most common was providing instructions on how to perform the required behaviour. The studies incorporated 41 different outcomes, the most common of which were daily weight gain, length of stay in NNU, and length of time to full oral feeding. Although we found one additional small trial, our results do not change the conclusions of the Cochrane review22 that the evidence in favour of cue-based feeding is of low quality and should be treated cautiously. The analysis of policies and guidelines included 15 documents: six from the UK, five from Canada, three from the USA and one based on a literature review. The findings highlighted common features that were taken forward to the consensus-building workshop to inform the development of the Cubs intervention (see Chapter 7).

Aims

The aim was to generate a contextualised picture of cue-based feeding strategies for preterm infants in NNUs by exploring experiential knowledge and clinical or behavioural approaches that facilitated the practice. The case study goals were to confirm and detail key practices identified through literature review and consultation, with neonatal medical and nursing expertise provided by the Stakeholder Advisory Group.

The three participating NNUs were the Princess Royal Maternity (Glasgow) in Scotland, and Uppsala University Hospital (Uppsala) and Falun Hospital (Falun), both in Sweden. These units were identified by members of the Stakeholder Advisory Group as suitable for inclusion on the basis of pre-existing or embedded cue-based feeding approaches.

Method

Case studies can be focused on a group of people for a particular purpose and be designed to examine examples or phenomena that illuminate activity and behaviour within a real-life context. 75 The case studies described here involved observational visits to the clinical area, informal interviews with key informants (including senior nurses and consultant neonatologists) and access to relevant documentation, policies, guidelines and training materials.

Based on the characteristics of the trials included in Watson and McGuire,1 key features that were anticipated would inform the Cubs intervention included how to recognise infants’ readiness-to-feed cues; frequency of assessment of readiness to feed; understanding infants’ cues of physiological stability or instability during a feed; how to recognise satiation cues and when to stop an oral feed; minimum and maximum time between feeds; how to assess need for and when to give ‘top-up’ feeds by tube; stages of transition to full oral feeding; monitoring infant well-being, and mother/parent confidence and satisfaction with feeding. This information was reviewed and refined by expert neonatal medical and nursing partners to create a template for case study data collection, prior to visiting the units.

The template consisted of 46 questions grouped into broad categories to organise the collection of observational, documentary and verbal information on cue-based feeding strategies. The categories included population (infants eligible for cue-based feeding); feeding cues (how cues were defined); practicalities of feeding (‘the what’ and ‘the how’ of administering enteral nutrition); clinical markers (what guided clinical decisions or precautions were taken); written resources and policies; parent and staff experiences, and general reflections.

Each unit was visited once between July and October 2018. The visits lasted between 4 and 5 hours and were conducted by two members of the research team: a health psychologist and a neonatal nurse specialist, each experienced in their respective fields. Both researchers were always present and completed the template separately, making contemporaneous notes and observations during the visit. After visits were completed, notes were transcribed, compared and analysed based on the aims and goals of the case studies.

Findings

The results of each case study are presented based on key differences in their approach in terms of the population of infants eligible, how the cues were assessed and implementation considerations.

Clinical considerations

Although there were key differences in the approach to cue-based feeding, there were clinical similarities across the three sites; for example, a maximum limit of 3 hours between feeds was applied regardless of cues, and this was relaxed only when feeding was established. There was a clear focus on the importance of assessing the efficacy of breastfeeding rather than just applying the tools available to support the identification of feeding cues. All units used prescribed daily feed volumes and relied on weight and head circumference trends to ensure that nutritional, growth and development needs were achieved. This was an important feature of NNU care and a key driver in the clinical management of infants in this environment.

Facilitators

Key learning

In Sweden, cue-based feeding practices were more established than in Glasgow, where the practice had been introduced only during the preceding year. Both Swedish units reported that, historically, there were some challenges, with older, more experienced, staff expressing a preference for scheduled feeding. However, there were key factors that helped address their concerns about risks. NNUs across Sweden collected extensive mortality and morbidity outcome comparison data for their infants. The unit in Uppsala was able to identify better outcomes than other units across several measures in these data. Staff in the unit believed that this was attributable to its approach to kangaroo care and, by extension, the fact that practices that were facilitated by this, such as cue-based feeding, became more acceptable. Staff at the unit in Falun also described similar challenges, with some staff resisting cue-based feeding, but they believed that a recent move to a purpose-built unit with better facilities to facilitate zero separation and increased kangaroo care helped to reduce resistance. Staff in Glasgow did not report any significant challenges in implementing their cue-based feeding strategy, identifying a willingness to accept the change as quality improvement.

Access to training was not perceived as sufficient in every area. Staff in Falun expressed a desire for more formalised education to be available either for or by NIDCAP trainers, particularly to help to support decision-making on the quality of feeds. Experience was seen as an important facet of managing the approach to top-ups in Falun, and there was some difficulty in articulating the clinical decision-making that guided this, suggesting that unconscious competence77 was directing the decision. It was evident across the case study sites that assessing the quality of feeds and the approach to top-ups required some degree of clinical judgement, as it was impossible to ascertain the volume an infant had swallowed at a breastfeed. This may explain the preference for scheduled feeding among more experienced staff in Sweden. Despite differences in approach, each unit had operationalised elements of top-up management to provide a degree of certainty in achieving prescribed volumes and nutritional needs by reducing reliance on interpretation or judgement. In Glasgow, this involved using minimum gestational age, increasing the infant’s likelihood of sufficiently mature reflexes, as well as a quality feed tool to guide the decision. Falun’s semi-scheduled approach meant that relatively small incremental volume reductions were of low impact and could be carefully guided by weight trends over time, and Uppsala utilised test weighing to make assessments of actual intake.

What should not be underestimated, in terms of the impact on families’ experience of cue-based feeding approaches, was the sociocultural difference evident between Scotland and Sweden. Very few infants received bottles in Falun or Uppsala and there was very little bottle-feeding equipment and very few breast milk substitutes available in the units to offer this, as breastfeeding was the accepted norm. Sweden has a very high breastfeeding rate compared with the UK. Staff here also expressed their ardent belief in preterm infants’ ability to demonstrate and follow through on cues in their own time, especially if they received unrestricted kangaroo care. Their commitment to unrestricted kangaroo care was demonstrated by having adult beds at the cot side and the use of ‘open’ incubators to reduce physical barriers. Sweden also had many advantages in terms of well-funded social welfare benefits, extensive parental leave and spacious well-facilitated ‘living’ environments available to Swedish families. There was an implicit recognition in these measures that families’ and infants’ normal state is to be together at all times. This did not just facilitate a zero-separation philosophy from staff and families, it demanded it. The feasibility of this approach is unlikely to be replicable to the same extent in the UK. Reduced family–infant interaction has an impact on the opportunity for kangaroo care and may affect opportunities for cues generally to be both recognised and responded to from a very early stage. This, arguably, suggests that a higher level of nursing input into the delivery of care and caveats for introducing cue-based approaches may be necessary.

Limitations

The limitations of these case studies are that they represented an informal, time-limited snapshot of cue-based feeding approaches in three purposefully selected NNUs. A large amount of information was collected, and presentation of the results represents a rapid ethnography78 rather than an in-depth analysis of behaviours and practice. A key strength of these case studies was using two researchers with significant experience in neonatal intensive care nursing and in health psychology. The combination of the differing lenses on the clinical and behavioural aspects of cue-based feeding afforded a rounded perspective on which to inform the development of an intervention.

Aim

The aims of the survey were to map and understand the range of approaches to the transition from tube feeding to full oral feeding in the UK including the scope of routine data collection systems, training needs of staff, and the variation in practice to understand ‘usual care’ that would form the control arm of a future trial.

Method

A purposive sample of 20 NNUs was selected from across the 220 units in the UK. The selection of units was based on the geographical location, designated unit level, cot capacity, population and urban–rural setting. Initially, units were contacted on behalf of the study team by regional representatives of the Neonatal Networks. Where there was no response, contact was followed up by a representative from the relevant Clinical Research Network for the trust/health board. Out of the 20 units sampled, 18 units indicated a willingness to take part; two units did not respond to the invitation.

