Psychosocial support for families of children with neurodisability who have or are considering a gastrostomy: the G-PATH mixed-methods study

Research objectives

1. Establish a national (UK) picture of models and practice via an online survey.

2. Compare the implementation and operation of four contrasting service models and practice using a collective case study research methodology.

3. Analyse how support is accomplished in different organisational structures, including health and social care sectors, and the role of schools in supporting children and families.

4. Provide rich description of contrasting supportive practices and the interface between services and other key agencies.

5. Identify different features of service configurations and supportive practices and analyse their costs, specifically to (1) assess the resources required for each of the models considered and how these costs impinge on health and social care services and (2) estimate the costs of providing psychosocial care.

6. Identify whether or not any models of support utilise service user involvement or third-sector organisations (i.e. families with experience of GFT feeding or voluntary organisations) and analyse how service users/family support organisations interface with the formal health and social care sectors and how they are resourced.

7. Analyse professionals’ understanding of support issues and how services provide support, the support needs of young people and their families and how these were met.

8. Derive best practice guidance on support issues from an explication of the service models identified in the study and service specifications and make recommendations for change to inform commissioning decisions and set standards of support for children and families.

9. Make recommendations for embedding support for a wider constituency of children with complex health needs and their families, including recommendations for training and skilling the wider children’s workforce, by deriving general principles of how services can provide support and where in the care pathways this should happen.

Selection criteria case study sites: integration and support

Previous research on organisational case studies has selected sites according to best practice or performance criteria; for example, the Birthplace study45 selected sites according to the findings of the 2007 Health Commission Services Review. However, there were no policy recommendations to inform the selection of neurodisability services. Given the lack of guidance on models of service delivery, and how services should provide support, we identified cases in terms of the integration of PSS as a proxy for the structured support recommended in evidence reviews and also aimed to look at variation. We developed a four-step approach to the selection of cases (Figure 2).

FIGURE 2.

Development of a case study site selection framework. CSO2, case study site 2; CS03A, case study subsite 3A; PI, principal investigator.

Steps 1 and 2

We initially aimed to identify services from the WBS using the criteria of support (step 1) and integrated care pathways (step 2) (Figure 3). We operationalised ‘higher’ levels of support as involving designated staff offering PSS and written care pathways that included PSS. Our operational definition of integrated care, as a care pathway approach, was ‘structured multidisciplinary care plans which detail essential steps in the care of patients with a specific clinical problem’. 46 The standards of integrated care pathways are provided in step 2. We asked respondents in our WBS to indicate which of the criteria outlined in steps 1 and 2 applied to their service to guide our case study selection. 47

FIGURE 3.

Preliminary selection criteria integration vs. support.

Step 3

Step 3 involved using the learning garnered from our first case study and WBS and applying these findings to the selection of subsequent cases, drawing on ideas from Stake’s progressive focusing methodology,48 which allowed additional criteria to emerge in relation to our study aims. For example, as it became clear that some services had examples of best practice but no formal care pathway, and on the advice of members of the PAG, we adopted a more flexible approach to the selection of our cases using theoretical criteria. 48

Step 4

Willingness to participate was also an important criterion as an indication of staff or organisations’ willingness to learn or show case examples of best practice. Interest and ‘opportunity to learn’ were therefore privileged over ‘typicality’ of service. 39

It should be noted that, although the initial criteria of integration and support (see Figure 3) informed the selection of case study 1, the results of our WBS, and our PAG, suggested that not all services would have written care pathways and that support was often provided generically as part of the multidisciplinary team (MDT). Assumptions that care involving designated staff equated to a higher level of support was problematised through our findings. Indeed, the concepts of ‘high’ and ‘low’ integration and support are themselves discursive constructions and highly contested in the absence of evidence and guidance. Therefore, we chose additional sites according to theoretical sampling and what those services could tell us about PSS initiatives using theoretical sampling based on the learning and experience garnered from the field work.

Selection of case study sites

All case study site services, apart from the service at case study site 2 (CS02), which was described as a generic service, were specialist services in which MDTs delivered care to children with complex feeding needs. All sites referred to specialised tertiary centres for the placement of a GFT. Three of our cases were based in England and one in Scotland (we do not provide exact regional locations to preserve anonymity). Two of our case study sites were in the 20% most deprived authorities in England, with approximately one-fifth of children living in low-income families; one of our case study sites was in one of the least deprived authorities, with approximately 7% of children living in low-income families. 49 The cases and rationale for their selection were as follows:

• Case study site 1 (CS01) was selected because it met our initial criteria of integration and support. We initially designated this site as high in integration, between health and education via a co-located special school, and support (defined as availability of designated staff). In addition, there was a strong parental network organised around the school.

• Case study site 2 (CS02) was chosen because it was an example of a generalist, rather than a neurodisability, service, where children were cared for by a MDT and staff had generic responsibility for all children but specialised in caring for children with complex feeding needs. Staff were supported by the NMCN CEN, which serves as a forum for disseminating best practice across Scotland. The WBS identified that the NMCN CEN had developed a training module for staff specifically on the importance of providing emotional support to parents when making decisions about GFT feeding. This was also underpinned by policy, that is, a best practice statement and guideline based on research findings. 50 Education and training were identified as aspects of best practice in our WBS and as gaps by some staff in our first case study.