Each unit that indicated a willingness to participate nominated an individual, or individuals, to be interviewed. Once units had agreed to be contacted, an introductory call was made by a researcher from the study team. The purpose of this call was to discuss the aim of the telephone survey, to discuss the participant information sheet and to arrange a suitable time and date for a telephone interview. Verbal consent was sought at the outset of the telephone interview.

The interviews were conducted by a member of the research team using a semistructured interview schedule based on the aims of the survey. The duration of interviews ranged from 18 to 56 minutes; the mean length was 35 minutes. The units varied from level 1 (special care) to level 3 (neonatal intensive care). The number of cots in the units ranged from 6 to 53. The job roles of interviewees included infant feeding advisor (n = 6), specialist speech and language therapist (n = 5), staff nurse (n = 4), senior charge nurse (n = 2), clinical/practice educator (n = 2), unit manager (n = 1), clinical nurse lead (n = 1), neonatologist (n = 1) and neonatal consultant (n = 1). There were four participants in one interview, two in another, and the remaining participants had individual interviews.

Interviews were recorded, transcribed and analysed using a thematic approach. 79 Initial coding was conducted independently by two researchers. The final themes were discussed and agreed with the research team.

Approvals were granted by NHS East of Scotland Research Ethics Committee (LR/18/ES/0059) and the Health Research Authority.

Findings

An overarching theme was typologies of change. This referred to the various stages in the process of change towards introducing cue-based feeding described by participants. Three typologies of change were evident in the descriptions of units’ approaches to the transition from tube to oral feeding: ‘not considering change’, ‘considering change’ and ‘making changes’. A further subtheme was ‘variations in practice’. Descriptions of these subthemes utilised the characteristics, ethos and practices of units, as well as the micro language, namely change talk,78 to categorise the stage of change of each unit at the time of the telephone survey.

Summary

This chapter describes the findings of a telephone survey of 18 purposively selected NNUs across the UK. The findings show a wide range of practices and approaches to the transition from tube to oral feeding for preterm infants. The analysis suggested three stages in the change process towards cue-based feeding; a smaller number of units were not considering making any changes, or were actively implementing cue-based feeding, while the majority were considering but had not yet implemented systemic change to cue-based feeding. Also of interest is that in all units, regardless of where they are situated on this typology of change, there are individual staff who are enthusiastic about cue-based feeding, and those who were described as resistant to change.

Aims

The aim of this component of the project was to understand parents’ and health-care professionals’ views and understanding of cue-based feeding to inform the intervention, particularly the training component.

Methods

We conducted focus group discussions and/or individual interviews with a convenience sample of 15 parents (12 mothers and three fathers) and 32 health-care practitioners. Health-care practitioners were invited to take part in the interview through an e-mail sent by the senior charge nurse or matron in each unit. Interviews took place during working hours. Those who were interested in participating in the study contacted the site research nurse, who arranged the time and location of the interview. Where possible, interviews were conducted as focus group discussions, but, owing to staffing constraints, it was necessary to conduct some interviews individually. Participating health-care practitioners included neonatal consultants, speech and language therapists, occupational therapists, neonatal nurses, nursery nurses, health-care assistants and infant feeding leads.

Parents were invited to an interview by a member of their infant’s care team. When parents indicated an interest, they were contacted by the site research nurse to arrange a time and date for the interview. Although these were intended to be conducted as focus group discussions, those who were interviewed indicated a preference for individual interviews. In addition, the reality of parents needing to attend to their infants at various times meant that it was not always possible to arrange group interviews that were suitable or convenient for everyone.

Interviews and focus group discussions were conducted by site research nurses guided by a semistructured interview topic guide. Staff interviews lasted 37–52 minutes and parents’ interviews 10–25 minutes. The interviews were audio-recorded. We took a pragmatic approach to analysis; each interview was listened to by a member of the research team, and notes of key points were taken. The key points were organised into common themes.

Approvals were granted by NHS East of Scotland Research Ethics Committee (LR/18/ES/0059) and the Health Research Authority.

Findings

Five themes were identified from the analysis of the data from health-care practitioners: knowledge and attitudes; concerns about cue-based feeding; variations on practice; role of medical staff; and education. Six themes were identified from the analysis of the interviews with parents: experiences, identity, getting home, feeding cues, support and suggestions.

Summary

In this chapter we describe the findings of qualitative interviews with staff and parents regarding their current experiences and approaches to the transition from tube to oral feeding. From the analysis it was evident that the health-care practitioner perspective was that most staff in their unit were implementing a cue-based approach. Although health-care practitioners reported implementing cue-based feeding in practice, their descriptions suggest that a version of cue-based feeding was being used but not as it is defined in the literature. It was also evident that staff have concerns about the safety of cue-based feeding that may hold back full implementation. A further educational issue for consideration is that staff did not appear to be familiar with stop cues. It was clear from the staff data that a multidisciplinary approach is needed to implement a change to cue-based feeding.

In contrast to the staff perspective, parents’ experiences suggest that a scheduled approach is more common. The variations in practice reported by health-care practitioners suggest inconsistency. This inconsistency could explain why parents’ experiences were different from those of health-care practitioners. From the parents’ perspectives, it also appeared that some health-care practitioners struggled to explain the reasons for different approaches (e.g. for waking an infant for feeds during the night). Discharge from the NNU was clearly a focus for parents, and this appeared to lead to some pressure around oral feeding. Parents clearly appreciated the support for feeding provided by nursing staff. Parents’ suggestions for supporting cue-based feeding included learning resources, such as posters and a film, and a means of tracking the progress of their infant.

In Chapter 7, we describe how the information reported in this chapter, together with the literature review, analysis of policies and guidelines, case studies and telephone survey, was used to inform the development of the intervention.

Introduction

The intervention was co-produced with stakeholders and was informed by the evidence gathered in the previous stage: the systematic review; the analysis of policies and guidelines; three case studies of NNUs with embedded cue-based feeding; telephone interviews with 18 NNUs across the UK; and qualitative interviews and focus group discussions with staff and parents in three NNUs. In this chapter we describe the process of developing the intervention and the intervention components. The overall aim was to co-produce an evidence-informed, adaptable, manualised intervention that included staff and parent educational support for feeding preterm infants at the transition from tube to oral feeding in response to feeding cues and signs of infant stability.

Methods

The aim was achieved through three key activities:

1. development of a matrix of intervention options

2. a consensus-building workshop

3. co-production of the intervention.

Results

Summary

The Cubs cue-based infant feeding intervention was developed based on evidence from a literature review, analysis of polices and guidelines, case studies and primary qualitative research, as well as the expert opinion of the research team and stakeholders including parents. The intervention was developed using an iterative process of synthesising the evidence base, gaining consensus from stakeholders (including parents) at a workshop, co-producing the intervention with parents, and having discussions with the research team, Stakeholder Advisory Group and the nurse managers of the three sites in which the intervention was implemented. The final version of the intervention was manualised (i.e. written as a manual). One area of contention was the exclusion of infants on high-flow oxygen; however, it was felt best to adopt a cautious approach. Although there was consensus that there should be no minimum gestational age for commencing the intervention, one implementation site modified the intervention for bottle-fed infants, applying a 34 weeks’ gestational age limit to align with the site feeding protocols.

In the following chapters, the methods (see Chapter 8) and results of the feasibility study are described, incorporating quantitative results including recruitment and retention (see Chapter 9) and qualitative findings (see Chapter 10).

Study design and theoretical framework

The mixed-methods feasibility study comprised quantitative and qualitative approaches to meet the above objectives. The study involved implementation of the intervention in three NNUs. The study was designed as a single-arm feasibility study; no comparison group was included. Feasibility outcomes of acceptability, adoption, appropriateness and fidelity were based on the model by Proctor et al. 83 We judged feasibility as whether the intervention was practicable to implement based on the views of the participants, as well as the expert opinions of the research team and Stakeholder Advisory Group. We used the Proctor et al. 83 definition of acceptability (i.e. that the intervention was ‘agreeable, palatable or satisfactory’).