• The services of a psychologist were available at case study subsite 3A (CS03A) but were not available at case study subsite 3B (CS03B), reflecting an inequality in access to resources, although both services were homed in the same NHS trust. Both sites referred children who needed a GFT to the same tertiary centre [case study tertiary site 3C (CS03C)], which had recently implemented a nurse assessment model. We selected CS03A to specifically examine (1) the experiences of parents of younger children and (2) the role and function of designated staff to offer support (e.g. a psychologist), a theme raised in the WBS. The comparison was felt to be useful in understanding commissioning, staffing arrangements and resources: themes identified in our WBS as barriers and levers in the provision of PSS.

• Our final site, case study site 4 (CS04), could be described as a hospital-based, district service. There was no written care pathway. This site was a mini case study because the recruitment of parents was particularly difficult. Moreover, the tertiary referral centre did not have capacity to participate.

Interviews with key stakeholders

We conducted semistructured interviews and focused group discussions in each case with a theoretical sample of service managers, staff (health care, social care, education) and parents and children (the children were included whenever possible). In addition to face-to face interviews we offered participants telephone or Skype™ (Microsoft Corporation, Redmond, WA, USA) interviews. We used visual mapping of service configurations as an interview aid with professionals, where possible, to better understand referral pathways in local systems of care. 51 Interview schedules were semistructured and required a degree of flexibility depending on where families were in their feeding journey (see Report Supplementary Material 2 and 3). Flexible interviews were necessary for children to accommodate their communication preferences.

Observations and document review

This was not an ethnographic study so the role of observation and review of documents operated as follows: prior to the interviews we obtained key documents relevant to the case study (e.g. service documents, care plans, care pathways, mission statements, protocols on eating and drinking) to sensitise us to the context of service delivery and identify aspects that could be corroborated in interviews. We conducted observation of selected aspects of services at key junctures, including appointments and meetings with family groups. Non-participant observations aimed to provide a baseline understanding of the context of care and sensitise us to the influences of this on care delivery. Both observation and document review helped to highlight issues of relevance that could be explored in interviews and assist with triangulation of data.

Data management

Interviews were audio-recorded, transcribed, de-identified and stored in NVivo version 11 (QSR International, Warrington, UK). Key documents and notes of observation were added to the NVivo database alongside interviews.

Analysis of data

The lack of research on how services provide support to children and families, coupled with diversity of provision due to a lack of guidance on how services should be organised, and the complex nature of family journeys (which often traversed multiple health-care organisations, education and social care), provided the rationale for a largely descriptive approach to the case study to provide sufficient contextual detail. 52 Our approach to analysis involved four levels:

1. description of services to highlight how support was accomplished across each service configuration and their contexts, building on the findings of the WBS

2. visual service mapping highlighting linkages in and between services to illustrate pathways and complexity derived from interviews and checked in feedback sessions

3. themes highlighted in the WBS

4. theoretical model of decisional conflict.

The original plan was to analyse the qualitative interviews using a framework analysis53 combined with a narrative approach to provide rich illustrations and storied accounts. However, the case study generated to some extent heterogeneous data in line with our progressive focusing methodology, which allowed us to focus on different strengths of services in each of our cases to illustrate how support was accomplished across a diverse array of organisations we have loosely termed ‘local service configurations’. Therefore, our cases cut across health care, social care and educational provision and across different sectors of provider. This provided a rationale for a prioritised focus on a thematic analysis,54 which we felt was more sensitive to the different perspectives and the dynamic nature of the project.

An analytical framework was developed in relation to the research questions, findings from the WBS and model of decisional conflict. 1,55 Care was taken to attend to similarities and differences in the data in and across case study sites using principles derived from constant comparative analysis where possible. 37 Data were organised in relation to our theoretical framework and are presented in a synthesis model in Chapter 6. It should be noted that not all key themes are presented in every case study site to the same degree of analysis; in some cases there were no data relating to that theme or there was less evidence of a particular phenomenon in that setting.

Theoretical model and data synthesis

We drew on Manhant et al.’s1,55 model of decisional conflict as a way of organising and categorising data specifically on decisions about feeding. Based on a systematic review of qualitative interview studies, the model outlined three sources of decisional conflict between HCPs and families arising from the recommendation of a GFT, including the values that parents attach to the meaning of feeding and GFTs; the child and family context, including unique family circumstances; and processes of care, which includes characteristics of organisations or care configurations, how these and HCPs interact with families to make decisions and the role of support and information. This theoretical framework informed the interview schedules as well as the analysis and synthesis of data.

Data synthesis

We collected and analysed the WBS (phase 1) and the case study data in two consecutive phases, with the qualitative case study being the primary component. Themes identified in the WBS were explored in greater depth in interviews in the case study and used to identify case study sites for phase 2. The phase 2 case study data were used to inform the scenarios in the WTP study in phase 3; otherwise, analysis in each phase remained distinct. We present findings in terms of a synthesis model that takes into account structures and outcomes in addition to processes of care and populate this model with our descriptive themes in Chapter 6. 56

Recruitment of staff and families

All staff and families were approached by the appointed principal investigator or research nurse at each case study site. The research team did not have direct contact with families initially. The research nurses contacted families by phone to advise them an information pack would be sent out, with their permission, inviting them to participate in the study. Packs were then sent out and followed up with a telephone reminder by the local staff 2 weeks later. Health-care staff received e-mail invitations advising them about the research, followed by the information packs and e-mail reminders. The local principal investigators and research nurses also sent out packs to school-based staff where the clinical teams delivered clinics. Consent forms were returned to the research team. Parents consented for their children to participate. Inclusion criteria are described in Appendix 3.