Setting

The intervention was implemented in three NNUs, two in England and one in Scotland. The sites were selected to provide different sizes and levels of unit, and diversity of populations. For pragmatic reasons, units where members of the research team had established links were selected. Although all three units have full accreditation for the UNICEF UK BFI neonatal standards,68 at the time of selecting the units only site 2 had achieved this status. Table 8 shows details of the three units.

Implementation

Implementation of the intervention commenced with a site visit by the research team members, who provided an initial 2-hour training session for the research nurse and the key cascaders on the intervention and the study procedures. Each unit was provided with paper and electronic copies of the intervention manual and resources, a pack for the parents of each infant containing the intervention information and resources, and a set of posters to be displayed around the unit. Links to the Cubs film and narrated PowerPoint were also included. It was planned that the intervention would commence once training had been cascaded to most staff. Commencement of the study was delayed for several reasons, including gaining approval from the study sponsor (University of Dundee) taking longer than anticipated and a delay in being able to circulate the film (one of the infants featured in the film died and it was felt that permission should be sought from the parents to retain that section of the film). In one unit, a planned move to temporary accommodation was delayed meaning that instead of taking place before the study started, it took place just as the study was starting. The principal investigator (PI) asked to delay the start of the study until the move was completed and the staff and parents were accustomed to their new environment. Recruitment took place over 7 months (August 2019 to February 2020). All follow-up was stopped in early March 2020, as NHS research was stopped because of the COVID-19 pandemic.

Quantitative methods

Quantitative methods were used to assess recruitment and retention, and clinical outcome data on infants included in the study.

Qualitative methods

The quantitative data were supplemented with qualitative data as part of a process evaluation to provide a more in-depth understanding of implementation outcomes of acceptability, feasibility, adoption and fidelity. 83 The study also included qualitative data from interviews with parents of infants receiving the intervention and staff involved in its implementation. Further data on fidelity were collected through observations.

Ethics approvals

Approval for the feasibility study was given by the North of Scotland NHS Ethics Committee (19/NS/0055), the Health Research Authority and NHS Research Scotland. Research and development approval was given by the relevant NHS trusts in England and health board in Scotland.

Summary

In this chapter we have described the methods used for the feasibility study. This includes the study objectives, design and theoretical framework, the implementation process, the research settings and the methods for the quantitative and qualitative components of the study. The following two chapters present the findings of the quantitative analysis (Chapter 9) and the qualitative analysis (Chapter 10). These findings were then taken forward to the final stage of the research (WP 4), as described in Chapter 11.

Recruitment and retention

The flow of infants in the study and the reasons for non-recruitment are shown in the Consolidated Standards of Reporting Trials (CONSORT) flow chart86 (Figure 3).

FIGURE 3.

The CONSORT flow chart.

Recruitment overall was about one-third of those screened, but varied by site from a low of 20% in site 2 to 47% in site 3 and 42% in site 1 (Table 10).

Overall, 13% of those screened were not eligible. The main reasons for this were that oral feeding was already established (n = 16) and medical conditions (n = 4). The fact that some infants were ineligible because they had already transitioned to full oral feeding suggested that screening took place too late in the infant’s feeding journey. Only 37% (n = 50) of eligible infants were recruited to the study. This included 28% (n = 38) whose parents declined to participate and 10% (n = 13) who were not consented (owing to either the research nurse or the parent not being available); for 26% (n = 35) of infants, the reason for non-recruitment was not documented. Although reasons for declining were not recorded, feedback from study research nurses suggested that many parents felt too overwhelmed at this vulnerable time to think about participating in research. Site 3 experienced a particularly quiet period when there were few infants in the unit, and an extremely busy period when staff reported that they lacked time to approach parents about the study. This affected the recruitment rate.

Table 11 shows recruitment in relation to the target of n = 60, with overall recruitment of 83%. The mean recruitment rate was seven infants per month, with a range of 3–12.

Of the 50 infants recruited to the study, 49 received the intervention (one was withdrawn, the reason was not documented) and 48 were followed up until discharge from the NNU (one was transferred to another NNU). Follow-up rates at 2 weeks post discharge were much lower with only 38% (n = 19) followed up giving a 62% drop-out rate. Non-response to telephone calls was the main reason for loss to follow-up. There was wide variation between sites with the lowest drop-out rate at site 1 (30%), followed by site 3 (77%), and the highest at site 2 (87.5%). Why there was such variance across sites is unknown, but feedback from the research nurses suggested that it may be partially related to how much effort was made to contact parents. For example, one research nurse described making multiple attempts to reach parents. Another research nurse had informed parents in advance that the follow-up telephone call would come from a ‘withheld number’. This advance knowledge may have increased the likelihood of parents responding to the telephone calls.

Evaluation of outcomes

Table 12 shows the baseline characteristics of the infants in the study including weight and gestational age at birth and at the start of the intervention.

Among the 49 infants with evaluable data, the mean gestation at birth was 30.3 weeks and the mean birthweight was 1530 g. Of these infants, 78% were singleton births and 22% were multiple births. The mean gestational age at the start of the intervention was 34.6 weeks, with a mean weight of 1990 g.

Table 13 shows the characteristics of the infants at transition to full oral feeding and at discharge from hospital.

At time of discharge from the NNU, the mean corrected gestational age was 37 weeks and 4 days (range 34⁺³–44⁺² weeks) with a mean time of 14.4 days from the start of the intervention to discharge. The mean number of days from the start of the intervention to transition to full oral feeding was 10.8. Mean weight increased from 1990 g at the start of intervention to 2434 g at discharge. The mean daily change in weight from intervention start to discharge was 25 g. The change in weight from birth to 2-week follow-up is illustrated in Figure 4.

FIGURE 4.

Weight from birth to weight at 2-week follow-up.

The mean age at the 2-week follow-up was 71 days from birth, with a mean corrected gestational age of 40 weeks and 2 days. The mean weight was 2842 g (Table 14).

Feeding outcomes

Prior to their infant starting the Cubs intervention, most of the parents (n = 37) indicated that their feeding preference would be to exclusively breastfeed their infant. Figure 5 shows the feeding outcomes for these infants. On discharge, 21 of the 37 infants were being breast fed exclusively or were receiving EBM only or were fed with a combination of breastfeeding and EBM. A further six were breastfeeding and/or receiving EBM supplemented by infant formula and seven were fed exclusively with infant formula. One infant who was discharged was breastfeeding with top-ups of EBM via nasogastric tube. The remaining two infants were transferred to another unit before discharge, and were therefore lost to the study. Thirteen of the 37 infants were followed up at 2 weeks. At this time point, five infants were breastfeeding and/or receiving EBM exclusively, four were breastfeeding and/or receiving EBM supplemented by infant formula and four were fed exclusively with infant formula.

FIGURE 5.

Feeding outcomes for infants of parents who intended to breastfeed exclusively.

Of the remaining parents who joined the study, two planned to combine breast and infant formula feeding. On discharge one parent was feeding their baby infant formula exclusively and the other baby was transferred to another unit prior to discharge and was therefore lost to the study. The remaining nine parents stated that they planned to feed their baby with infant formula and were doing so at discharge. Two parents’ preferences were not documented.

At follow-up, seven mothers reported that they felt their infant was having feeding complications. This was cited as the reason for introducing infant formula feeding after discharge (e.g. they were concerned that their infant was not getting enough milk at the breast). However, 17 out of 19 mothers reported that they were confident and satisfied with their infant’s feeding.

Feeding journeys

The ‘My Feeding Journey’ document enabled parents and staff to chart an infant’s journey from tube to oral feeding. They were asked to detail the start and stop cues shown by the infant; to provide detail of feeds (such as breast or bottle feed, type of milk fed and duration of feed); to provide detail of top-ups given; and to score the feed using feeding assessment charts. Free-text boxes enabled comments to be made on the current feed and any plans for the next feed.

Of the 50 infants recruited to the study, journals were returned for 36. Others may have been completed but they were taken home by parents. Data for 483 feeding days were collected. Owing to the variation in completion of the journals, we have selected those with the most complete information as case studies. Feedback from the Stakeholder Advisory Group suggested that this would be the most informative way to present the data. We present case studies of three infants, one from each site, to show variations in how the intervention was implemented and to include breastfeeding and bottle feeding experiences.

Case studies

Infant 17, site 1

This infant was from a singleton pregnancy born at 25⁺² weeks’ gestation, weight 970 g; this infant was one of the most preterm in the study. Prior to birth the mother had intended to breastfeed.

The infant started on the intervention at 35 weeks’ corrected gestational age and weight 1710 g. The infant’s first breastfeed was on day 1 of the intervention in response to start cues of rooting and hand sucking and took place 90 minutes after a tube feed.

By day 7, the infant was awake and showing cues at five out of the eight feeds documented. The infant was tube fed overnight when the mother was not present, but breastfed throughout the day. The infant was said to tire at the breast and half top-ups were given in line with breastfeeding assessment scores. This feeding pattern continued until day 15, after which no feeding data were documented until the infant was recorded to have achieved full oral feeding on day 18. Discharge was on day 26, when the infant was 13 weeks old, with a gestational age of 38⁺⁵ weeks, and weighed 2090 g. When the infant went home, they were fully cue-based breastfeeding.

At follow-up the infant was described to be cue-based breastfeeding frequently, with additional breast milk given by bottle to aid medication administration. The infant had gained a further 490 g over the 2 weeks since discharge and the parents reported feeling confident and satisfied with feeding.

Infant 14, site 3

This infant was from a singleton pregnancy born at 27⁺⁴ weeks’ gestation, and weighing 726 g. The parent’s pre-birth intention was to bottle feed infant formula.

Oral feeding was introduced when the infant was just over 6 weeks old at corrected gestational age 34⁺² weeks, weighing 1310 g. This was the lowest weight of all infants in the study at commencement of oral feeding. The first oral feed documented was a bottle feed given when the infant was showing cues of mouthing and putting hands to mouth. The infant was described to have taken the bottle well and the feed was scored as D, indicating that the infant was alert and fed with strong co-ordinated sucking and breathing. However, after a desaturation episode at the next feed attempt, the remaining feeds on day 1 were full tube feeds.

By day 9 of the intervention, the infant was fed at 3-hourly intervals. Half of the infant’s bottle feeds were given when the infant was showing start cues and half top-ups were given regularly at this stage. By day 16 the infant was fully orally fed by bottle and received only one small top-up. Feeding intervals ranged between 2.5 and 3.5 hours. Feeding cues were recognised for every feed, although sometimes the late cue ‘crying’ was documented. The infant was described as feeding slowly but all feeds are scored as ‘E’, showing that feeding ability had progressed well over the week.

This infant was discharged home on day 17 at 36⁺⁴ weeks’ corrected gestational age, weighing 1730 g (an increase of 1004 g since birth) and fully bottle feeding. Feeding was described as a combination of cue-based and scheduled feeding.

At the 2-week follow-up, the infant was 11 weeks and 2 days old, corrected gestational age 38⁺⁶ weeks, and weighed 2100 g, a further increase of 370 g since discharge. The infant was fully bottle fed to a schedule and the parents did not report any feeding concerns, although the infant required a surgical procedure for an inguinal hernia following discharge.

Infant 8, site 2

This infant was a singleton born at 35⁺¹ weeks’ gestation and weighing 3018 g. The mother had documented in the feeding journal that she was very keen to breastfeed as she had felt that her feeding experience with her first infant could have been better.

The infant quickly progressed on to the intervention at 1 week and 2 days old (36⁺³ weeks’ corrected gestational age), weighing 2760 g. The infant progressed well, and the parent had commented that the infant ‘needed to build stamina feeding at the breast’. Feeding assessment scores and the use of top-ups were consistent with these comments.

As can be seen from Figure 6 (which is a segment of the infant’s transition), this infant was fed according to cues from the start of the intervention. The straight line in the middle of the graph is an overnight period when the mother was not present and the infant was fed by nurses on a schedule. When the mother returned the next morning, the infant was again fed in accordance with cues. The infant was discharged home after 6 days on the intervention, and was fully cue-based breastfeeding.

FIGURE 6.

Feeding journey extract.

Analysis of feeding cues

Within the journals there were over 770 documented feeding start cues. The most common cues recorded were awake and rooting. The full analysis of the range of cues is shown in Figure 7.

FIGURE 7.

Start cues.

Crying was documented 75 times across 24 infants and was the third most common feeding cue. This is a late cue, which would indicate that the infant’s early start cues had probably not been recognised. There did not appear to be a pattern to the infants who cried before feeding, although two infants transitioning to feed at the breast were both reported to cry regularly on a day that they were fed frequently.

Stop cues were not reported as often as start cues (Figure 8). The most frequently reported stop cues were ‘tires/falls asleep’ and ‘stops sucking’. More subtle cues, such as the infant raising their hands, were documented only once.

FIGURE 8.

Stop cues.

Although a large number of start cues were identified within the ‘My Feeding Journey’ record, a large number of feeds appear to have been given on a 3-hourly schedule. This may indicate that the recognition of these cues was not the driving factor for feeding the infant.

At discharge, 10 infants were fed through a combination of cue-based and scheduled feeding; at the 2-week follow-up this number was five (out of 19). Eleven infants were fully cue-based feeding at discharge and by follow-up this had dropped to six (out of 19). One infant was discharged on scheduled feeding; this increased to two infants by follow-up. For a large proportion of the infants at discharge (n = 26) and at follow-up (n = 5), the approach to feeding was not documented.

Summary

The study recruited > 80% of the target sample over 7 months, with an average recruitment rate of two infants per site per month. Retention in the study until discharge from the NNU was high, but there was a low rate of follow-up at 2 weeks after discharge. We were able to collect data on important outcomes including weight, number of days to transition to full oral feeding and time from intervention start to discharge from the NNU.

The data on infant feeding outcomes were not as comprehensive, and we had to rely on the ‘Our Feeding Journey’ documents, which varied in completeness of data. Compared with the stated feeding intentions of parents, there was a gradual overall decrease in the number of infants who were fully breastfed at discharge, and at the 2-week follow-up. The analysis of case studies suggested reasonable fidelity to the intervention, although there were indications that a 3-hourly schedule of intervals between feeds was often followed. In terms of the cues recorded, start cues were documented more frequently than stop cues.

In the next chapter we present the findings of the qualitative analysis which add depth and explanation to some of the quantitative findings.

Recruitment and participants

Fourteen interviews with parents took place, 47% of the target of 30. Six took place in site 1, three in site 2 and five in site 3. A key challenge to conducting the interviews was finding a time and place suitable for both the parent and the research nurse. Many parents did not wish to leave the cot-side or be distracted from their infants, and it was difficult to achieve quiet and privacy on the NNUs. One research nurse reported that she had needed to make several visits to find a time when the parent was not busy with the infant. It was easier to conduct the interviews if the infant was in a room on their own. In some cases, the infant was discharged before the research nurse was able to conduct the interview with their parent. The Parents’ Panel suggested that one way to overcome this was to undertake the interview by telephone following discharge; the study protocol and approvals were amended to offer this, but no parents took up this option.

Sixteen interviews with staff took place, 53% of the lower target of 30. Three took place at site 1, five at site 2 and eight at site 3. Finding a suitable time and location for the staff interviews was challenging in the context of busy workloads and units’ layouts. Feedback from the research nurse at site 1, where there was a particularly low level of recruitment (this was the unit with the largest number of staff), suggested that time was the greatest issue, and that the unit management was not particularly supportive. It was particularly challenging for the research nurses to find a time to interview the medical staff and only one was completed.