Study 1: estimation of resource use

Questionnaires on resource use concerning health service appointments were sent out to 103 parents and carers of children who had had a GFT or in whom placement of a GFT was being considered (see Report Supplementary Material 4). Information requested included demographic data concerning the child; details of HCP appointments related to the GFT in the previous 12 months; the length of each appointment, who was present and what issues were discussed; and whether or not emotional PSS was given and, if so, for how much time. Further questions asked if their problem was resolved and if they would have liked specific appointments to discuss emotional psychosocial issues. Questions were also asked about their satisfaction with each appointment and if they were offered the opportunity to talk to other parents or families and, if not, if they would have liked the opportunity to do so. The questionnaire also asked how satisfied parents were overall with their care on a scale from 1 to 5, and provided free-text comments on their child’s feeding and the PSS that they received or would like to have received. Questionnaires were also sent to HCP staff (see Report Supplementary Material 5). In Chapter 4, we report mainly on data from the parental questionnaire as well as reporting on the training and confidence in supporting families from the professional questionnaire.

Study 2: willingness to pay

Parents and HCPs were asked to value two different services: a hypothetical model of care that reflected usual practice across all four case study sites (service A) and service B, which reflected an enhanced care package of support based on data gleaned from the interviews and organised around the principles of structured and consistent support. 1 They varied slightly in terms of professional support and the frequency and type of psychological support offered. The aim of such questioning was to determine what value parents would put on each service relative to the other and, therefore, which model of care (service A or B) they would prioritise and thereby express a strength of preference for, using a hypothetical ‘purchasing price’ as a measure of such value (see Report Supplementary Material 6 for descriptions of the two hypothetical service scenarios).

The questionnaire and scenarios were piloted with our PPI parent group for ease of completion and among members of the PAG. Owing to the amount of time it would have taken to gain permission to disseminate the questionnaire through our case study sites, we decided to seek ethics approval through the University of Hertfordshire and distribute our questionnaire through professional and parental networks and social media, including Facebook (Facebook, Inc., Menlo Park, CA, USA; www.facebook.com) and Twitter (Twitter, Inc., San Francisco, CA, USA; www.twitter.com). The questionnaire was distributed via Qualtrics from 3 April 2019 to 28 May 2019.

Involving parents and young people in the process of research

A PPI reference group was formed comprising three parents and three young people and organised by Triangle (Brighton, UK) (co-applicant), an independent organisation that provides consultancy and advocacy services that enable children and young people to communicate about important matters. Triangle conducted the PPI on our behalf. The research team provided Triangle with a brief, or instrument, and asked for comment. Triangle liaised with parents and children on our behalf. In relation to children’s participation, a member of the research team usually observed the consultation. Our lay chairperson was involved in all aspects of the research study.

Generalisability

We aimed to use the case study to bring into relief approaches to embedding PSS in a variety of settings through interviews with a range of stakeholders, observations of care processes and a review of service documents. We concur with Sharpe et al. ,40 who argue that the different views of a range of participants across diverse contexts allows for some degree of generalisability, particularly when supported by additional data from other sources, in our case from phases 1 and 3 and the case study with data from across the four cases.

Interviews were coded by an independent researcher (JL) and checked by another researcher (EB). The chief investigator (GC) also reviewed 10% of transcripts for coding.

The case study has also been reported with regard to Rodgers et al. ’s58 consensus standard for the reporting of case studies in organisations (see Report Supplementary Material 1).

Member checking

Feedback sessions were conducted at two case study sites (CS01 and CS04), which involved face-to-face meetings with HCPs, and descriptive information about the case studies was sent to two sites (CS02 and CS03) for comment to validate the findings. The mapping of services was either sent to the principal investigators at each site for comment or discussed in feedback sessions.

Triangulation

Cohen et al. ’s59 definition of triangulation as an approach that maps out and explains the richness and complexity of a phenomenon by studying it from more than one standpoint reflects the way in which triangulation was handled in this study. To this end, a range of professionals and services were identified using theoretical sampling and we employed different methods to provide data (interviews, document review and observation). These different data sources provided a more balanced and holistic picture of services, which can often look, feel and be experienced as different from varying perspectives and across case study sites. Theoretical triangulation and synthesis was used by applying Mahant et al. ’s1 model of decisional conflict, and themes were compared across case study sites.

Limitations

It proved very difficult to disseminate a WBS nationally, despite extensive efforts, following a poor response rate using a professional database. Appendix 2 illustrates an extract from our dissemination log using the BACD database. This suggests that only 43 out of 225 recipients initially opened the e-mail containing the survey link sent to CDCs (our target audience) by the BACD on our behalf, with 11 out of 43 opening the survey link. Low response rates to surveys involving doctors are commonly reported, including rates of 29% in Canada61 and 30% in the UK. 62 Moreover, the research team had to rely on gatekeepers to disseminate the survey. Future efforts could second a member of the BACD to help with telephone reminders to improve response rates, and there is evidence that incentives increase participation;63 however, these approaches would have implications for resourcing.

Information request overload may have had some bearing. Anecdotal evidence from our networks suggest that clinicians receive many requests for information, including for research, commissioner requests and national professional body surveys. The length of the WBS may have been a factor affecting the differences in responses to questions, although evidence that length affects response fatigue is not conclusive;64 rather, WBS content, interest and motivation in the context of time pressures may be factors. However, responses to questions later in the survey did appear to wane. Given the lack of formalised and documented approaches to recording PSS, it is possible that respondents may have had to find the answers to the survey from different data sources and hence left the survey but then did not return to complete it.