Twenty-one hours of observation took place across the three sites on seven occasions (two each in sites 1 and 2, and three in site 3) representing 38% of the target of 54 hours on 18 occasions. The main barrier was gaining consent from staff. Feedback from the research nurse in site 1 suggested that staff felt that their practice was being judged.

The COVID-19 pandemic affected the number of interviews conducted, which otherwise would have continued throughout March and April 2020.

We present the findings in three key sections: contextual factors that influenced the implementation of the intervention, implementation outcomes and views on a future study. To preserve anonymity, as there were small numbers of parent and staff interviews, we provide only the site and whether or not the participant was a parent or staff member as contextual information for participant quotations.

Contextual factors

There were several contextual factors, (external to the intervention) identified across parent and staff perspectives with the potential to positively or negatively affect the implementation process.

Implementation outcomes

Cost

In terms of cost, participants were asked if cue-based feeding required additional resources compared with scheduled feeding. Overall, both staff and parents suggested that this was not the case. However, staff reported that they had to shift the time allocation or organisation of some of their usual tasks to enable them to address the intervention procedures. In particular, staff noted a shift towards needing more time with parents in the early stage of the transition to oral feeding, with less time needed later as parents became confident in recognising cues. This was not perceived as problematic. Sometimes it was difficult to extrapolate whether the additional time related to implementing the intervention or to the study procedures:

So I would say probably, dependent on those things that I’ve said, I would like at least an extra half an hour to talk through how, what they’re seeing relates to the paperwork. So let’s say for example half an hour, 45 minutes, you might generally talk about cue-based feeding, an extra half an hour to go through that properly as well I think, and that would include your visit back just to check that they’re OK with it and understand and are managing to fill it out.

Staff 1, site 2

From the parents’ perspective, there were no major issues associated with implementation costs. Although some parents reported an emerging need to buy new bottle-feeding equipment to better support cue-based feeding, this was not perceived as a barrier to engaging in cue-based feeding:

The only thing I’ve had to buy is different kind of bottles, purely because the ones I’ve got were too big to start with so I had to go and get the smaller teat ones which then really, it’s almost like, they showed more cues knowing that they were able to feed from a different bottle. So yeah, I went and bought a set of bottles.

Parent 9, site 2

Other costs mentioned by parents, such as parking charges, taxi fares and buying meals in the hospital, were not related to the intervention. Parents and most staff thought that parents did not spend any more time with their infants for cue-based feeding than for scheduled feeding. However, a few staff participants commented that they thought that it had encouraged parents to stay longer at the unit:

I think the parents that have been on the Cubs study tended to be here a bit more often and would stay for maybe a few feeds in a row rather than just coming in for the one feed they knew and then going home.

Staff 6, site 3

Nursing staff noted the importance of having appropriate resources to overcome the limitations they experienced in their ability to train other members of staff, highlighting the implications that this may have had in terms of uptake and consistency in the delivery of the intervention:

. . . train more nurses to get the information across to the parents, so that then we can be then a bit more thorough, because obviously they’ve built up a relationship with us, so if someone has come in and given them this information, the research and then they show it to the nurse, ‘Oh, such and such, has just come in, and do you think this is a good idea?’ At least then we can then promote that and say, ‘Oh, yeah, this is actually really, really good, this will fit in with what I’ve just discussed with you about baby led.

Staff 3, site 2

A doctor noted that the intervention reduced the time he needed to be involved:

I suppose as a consultant doing the ward round or being in charge of the unit, my role would be to flag at ward round times to think about whether the baby is ready to feed orally yet or not. [. . .] So as far as the Cubs intervention has been concerned, I’ve found that it very much decreases what intervention I need to make, in terms of moving the baby’s feeding on. People would wait for the ward round and wait for me to say, OK move onto alternate treatment bottle or move onto two bottles to a tube.

Staff, site 3

Mechanisms of impact

We analysed the data to find any information on the mechanisms that might lead to the outcomes and assessed whether they supported our logic model (see Chapter 7). The main themes were building parents’ knowledge, confidence and skills, the supportive relationship between staff and parents, the perceived consistency between the intervention and current staff practices, and increased opportunities for parents to understand their infant’s behaviour leading to enhanced opportunity for bonding.

The intervention procedures and materials allowed parents to build up their knowledge and confidence to identify and act on cues:

My confidence has grown loads over the time. My confidence to start with, to identify the cues was I doubted myself because I was never sure whether I was seeing the right cues or was I trying to see something that wasn’t a cue so yeah, it took me a while to build my confidence but now I’m pretty confident that I know exactly what the kind of cues they’re going to show me.

Parent 9, site 2

The support and guidance provided by members of staff during the intervention was perceived as an integral part of the intervention by both staff and parents, suggesting that this relationship was a likely key factor to ensure successful implementation and intervention outcomes. A helpful, positive relationship between staff and parents during the intervention process was greatly valued by parents:

Yeah [the staff support with cue-based feeding was] amazing. They honestly, they’re so good and they’re good in that if you want their help, they’re there but they’re also good enough to give you space to let you just do it yourself and learn yourself.

Parent 8, site 1

Staff’s perceived consistency between the intervention and their current practices supported uptake and engagement with the intervention. However, in some instances, this resulted in a reinterpretation of the principles of the intervention whereby cue-based feeding was undertaken within the confines of scheduled feeding, thus avoiding the challenges associated with changing well-established practices. This links to the previous points made about fidelity to the intervention:

We kind of do it [support parents with cue-based feeding] anyway. We talk to parents, even though we do feed scheduled, shall we say, like, every 3 hours, I think we talk about — does he want a feed, is he looking, is he awake or is he not? So, I think we do that anyway so, I don’t think it was much of a change.

Staff 4, site 3

The intervention procedures and materials, alongside the support provided by members of staff during the intervention, enabled parents to master and embed cue-based feeding skills into their interactions with their infants:

At first, I didn’t know kind of what I was looking for as such. I had an idea in my head but overall, I didn’t entirely know what I was looking for. But now I do. [. . .] it gives parents an idea as to what you need to look for, you know, when you’re at home, for example, because that’s what you’re going to be doing when you get home. So, that’s given me an understanding of that, in that respect.

Parent 26, site 3

During this process, parents noted that their journey through the intervention and their experience of cue-based feeding allowed them to learn about a range of cues that went beyond their previous knowledge, building up their confidence to identify and act on cues, and ultimately enabling them to better understand and meet their infant’s needs in a more responsive way:

I didn’t realise there was so many cues that babies do before they want to feed. I thought it was just like looking around and crying generally, which is then you’re too late but, yeah, it’s been interesting to pick up the other cues that they have. [. . .] I feel much more confident to know when she’s ready.

Parent 1, site 2

I think it’s a great way of doing it, rather than get to the point of her screaming and then you’re behind them, aren’t you because you’re rushing around trying to get the food ready. I think this way, you get an inkling and then we’ve got a little — mainly because of the way they’ve shown me here, but we’ve got into a routine now that when she starts doing it, we change her nappy, so, she knows that. Then she sits out in her cot for a little bit to get awake and a bit more alert and by that point, she’s ready but not distressed. So, it is a nice experience, it’s not like, oh, my God, she’s hungry, I’ve got to feed her.

Parent 8, site 1

Alongside this, parents also noted that their journey through the intervention and experience of cue-based feeding afforded them increased opportunities to engage with breastfeeding and bond with their infant:

It’s quite a helpful thing for parents. I think most staff are that kind of busy with so many other things that that’s how the way it is in most jobs but as an overall experience, it has been very helpful. It helps you get to know your baby a bit more so.

Parent 9, site 2

I think it’s really helped with bonding because she’s needed something, and I’ve been able to give it to her.

Parent 15, site 3

Views on a future study

We asked participants for their views on the important outcomes for a future trial and whether they would be willing to be randomised to a cue-based compared with scheduled feeding trial.

Summary

In this chapter we have presented analysis of qualitative interview and observational data. Recruitment to these components of the research was challenging owing to a variety of factors, including time and location for staff and parents, and processes for gaining informed consent from staff for observations. However, the data from the interviews with 14 parents and 16 staff are rich and provide an insight into how cue-based feeding was implemented in accordance with the views and experiences of staff and parents.