As a result of the above, we are unable to claim that the results are representative, although we did identify topics worthy of further exploration in our case study. Methodologically, surveys are a crude tool for capturing data about complex services crossing boundaries. For example, there was some evidence to suggest that responses reflected PSS across services rather than individual services. Similarly, it proved difficult to classify services and models of psychosocial support from the responses provided in the WBS. There was a tendency of staff to respond with stock responses such a ‘MDT working’ as an aspect of best practice, which provided little insight into how teams functioned. This is a limitation of the method. There are also methodological issues regarding individual responses that ‘speak’ on behalf of whole teams and services, hence the need for a mixed-methods study that allowed us to explore the themes raised in this WBS.

Overall description of the case

Case study site 1 is described as a specialist clinical service and a co-located, charitable-funded special school and associated services. The clinic saw children attending the co-located special school and children in the local catchment who may have attended other schools in the area. Figure 4 shows the configuration of care at CS01. There had been a recent move in this setting to pool budgets for health and education, and clinical services received payment from education and NHS Clinical Commissioning Groups (CCGs), with agreed levels of funding for each child. However, for older children (aged 19–25 years), funding for health was described as ‘piecemeal’ (in an orientation interview) and dependent on NHS Continuing Healthcare assessment funding, reflecting inequities in service provision for young people.

FIGURE 4.

Configuration of care: CS01. CCN, children’s community nurse; GP, general practitioner.

The CS01 clinical service provided a range of specialist health-care services for children and young people with neurodisability and complex feeding needs/GFT. Referrals to outpatient services could be made through any NHS HCP and were accepted without prior approval, provided that they could be accommodated by the outpatient clinic allowance set out in the contract financial schedule. The allowance for outpatient clinics was therefore used to provide outpatient access to non-pupils (as described in the service specification document for the CS01 clinical service; details omitted to preserve anonymity). Pupils attending the co-located special school had an assigned paediatrician who visited the school and was linked with the school therapists, nurses and teachers. This helped to simplify referrals to the clinical services and the joint assessment of children.

Staffing arrangement

The nutrition team, which was part of the nutrition service that provides clinical care to children and young people with complex neurodisability, included a paediatrician, dietitian, nutrition nurse specialist and SLT and offered a dietetic clinic. Team meetings, which were attended by clinical staff, parents and school representatives, aimed to address any issues around feeding. A paediatrician was linked to the nutrition clinic and therefore came in direct contact with pupils at the CS01 school. There were three main clinics where children and families could be seen: a dietetic clinic, a drinking and management clinic and a joint surgical clinic (discussed later as an aspect of integrating community and tertiary services).

The dietetic clinic was run by a senior paediatric dietitian, lead clinician and nutrition nurse specialist. The clinic was for children and young adults for whom there are dietary or nutritional concerns, and all individuals fed by a GFT or nasogastric (NG) feeding tube were reviewed in this clinic once per year to ensure that their intake was nutritionally adequate (sources: Dietetic Clinics leaflet developed by the CS01 clinical service; CS01 dietetic clinic observation notes).

The eating and drinking management clinic was run by the specialist SLT, who was the clinical lead for eating and drinking difficulties. The service was for children and young adults who have complex eating and drinking needs and who did not attend the co-located school. Referrals could be made to this service by the SLT, general practitioner (GP) or paediatrician. The clinics were held monthly throughout the year. Funding was covered through a service-level agreement or individually commissioned service (source: Eating and Drinking Management Clinic leaflet developed by the CS01 clinical service).

Integrating support

A named paediatrician co-ordinated the MDT for nutrition and took responsibility for ensuring that PSS was provided. A key worker scheme operated through the school to cater for social care needs and represent children at health meetings. In general, however, the health team viewed the paediatrician as the ‘key worker’, although this term was not used. The joint surgical clinic included a consultant paediatrician in neurodisability, visiting paediatric surgeon from the local children’s hospital (tertiary centre), paediatric dietitian, paediatric SLT and nutrition nurse. This clinic was for children aged < 19 years with feeding issues that required a surgical opinion about the need for a GFT. Investigations were organised from the clinic. Referrals could be made to this service by either a paediatrician or a GP. Clinics were held six times per year (source: Joint Surgical Clinic leaflet developed by the CS01 clinical service). Surgery for the insertion of a GFT was performed at the tertiary hospital, with the surgeon leading the process and community nurses supporting the parents before and after surgery (CS01, S10, FG).

Psychosocial support

Parent peer support

Case study site 1 was characterised by a strong parental network facilitated by the unique and integrated relationship between the school and clinical services, an active group of parents, a support worker available to facilitate communication between the school and clinical services, a space for parents to meet, an on-site café and a site-specific social media site. Those parents who drove their children to school (car parking facilities are available) were often able to meet with each other. Both HCPs in the clinical services and staff members in the school commonly expressed the view that parental peer support was highly beneficial because it enabled parents to discuss issues openly, including the benefits of GFT feeding, without feeling undue pressure from professionals, and offered advice on post-gastrostomy care. This was confirmed by the parents themselves:

And that’s a lot of how you get information with kids like ours; it’s not because the professionals are telling you but because you hear it from other people, other parents.