The context of the NNUs facilitated implementation of cue-based feeding. Barriers related to parents’ other responsibilities and the wide variation in experience of staff. In terms of implementation outcomes, the intervention was acceptable, with the resources well received, although there was some dissatisfaction among staff about the amount of documentation. The main issue in adoption of the intervention was the failure of the cascade approach to training to reach all staff in a timely manner. The intervention was perceived to fit well with current neonatal care practice, although resistance to change from some staff was mentioned. There were a considerable number of data relating to fidelity and, in general, there was good evidence that the intervention was implemented as intended, especially in relation to not applying a lower gestational age to the start of oral feeding and not having a set rate of transition. However, there was evidence that there was a 3-hourly feeding schedule for most infants. There was little information on which to assess cost but the main perception was that the intervention did not require additional staff and parent time compared with scheduled feeding, and may have reduced the time required by medical staff.

Several mechanisms of effect were suggested, including building parents’ knowledge, confidence and skills, relationships between staff and parents, consistency between the intervention and current practice, and increased opportunities for parents to understand their infant’s behaviour leading to enhanced bonding. The key perceived benefits related to empowering parents, providing evidence that underpinned decision-making and some tentative views that the intervention may lead to earlier discharge from hospital and faster transition to oral feeding.

For a future definitive evaluation, measuring infant growth and weight, and time to full oral feeding and discharge were the most important outcomes to parents and staff. Staff would also like to see longer-term follow-up of infants and the impact of cue-based feeding on feeding and obesity. There were mixed views on randomisation, suggesting that many parents and staff were not in equipoise for cue-based feeding, believing it to be the best approach. In the next chapter we describe stakeholder views on how to optimise the intervention and their views on options for the next steps for research.

Methods

The primary aim of this stage of the project was to determine stakeholder views, based on the evidence of the feasibility study described in Chapters 8–10, of whether or not a RCT of the Cubs cue-based feeding intervention is feasible and what the components of a future study might look like. A secondary aim was to explore stakeholder views on how to optimise the intervention.

This stage of the work was guided by the ADePT process, which aims to provide transparency in decision-making processes after feasibility studies, and involves systematic identification and appraisal of problems and potential solutions. The process involves three key steps: (1) decisions about the type of problem (is it an issue for a future trial, an issue for the real world, or for both?); (2) assessment of all potential solutions; and (3) solutions are ranked, combined and refined. 87

We adapted this process to include stakeholder engagement in proposing potential solutions through a stakeholder workshop and in refining the final solutions through discussions with the Stakeholder Advisory Group.

Findings

Table 15 presents the methodological challenges, findings and evidence that were taken forward to the stakeholder workshop, where the focus of the discussions was on recruitment and retention, fidelity and acceptability, appropriate outcomes and the design of a future study.

Next, we present a summary of the solutions that were ranked high for acceptability and feasibility at the stakeholder workshop.

To increase recruitment of infants, it was proposed that the window of opportunity for recruitment should be longer so that infants could be recruited and consented before oral feeding started. It was also agreed that the study information (i.e. the participant information sheets) should be more attractive and easier to read, with visual images replacing some of the text. The information also needed to be translated into relevant languages so that infants are not excluded because their parents cannot read English. Training for clinical staff on the study recruitment processes should be clearer, and reinforced regularly, with minimal time lag between training and study implementation. The study could also be more visible (e.g. with stickers and visual materials in the NNUs). In terms of recruitment for the interviews and observations, there should be an ‘opt-out’ rather than an ‘opt-in’ approach for the observations, and the interviews with staff should be shorter. An online questionnaire was thought to be more feasible for staff. It was felt to be particularly important that the site PI had the necessary influence and understanding of the research to promote the study and facilitate buy-in from staff. It was suggested that the training needs of each unit be assessed, and the training tailored accordingly.

Most of the discussion around fidelity focused on the training component of the intervention. The key suggestion was that there should be someone in each unit who had dedicated time to provide the training for the intervention to support the staff in its implementation. Therefore, the person delivering the training for the intervention would be different from the person delivering the training on the study procedures; for example, the latter could be a trial manager. Other suggested solutions were including the intervention training in mandatory training days and study days, and providing online training. The second main area for discussion around fidelity was the documentation. The key solution proposed was to capture data, especially the feeding data, electronically via a mobile application (an app). It was also proposed that the training resources for parents and staff could be included in the app, which would overcome some access problems. Further important solutions were making sure that medical staff were aware of and understood the intervention. Discussions on acceptability overlapped considerably with fidelity and similar solutions were proposed.

There was significant debate about the most important outcomes for a future trial, and the consensus was that a composite outcome was needed that covered feeding and parent–infant attachment. Short-term quantitative outcomes were said to be important, but focus on weight gain was felt to ignore the quality of feeds. Longer-term outcomes, such as feeding difficulties and oral aversion, were agreed to be important.

In terms of a future trial, it was agreed that a cluster design would be the most appropriate and that stratification would be needed to account for the different stages of NNUs’ transition towards family involvement and responsive feeding. Stratification by UNICEF UK BFI accreditation status was proposed to be the most practical way to achieve this.

The ADePT process

In the next stage the research team worked through the ADePT process to identify key problems. Type A problems (for the trial only) comprised the small proportion of eligible infants recruited, challenges in engaging staff and parents with documentation and data collection, and challenges recruiting staff to the study. There was one type B problem, namely parents and staff not adhering to all intervention components. Boxes 2 and 3 show the final evaluation and ranking of the solutions with further detail of the process presented in Appendix 5.

Avery et al. 89 suggest that the three predominant types of progression criteria from an internal pilot to a full trial are recruitment, protocol adherence and outcome data. Although our study was not a pilot trial, working through the ADePT process highlighted similar issues from our feasibility study.

In terms of recruitment, a key issue related to the point in their feeding journey at which infants were screened for eligibility and recruited to the study. The fact that 16 infants were ineligible because they had established oral feeding suggests that they were screened too late. Screening is further complicated as it is not a one-off event; infants progress at different rates and frequently not in a linear fashion, meaning that some infants may move in and out of eligibility over time. There appeared to be a short window of opportunity for infants to be recruited to the study as, once they showed readiness to start oral feeding, their progress could be rapid. Therefore, in a future study, earlier screening should increase the number of eligible infants. Furthermore, if infants are screened earlier, there may be more opportunity for the person taking consent to meet the parents to discuss the study. This could increase the proportion of eligible infants who are recruited. Therefore, we assessed training for care staff in the trial procedures in terms of timing of screening to be an important solution. However, a NNU is a very stressful environment for parents, and feedback suggested that many feel too overwhelmed to consider participating in research; therefore, low recruitment rates as a proportion of eligible infants may be inevitable. A further solution to increasing recruitment could be to have a research nurse who is embedded in the NNU to support screening and recruitment, and to increase the visibility of the study. The role of the PI is also critical to support the research. In one site the PI had limited research experience, and in another the PI was not sufficiently embedded in the NNU. Stakeholders proposed providing funding to backfill staff so that they could be released for interviews. In fact, this was provided to each site, but it is possible that it was not used in the most effective way to incentivise participation. Feedback from the PIs suggested that there is a shortage of suitably skilled practitioners to enable the release of staff.

There were also issues with recruiting parents and staff to the qualitative elements of the study. As a future trial would need to incorporate a process evaluation, this needs to be addressed. There appeared to be three challenges: time and privacy for interviews, consent procedures for the observations and, in one site, an issue of mistrust regarding confidentiality. Of the solutions suggested by stakeholders, we assessed as important shortening the interviews with staff and/or using an online questionnaire as an alternative to interviews. For parents, more flexible options could be available, such as conducting the interviews at home following discharge.