CS01, P1, JI

I think often parents have more useful information than professionals . . . and they are easier to get hold of than, for example, consultants.

CS01, P2, FG

Seeking online medical advice and support from other parents about a GFT (pre and post GFT insertion) seemed to be a common practice:

Facebook is a great tool for parents to chat and share experiences and information. There’s a lot of groups now on Facebook, feeding groups and blended diet groups, reflux groups, so I got a lot of information from groups.

CS01, P2, FG

This was confirmed by a teaching assistant:

[The group] only started a couple of years ago; it’s a Facebook group and they also meet; they have regular social events. They have clinical information meetings as well, whereby whether you’re a parent that’s over in [name of place] or whether you’re a parent that’s 2 miles down the road, and you’ve got a query about your child but you don’t know whether to take it to a clinician, you can just pile it in and other parents are quite often very helpful at saying, ‘Oh, have you thought of this support or that support?’, or somebody says, ‘Oh, the gastrostomy is looking a bit sore; have you tried any tapes underneath it that will work?’.

CS01, S5, II

Parent coffee mornings were a feature of the partnership with the co-located school and information sharing at CS01. These sessions ran every half-term and were led by the support worker, an individual whose role was to link the school and clinical services but who also had a key role in supporting families around funding issues. The sessions also offered parents the opportunity to Skype, which was useful for parents who did not live within a reasonable commuting distance. An observation of the parent coffee morning session that aimed to address a specific issue (BDs) revealed that, in addition to parents/carers, they were attended by various professionals at both clinical services and the school, including a SLT, nurse, consultant paediatrician, dietitian and support worker as well as school staff members. Parents were able to share their concerns and experiences; staff members listened attentively and offered solutions.

The success of the parent support network was evidenced by the fact that parents at other schools (where parental networks were absent or less well developed) also made use of the network at CS01, which served as a hub of support. However, it was reported that not all parents accessed parental support events or group meetings at CS01 or on Facebook (CS01, P1, parents FG). This could raise the issue of equity of access to support for those parents who would have liked peer involvement.

Decision-making

Parents at CS01 were asked about the process of decision-making for a GFT. The emotional side of the decision was mentioned extensively by all parents, who referred to the decision as ‘horrific’, ‘unnatural’ and ‘the hardest decision as parents’ they ever had to make. There was a suggestion that having a GFT was not ‘what normal able-bodied people would do’ (CS01, P5, IPI). The importance of food and feeding was mentioned and a sense of failure when unable to feed a child. As one mother said:

I felt like I’d failed because the only thing you want to do for your child really is feed them, isn’t it? So, it’s the most important thing you can do for your child, it’s the first thing you do for your child and you continue to do it until they learn to do it for themselves, don’t you? . . . They were asking us to make a decision which, for me, was a very scary decision; again, it feels so abnormal.

CS01, P5, IPI

There were also concerns about how parents would manage feeding with a GFT in the context of family mealtimes, and how others beyond the family would react or cope:

I was worried about other kids feeling alienated by it, not understanding, and I didn’t want people to have any more reason than they already had to be disconnected from [name of child] really, to kind of judge him as different . . . I worried about the extended family; you know, so much revolves around eating together, going out and eating; I just couldn’t imagine it really.

CS01, P5, II

Despite the concerns and the emotions regarding the decision, there was a recognition that in many cases there was no option: the parent(s) had ‘run out of options’, were exhausted with the efforts of trying to feed the child and understood that the child’s health was deteriorating. Parents felt that presenting the GFT as a temporary, rather than a permanent, intervention was something that would have helped, giving them hope that the child might eat by mouth at some point in the future.

Thus, the key concepts for parents included fear of the unknown, feelings of guilt and maternal failure, learning to accept a different (‘abnormal’) way of feeding, the potential for stigma or perceived difference, the conflict engendered when weighing up the options or indeed of not having an option, and having to manage new nursing procedures and medicalised ways of feeding post surgery. The management of equipment and supplies was particularly burdensome owing to local procurement policies.

Decisional conflict

Staff at CS01 were cognisant of the emotional aspects of the decision for parents and families. When asked how staff supported the decision-making process, a dietitian explained that it was often an individualised decision depending on a family’s acceptance of the situation and child and family circumstances:

It really depends on the family; I think you have to make a decision, a pre-emptive decision on how you may feel the parents will take it, depending on the amount of time you’ve worked with them, their personality, the experiences they’ve had. Also, the experience of the child and the young person and what they’ve been through, if they’ve had a tube in the past, if other people have said it to them, if even they’ve thought about it as an option. So, I would certainly want to tell the family that we need to give everyone the benefit of the doubt at first, and we’re not at all pointing the finger or giving any blame to anybody, because I think the first initial thought is of guilt from the parents that they’re not providing the right nutrition for their child.

CS01, S8, JI

There were instances where the decision was not straightforward and it was generally agreed that multidisciplinary working, where decisions were shared and made collectively, was necessary before going down the safeguarding route. In comparison, staff in the community were perceived to be more isolated:

My feeling is, talking to some other therapists in the community, that they’re much more isolated in that decision-making process with families, and that’s quite scary. Whereas here, I think it’s much more of a collective responsibility.

CS01, S8, JI

There was a perception that therapists working alone in the community were much more likely to prescribe a nil-by-mouth feeding regime following video-fluoroscopy. This had resulted in a protocol at CS02 to ensure that video-fluoroscopy was repeated as part of a MDT approach.

When asked about potential conflicts, staff suggested that they usually agreed among themselves on the decision, but where there was a difference of opinion it was necessary to be consistent in the presentation of information to parents:

So, we need to be consistent, not necessarily to say we disagree, but if we disagree, we need to have that discussion without the parents there, not to confuse them. Not on the decision they make, but on the information we deliver.

CS01, S2, II

However, because of the value parents attached to feeding children orally, and the assumed quality of life associated with oral feeding, HCPs tried to go the extra mile to support families to manage the risk. For example, building in physical exercise to improve muscle tone and swallow function prior to eating a meal was one strategy personalised to the needs of a child.

The HCPs also spoke about the emotional impact on them of managing conflict and issues about safeguarding:

I can think of another instance where the parents were very much into healthy eating and the child was not thriving . . . and you can see a very chaotic child in front of you and I was not being able to . . . I wasn’t getting anywhere in my conversations, so it can be intensely frustrating at that point. [Murmurs of agreement.] And also, in the back of your mind, there is always the question, is this a safeguarding issue? [Murmurs of agreement.]

CS01, S9, FG

Guidance on eating and avoiding choking and aspiration was available for parents/carers and school staff (source: eating and drinking guidance at CS01). This drew on a system of traffic light codes that identified the level of experience needed by the person assisting the child. According to the document the assigned assistance code is reviewed every term by the speech and language therapy team.

A member of staff who was the lead for safeguarding recalled only one instance of involvement with a child who needed to be fed in a particular way because of safety considerations. Another member of staff described the conflict she felt between her safeguarding role and her role providing support for parents:

Obviously if there are significant safeguarding concerns, they need to be addressed, but I think with my role it’s very much about building up trust and relationships with families, and I’m very keen for that to be done.

CS01, S4, II

School staff and clinicians at CS01 therefore aimed to support families with decision-making, establish a relationship of trust, provide consistency of information and involve parents in decision-making. Staff members were mindful of safeguarding issues and formal proceedings were rare.

Blended diets

Blended diets, in which blended food that is prepared at home (as opposed to formula only) and administered through a GFT, were highly topical at CS01, with a parent coffee morning devoted to the use of BDs. Although not officially recommended, at CS01 there appeared to be a permissive approach towards BD use, with some staff members offering more proactive support. However, it was suggested that conflicts arose – not just between HCPs and parents but also between HCPs themselves – when the child was considered ‘very malnourished’ (CS01, Pr FG). Although school policies allowed food prepared at home to be administered in school, they did not permit the blending of school meals, suggesting a form of risk management whereby parents rather than the institution owned the risk. 20

Staff training

In relation to the training of carers, parents at CS01 took on a major role as ‘feeding experts’ and provided guidance and training to school staff on how to feed their children orally, whether that be in the school or in the community. In the FG, one of the parents (CS01, P2, FG) described their role as follows, with others in the FG signalling agreement:

I come in to do training, I’m training someone up, a different person, and I’m quite surprised to see how many children are eating orally; it’s nice to see it. But on the other hand, I do understand with some parents, there’s no choice and it is too unsafe. [Murmurs of agreement.] They’re very good at listening and we’ve trained very good carers here I think.

CS01, P2, FG

Thus, the important aspects about the training of staff from the viewpoint of parents was that they considered it their responsibility to ensure that staff knew how to feed their children and that they were best placed to give this guidance because of their often considerable experience of looking after one or more children with feeding problems.

School staff members attend induction training that includes training on supporting communication and eating and drinking for children and young people with complex neurodisability. Training on GFT use is provided by the nursing team. When asked about any training or preparation they may have had to offer emotional support to parents and families, clinical and teaching staff members highlighted a gap. One professional shared a concern about the lack of formal training in psychology and how approaches should be tailored to the needs of individual children:

I think [I] find it very difficult because we’ve had no training in psychology, and I think [name of professional] is typically on the forefront of it and I do feel quite vulnerable, because I don’t know the exact strategy to give them. I haven’t been trained in cognitive behavioural therapy or behavioural change or anything, and I find it sometimes a struggle to work out how I can motivate a family without having learnt psychology knowledge.

CS01, S8, JI

Teaching staff interviewed at CS01 were also parents of children with gastrostomies. Thus, they had experiences and backgrounds that influenced their knowledge of caring for children with complex feeding needs. One described her training and rationale:

I am gastrostomy trained and jejunostomy trained, and I am able to do everything that needs to be done in class. I also support parents, and the reason I trained to do gastrostomies and jejunostomies was not only because my [child] had it, but also because I like to know what I’m expecting my staff to do, and I also like to be able to converse with parents about what their children need.

CS01, S6, II

This teacher described that through caring for her own child her knowledge was gained through experience rather than formal training. Since becoming a teacher, she had received formal training, with observation by nurses and SLTs until competent. This was made possible by having the clinical services on site.

There were ‘strict’ controls in place whereby staff were not allowed to feed children until the training had been undertaken successfully. However, none of the teaching staff members referred to training to cope with the emotional aspects of GFT feeding.