The main challenge to protocol adherence was implementation of the cascade training, which failed to reach all staff and took a long time. Some staff experienced too long a gap between the training and the commencement of the intervention, and, for some, there was confusion between training to implement the intervention (i.e. cue-based feeding) and training for the study recruitment procedures. The key proposed solution to overcome this was that there should be a dedicated individual in each unit whose role would be to provide the intervention training across all staff and disciplines. Training related to the research protocol and procedures, such as screening and recruitment processes, could be provided by a different person, such as an external researcher, to keep this differentiated from the intervention training. Although fewer challenges were identified with the information provided for parents, stakeholders proposed that, as well as providing information about cue-based feeding and referring parents to the study audio-visual resources, staff should model cue-based feeding for parents. This would probably require a cultural shift within units from the predominantly prescribed volume and scheduled feeding that was still evident to some extent in the study findings. Solutions for addressing this included aligning unit protocols and more engagement of medical staff in the intervention. Implementation of an audit and feedback process could contribute to such a culture change. 90,91 Our feasibility study achieved good capture of outcomes that were available from routinely collected data. The challenge was to obtain data on feeding outcomes. These data were intended to demonstrate fidelity to the intervention as well as feeding outcomes. Infant feeding data are limited in routine data and, in their current form, are driven by an assumed scheduled approach with a focus on prescribed volumes taken at each feed (i.e. quantity rather than quality of feeds). The main solution that would cover both a trial context and the real world was to provide the study information and collect feeding data through an app. Stakeholders, including parent representatives, agreed that this would be an acceptable and feasible solution for parents. The only concerns were around the hygiene of mobiles phones and access for parents who do not have a smartphone. Provision of dedicated mobile devices could overcome both concerns but would require additional resource.

The Stakeholder Advisory Group discussed the above solutions and the options for a future evaluation. One issue of concern was that there are some staff and parents who believe that cue-based feeding is the best approach and that all babies should be fed in this way. This casts doubt on whether or not there is equipoise. There are contextual factors that suggest that many NNUs are moving towards cue-based feeding driven by the move towards family-centred care, UNICEF neonatal standards that encourage responsive feeding68 and the British Association of Perinatal Medicine toolkit on Optimising Early Maternal Breast Milk for Preterm Infants that alludes to a forthcoming toolkit on transitioning to responsive feeding. 92 This is further complicated by the evidence from our work that many staff believed that they were implementing cue-based feeding although parents’ experiences were not always consistent with this view.

The two options favoured by stakeholders were (1) a cluster-randomised control trial potentially using a stepped-wedge design or (2) a quality improvement project to implement and optimise cue-based feeding. Further work is probably needed before this decision can be made. The proposed next steps were (1) to co-design with parents and test an app that would incorporate both the intervention and data collection, and (2) to survey all 220 NNUs in the UK to explore the approach to transitioning to oral feeding and willingness to participate in a multicentre trial. The latter would extend and update the selective survey undertaken in the first stage of this project.

Summary

In this chapter we have described the process undertaken to co-develop with stakeholders the implications of our work for optimising the cue-based feeding intervention, and preferences for the future evaluation of cue-based feeding. A systematic approach guided by the ADePT framework was used. The key solutions to challenges relating to recruitment, protocol adherence and outcome data focused on screening infants earlier in their feeding journey, modifying and expanding staff training, and incorporating the intervention and data collection into an app. Equipoise is an issue, driven to some extent by current practice recommendations, standards and toolkits. Further work was suggested to be needed to co-design the app and assess comprehensively current practice across the UK prior to a decision on whether the intervention should proceed to be tested via a cluster RCT or be implemented using a quality improvement approach.

Summary of main findings

Strengths and limitations

There were several strengths and limitations of this study. We applied a rigorous, systematic and comprehensive approach to developing the evidence base for the intervention. The systematic review followed established review methodology to identify and assess for risk of bias in all relevant studies. We analysed the interventions not just for their effect, but also for theories and BCTs. A limitation was that most of the 24 included studies were assessed to be at high risk of bias. We extracted critical learning from rapidly but rigorously conducted case studies of three purposively selected NNUs, each using different approaches to cue-based feeding. A limitation was that the case studies were time-limited snapshots. The telephone survey of NNUs was informative in highlighting the range of approaches to transitioning from tube to oral feeding and intentions to implement cue-based feeding. A limitation was that owing to time and resource limitations, we were only able to sample a small proportion of UK NNUs. A further strength was that we generated primary qualitative data to increase our understanding of parents’ and practitioners’ knowledge, experiences and views of the transition to oral feeding. This helped to inform the development of the Cubs intervention.

A considerable strength of the study was that we were able to use the evidence to develop the intervention using a structured co-production approach. We were able to build consensus on components and implementation with involvement of a wide range of stakeholders, including health-care practitioners and parents. The resulting intervention was well received when implemented during the feasibility study. A limitation of the intervention was that we did not have the resources to consider translation to different languages to increase inclusion. A further limitation was an underestimation of the challenge of reaching all staff with the training materials, which led to a delay in commencing the recruitment phase of the study.

A strength of our work was the multimethod feasibility study in three purposively selected NNUs of different sizes and levels, which served different populations. A limitation was that, by the time we commenced the feasibility study, all three units had gained UNICEF UK BFI accreditation. We acknowledge that further challenges may have been evident in units that were not accredited. NNUs are challenging environments in which to implement change. Although challenging, we recruited > 80% of our target number of infants with high retention in the study until discharge. A limitation was the small number of staff and parents participating in qualitative interviews and observations. The small number of medical staff who engaged in the training and interviews was a significant weakness. Nevertheless, we generated rich qualitative data. A strength was our approach to analysis using two theoretical frameworks: Proctor et al. ’s83 implementation outcomes and normalisation process theory. 85 We also used a theoretical framework, the ADePT process,87 to facilitate a structured and transparent approach to decision-making.

Stakeholder involvement and parent engagement throughout the study were key strengths of our work. Parents were engaged through the Parents’ Panel, and their feedback and advice was crucial, as described in Chapter 2. The involvement of the charity Bliss was essential to facilitate parental involvement. We retained parental involvement throughout the study, although we had much stronger engagement in the early stages. Towards the end, only one parent was able to attend the second workshop and only one attended the final Stakeholder Advisory Group meeting. An alternative could have been to seek new members for the Parents’ Panel as the study progressed. It was inevitable that parental responsibilities and commitments changed over time as their infants grew older. A further limitation of our approach to parental engagement was that the parents did not represent the diversity of parents who experience care of an infant in a NNU; for example, we did not have parents from minority ethnic backgrounds. We chose a pragmatic approach to recruiting parents through the charity Bliss, but a more targeted process is needed to optimise inclusion and diversity. We had excellent engagement from health-care practitioner stakeholders, except for medical staff. This was a limitation, and alternatives to attendance at workshops may be needed.

There is no doubt that the COVID-19 pandemic had an impact on our study. We had to close the study to recruitment earlier than intended; even a few more weeks could have increased the number of qualitative interviews and follow-up rates. It probably also had an impact on maintaining parental engagement to the end of the study.

Implications for a future evaluation

Our study developed an evidence-informed cue-based feeding intervention for the transition from tube to oral feeding for infants in NNUs. Our systematic review identified that previous studies comparing cue-based feeding with scheduled feeding are small and have methodological limitations, suggesting a need for a larger-scale rigorous evaluation. The intervention was implemented in three NNUs to assess feasibility, although, without a control group or randomisation, some questions about progression to a full trial remain unanswered. Our study differed from most studies included in our systematic review as it was an intervention development and feasibility study without a comparison group. Our study also differed in the level of stakeholder engagement in the intervention design and in developing the recommendations for a future evaluation.

There were two further related challenges. First, there was evidence that health-care practitioners and, to some extent, parents are not in equipoise, believing that cue-based feeding is the preferred approach to the transition from tube to oral feeding. The second issue is that our telephone survey, albeit based on a small sample, suggested that most NNUs are at least considering implementing cue-based feeding. This is further influenced by contextual drivers such as UNICEF UK BFI neonatal standards. 68 This suggests challenges regarding the nature of standard of care that would be the comparator in a trial. This is complex, as there was some evidence that staff believe that they are implementing cue-based feeding, albeit while retaining elements of a scheduled approach. Given that the intervention incorporates staff training, resources displayed in NNUs and changes to a feeding protocol, a cluster stepped-wedge design was considered the most appropriate approach. Further work to undertake a comprehensive assessment of the 220 NNUs in the UK would provide a stronger basis for deciding if a trial design is appropriate. An alternative approach could be a quality improvement design.