Summary

In conclusion, while staff at the forefront of feeding, especially those in the school, were well trained for the physical procedures involved in feeding, it appeared that specialist training on how to provide emotional support to parents/carers or children was lacking. Informal sources of training through shared learning among members of the MDT, including psychologists where needed, were mentioned. Psychologists did not form part of the core team, although they could be drawn on as and when required to support staff. Some parents held the view that although emotional support was important there were potential barriers to how this could be provided. In a feedback session with parents, for example, concerns about referrals to professionals outside the team (e.g. a psychologist) were raised as these may be construed as the team ‘passing the buck’ and adding to the burden of appointments. Concerns were raised about the amount of time it would take to get an appointment with a specialist for PSS. Some parents also felt that, because the children often had complex needs, emotional support should be available for a broader spectrum of issues across the life course, not just related to feeding.

Overall description of the case

Case study site 2 was a generalist service with a highly specialised MDT for children with complex feeding needs. The team was employed by a Scottish Health Board within NHS Scotland. Children and families were seen at home, a clinic, a school or in other community locations. Referrals came into the service via paediatrician, paediatric dietitian, SLT or GP. The service served an area with a large rural catchment.

Staffing arrangement

Each child with complex feeding needs had input from a consultant paediatrician, dietitian, SLT and children’s community nurse (CCN) who cared for children until either GFT feeding was no longer required or they transition into adult services. The MDT aimed to see children with complex feeding needs as close to their homes as possible in line with the Health and Social Care Delivery Plan. 66

There were established links with specialists at the tertiary hospital (included a paediatric gastroenterologist and paediatric nutrition nurse specialist), although this was 45 miles away. Further assessment of children took place at the gastroenterology clinic at the tertiary centre before appointments with surgeons for the placement of a GFT. Post-gastrostomy care was the responsibility of the local MDT. The CCN team provided the immediate care regarding the gastrostomy site (stoma). The paediatric dietitian organised the initial delivery of the GFT feeds and equipment to the child and family home, after which the CCN team arranged the delivery of the feeds. Figure 5 shows the configuration of care at CS02.

FIGURE 5.

Configuration of care: CS02. HV, health visitor; OT, occupational therapist; SCBU, special care baby unit.

Integrating support

There were two mechanisms for integrating support: meeting around the child (MAC) and home enteral feeding clinic for children (HEFCC).

Psychosocial support

Parent peer support

There was no formal parental peer support group at CS02, although individuals were generally encouraged to engage with other parents and carers. The parents varied in the extent to which they felt that they needed or wished to relate to other parents. Reasons for not seeking out peer support included other work and family commitments, good existing personal and/or professional support networks, distance from the hospital and the feeling that there were unlikely to be other parents with similar issues.

Some parents had made informal contact with other parents (e.g. CS02, P8, II, and CS02, P6, II). One mother discussed informal meet-ups over coffee but stated that a more formal group event would not be something that appealed:

There are a couple of mums I meet up with occasionally and have a coffee with and just air everything out with them, and yeah, so I’ll have that but I’m not one for groups and things like that. That’s not my thing at all! I’d rather go out and have a nice cup of coffee and everything. Although that’s different though from having coffee with friends, because those mums have children with complex needs or disabilities, so they completely know where you’re coming from and they understand you.

CS02, P6, II

Clinical and teaching staff members at CS02 confirmed that they would suggest contact with other families if they considered that it would be helpful to the parents, though two did sound a note of caution that it would be important not to breach confidentiality. A paediatrician additionally raised several barriers to parents meeting up, including distance, geographical spread and lack of transport:

Part of the problem I think with this area is that you’re very aware that it’s quite difficult to put similar children together with other children, so you may be dealing with quite different conditions in their needs for gastrostomy feeding. And the parents are very spread out, finances and transport aren’t easy.

CS02, S1, JI

A social worker confirmed that they had experience of linking up families in the past that had resulted in unintended, positive consequences by families sharing the same carers, but issues of confidentiality were also raised:

We did run a support group for a short period of time, looking at linking families with other services and, as a result, they were then able to link up and to share: share difficulties, share positives, share advice of how to get things that they had previously got in the past. However, we can’t go around saying, ‘Oh, by the way, your neighbour up the street also has a child who is gastrostomy fed’. But by doing that support group, it linked them, and they were then able to link with each other and realise where in the locality [the other families] actually were. And as a result, quite a lot of them now use the same carers for their packages of care.

CS02, S2, II

Other clinical staff did not feel the need for a formal support group or for them to suggest linking with other families because there was a perception that families were doing this themselves using social media.

Decision-making

Parents at CS02 did refer to the process they went through when deciding to have GFT feeding, although these accounts were historical rather than recent. The decision to GFT feed was often a result of problems such as recurrent chest infections, compromised growth and an unsafe swallow:

. . . he needed more and at the time with the nasogastric tube, he was getting overnight feeds plus what they call bolus feeds. I think the decision was that it wasn’t quite enough and that fitting the PEG would make it a lot easier for him to cope with, and maybe less anxious about the whole business. And that kind of proved to be right, and we managed to get larger quantities into him that way.

CS02, P3, II

Not all parents felt negative about the decision if they felt that it was in the child’s best interest (e.g. CS02, P5, II) and that a GFT would benefit their child and help them to thrive. One parent (CS02, P6, II) admitted that she did not like the decision because it was ‘another step away from normality and into [a] medical [way of feeding]’ but accepted the decision when it was felt that there was no viable, safe alternative. The emotional and stressful nature of mealtimes also influenced the decision to elect for GFT feeding:

[The child] was about [age] when we got to the point where it was just becoming dangerous. [The child] was dangerously underweight, wasn’t eating, it was really, really stressful; every single mealtime, everybody was in tears. So, the nurse sat me down and said, ‘I think we need to be a bit more serious about this’. And it was the best decision ever!