The cluster design is ideal for interventions where unit-level implementation would be more practical and viable than individual randomisation. For this intervention, for example, individual randomisation would be difficult in a NNU, as nurses would be required to continually switch between feeding regimes for different infants. The design also reduces the risk of cross-contamination, as the two feeding regimes operating side by side would be likely to lead to confusion and potentially queries from parents about why other infants have different feeding regimes. Stratification would be worthwhile, as it would increase efficiency, and could be done on size and possibly by current type of feeding approach (fixed, mixed) and by UNICEF UK BFI accreditation status. It would also reduce potential biases. Alternatively, minimisation could be employed to provide balance on a number of key factors.

Recruitment bias is an issue in cluster designs and can be reduced in several ways. First, recruitment could happen at the same time as allocation to intervention, so that the units, and potentially mothers, do not know which regime will be followed at that time. However, this has the drawback that only infants in the NNUs at that time would be enrolled. This may reduce power but in a cluster trial the design effect is reduced with a lower number of recruits per unit. This effect could be reduced by collecting data on infants’ weights, if this is the primary outcome for all infants. Of course, it would have to be explained to parents before recruitment that selection of intervention is random, and before the unit gives consent to participate in the study. Any subsequent trial would follow the Ottawa Statement on the Ethical Design and Conduct of Cluster Randomized Trials. 93 Intention-to-treat analysis would be utilised to reduce the potential for bias due to non-compliance. Control units could, pragmatically, be a mixture of different approaches, so including co-interventions, though clearly excluding cue-based feeding, would help to maximise recruitment and reduce bias. Statistical issues of clustering would have to be considered in sample size estimation and subsequent analyses. 94

Key weaknesses in previous research are summarised in the Cochrane review. 1 A future trial of the Cubs intervention could overcome these weaknesses through testing an intervention that has been co-designed with relevant stakeholders; focusing on infants at the transition from tube to oral feeding; ensuring that the trial has an adequate sample size and length of follow-up; selecting outcomes that are important to parents and staff; and involving stakeholders in the trial design.

Through our feasibility study we were able to identify solutions to some of the challenges encountered that have implications for a future evaluation. To optimise recruitment, infants should be screened earlier in their feeding journey so that consent can be taken before the infant progresses to start oral feeding. The study procedures will need to be clear that the intervention should start only when the infant is assessed as being ready to start attempting oral feeds. Other factors that could be considered to enhance recruitment are embedding a research nurse or midwife with additional clinical duties within the NNU to support staff, and improving the training of staff for their role in approaching parents about the study. This could be highly effective in engendering trust among other staff, and could be cost-effective. If observations are included in a future process evaluation, a more pragmatic approach to obtaining consent from clinical staff is needed.

The main challenge to protocol adherence was the cascade training, which did not reach all staff in a timely manner. Alternative approaches are needed, and identifying a dedicated trainer within each NNU could overcome this. There was also evidence in the findings that there remained elements of a prescribed volume and schedule approach. A change in NNU culture is needed, which was beyond the scope of the intervention developed in this study. This will require engagement of medical staff, alignment of unit protocols and strategies to enhance staff motivation. The documentation of feeding progress and outcomes was too onerous and requires both a reduction in the amount of information requested and to be more user-friendly and accessible through digitalisation.

We did not have a control group and therefore were unable to test randomisation procedures or calculate the sample size needed for a future trial. Pilot work is needed to address this. Pilot work is also required to assess the feasibility and acceptability of a composite primary outcome that captures not only infant growth but also a measure of parent–infant attachment or interaction. There was considerable support for the assessment of longer-term feeding outcomes for a future evaluation, and this could also be assessed in pilot work. Finally, pilot work would also provide an opportunity to refine the entry criteria.

Conclusions

Our work has demonstrated that it is feasible and acceptable to implement an evidence-informed cue-based feeding intervention for the transition from tube to oral feeding for preterm infants in NNUs. The intervention was well received, but the training element needs to be improved. Further work is needed to digitalise the intervention and feeding outcome data collection, and to assess the feasibility of a cluster RCT, noting some evidence of an existing lack of equipoise.

Contributions of authors

Alison McFadden (https://orcid.org/0000-0002-5164-2025) (Professor of Mother and Infant Public Health) was the PI for the study. She designed the study, drafted the report, contributed to developing data collection tools and analysing all the data and contributed to the systematic review and developing the manual. She contributed to developing and delivering the training, facilitating the stakeholder workshops and conducting the ADePT process.

Bronagh Fitzpatrick (https://orcid.org/0000-0002-2068-2761) (Post-doctoral Researcher) project managed the study and contributed to drafting the report and conducting the case studies and telephone survey. She developed the manual and delivered the training. She contributed to the systematic review and facilitating the workshops. She chaired the Parents’ Panel.

Shona Shinwell (https://orcid.org/0000-0001-9369-9698) (Clinical Academic Research Fellow) contributed to drafting the report, developing the intervention, analysing the quantitative and qualitative data and conducting the ADePT process.

Karen Tosh (https://orcid.org/0000-0002-5724-3383) (Lecturer in Children’s Nursing) contributed to designing the study, drafting the report, conducting and analysing the case studies and contributed to the systematic review and developing the intervention and training.

Peter Donnan (https://orcid.org/0000-0001-7828-0610) (Professor of Epidemiology and Biostatistics) contributed to drafting the report, analysed the quantitative data and assessed the feasibility of a future trial.

Louise M Wallace (https://orcid.org/0000-0003-3770-0580) (Professor of Psychology and Health) contributed to drafting the final report, analysing the qualitative data and conducting the ADePT process. She chaired the Stakeholder Advisory Group.

Emily Johnson (https://orcid.org/0000-0003-4005-3169) (Specialist Speech and Language Therapist) contributed to drafting the report, conducting the case studies and developing the intervention.

Steve MacGillivray (https://orcid.org/0000-0002-1933-6431) (Reader in Mental Health) contributed to drafting the report and conducting the systematic review.

Anna Gavine (https://orcid.org/0000-0003-3750-2445) (Lecturer in Mother and Infant Health) contributed to drafting the report and conducting the systematic review.

Albert Farre (https://orcid.org/0000-0001-8970-6146) (Lecturer in Adolescent and Child Health) contributed to drafting the report and analysing the qualitative data.

Helen Mactier (https://orcid.org/0000-0001-6154-5758) (Consultant Neonatologist and Honorary Clinical Associate Professor) contributed to drafting the report, developing the intervention and assessing the implications for future research.

Data-sharing statement

All data requests should be submitted to the corresponding author for consideration. Access to anonymised data may be granted following review.

Patient data

This work uses data provided by patients and collected by the NHS as part of their care and support. Using patient data is vital to improve health and care for everyone. There is huge potential to make better use of information from people’s patient records, to understand more about disease, develop new treatments, monitor safety, and plan NHS services. Patient data should be kept safe and secure, to protect everyone’s privacy, and it’s important that there are safeguards to make sure that it is stored and used responsibly. Everyone should be able to find out about how patient data are used. #datasaveslives You can find out more about the background to this citation here: https://understandingpatientdata.org.uk/data-citation.

Disclaimers

This report presents independent research funded by the National Institute for Health 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, NETSCC, the HTA 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, NETSCC, the HTA programme or the Department of Health and Social Care.

Databases searched

The databases searched were EMBASE (1974 to 6 August 2018), HMIC (1979 to May 2018), Ovid MEDLINE(R) ALL (1946 to 6 August 2018) and Midwives Information and Resource Service (MIDIRS): Maternity and Infant Care.

Date range searched: 1988 to 26 April 2013.

Date of search: 7 August 2018.