CS02, P5, II

Various strategies for managing conflict were proposed by HCPs (e.g. CS02, S6, FG, and CS02, S4, II). These included explaining the advantages of GFT feeding for the child, engaging the parents in the decision, and referring the parents on to other professionals who may be better placed to advise or who could offer more weight or gravitas.

Decisional conflict

Professionals also stressed the importance of a team approach and relationships with families in arriving at jointly agreed decisions so that disagreements were uncommon:

I think we’re quite lucky that we know our families really well and because we’re such a small team, it’s not like the families are seeing loads of different people. I think we’ve always come to a joint decision with families.

CS02, S6, FG

Strategies also involved emphasising quality of life and challenging ideas that GFT feeding was regressive. Advice that oral feeding could become less pressurised and more enjoyable was another strategy adopted, although this was not safe for all children. There was a recognition that parents had different views about a GFT and simple questions asking where they were in their thinking helped staff to assess families’ preparedness to accept a GFT rather than any formal assessment:

I think we recognise that, well, for no two people is it the same, but for some they just cannot function . . . so functioning with a gastrostomy is the way they want to go and for others it’s the exact opposite, that a gastrostomy is a heart-rending medical intervention taking away their ability to orally feed their child. So we’re upfront and we ask them [what they think about it], because essentially we will take them through the fact that actually in the vast majority of cases, oral feeding becomes a much more pleasurable, enjoyable, less pressurised and social activity where actually your feed can be supplemented by gastric feeding.

CS02, S5, JI

Professionals were also aware of the possible tensions in managing disagreements, including the need to respect parental choice while ‘balancing up duty of care and medical needs’ (CS02, S3, II) and acting as ‘advocates for all of the children that we work with’ (CS02, S9, JI).

At the tertiary centre, which received patients for surgery from a wider catchment area, HCPs could not recall a case where safeguarding had been used around the decision-making phase for gastrostomy.

At the school, one staff member raised the issue of feeding and safeguarding. They had had concerns about parents orally feeding a child who aspirated and felt that, because schools were a front-line service responsible for the health and well-being of children, they were duty bound to act (CS02, S9, JI).

Blended diets

There was little evidence of parents using BDs at CS02, although at the tertiary centre, which covered a wider catchment area, staff reported an increase in the use of BDs more generally. It is of interest, perhaps, that professionals felt that there were more issues around safeguarding in relation to parental adherence post gastrostomy, particularly in relation to BDs, than at the decision-making phase:

Sometimes the more tricky bit comes where the family is managing the feed after the tube is in.

OK.

So you’re possibly going to run into situations there more than the actual getting the tube.

The compliance associated with it.

Yeah, because some families choose to do their own thing, and it’s not always the best thing, but they think they’re doing right. And then there’s the blenderised diet, it’s a big thing that’s coming out and it’s not the wrong thing to do, but it’s whether they’re doing it right. So that’s more your area where you might raise child protection for us as opposed to the getting of the tube.

CS02, Pr1, Pr2, JI

These risks and tensions (a pattern repeated across all sites) are discussed in more detail in Chapter 6.

Staff training

Training resources with a focus on the emotional aspects of GFT feeding were available. This included the NMCN CEN DVD How Does It Feel: The Emotional Impact of Tube Feeding, which was developed based on consultation with parents and carers, a young person who was previously GFT fed and professionals. There was evidence of formal educational provision: the NMCN CEN e-learning toolkit that had been developed in partnership with the University of Stirling as an online module. This toolkit explores best practice in caring for and supporting children, young people and their families and those who support them. It develops knowledge and experience around the potential emotional impact for families when changing from oral to GFT feeding and emphasises the need for continuing support. However, it is not mandatory to complete the module and it does not include a formal assessment tool for assessing the need for PSS.

When asked about training, school staff suggested that they had received ‘informal’ training to learn how to use the GFT, although the nurses who provided the training would have considered this to be formal. They also referred to care plans and guidance that, although not formal training, gave the ‘necessary information’ to assist parents and families in the care and utilisation of the equipment. They also mentioned that a commercial company was sometimes involved in informing staff and/or parents.

A health visitor and school nurse expressed in an interview that they felt that, because they rarely dealt with children and young people with feeding difficulties, there was a danger of them becoming deskilled. School nurses were not routinely involved in decisions about gastrostomy, or any care, in relation to GFT feeding; rather, their role focused on supporting the parents and other family members. They also commented that, although the ANAs in schools received training on the technical aspects of care, there was a perception that this placed a lot of responsibility on them. The CCN team would be responsible for offering one-to-one support for school staff and regular training updates (e.g. actions to take if a child’s GFT falls out and giving medication via the GFT):

For the ANAs, the outreach nurses tend to come from the hospital and train them, but because they don’t train them about absolutely every aspect that they’re going to come across, it’s really difficult, and they can get quite anxious.

CS02, S8, JI

The CCNs also received annual updates on managing gastrostomies run by the enteral feeding companies that offered hands-on sessions. The issue of resources and competing needs for training was also raised by staff in schools:

Training obviously would be good, but where do you stop, because there’s so many conditions that would be useful for us to know a bit more about in the community. [Murmurs of agreement.] And whether our managers would say, ‘Yes, it’s important for you to go and learn about that, as opposed to going to do something else it’s difficult to judge’.

CS02, S8, JI

These differences contrast with staff in the special school at CS01, although in the case of CS01 there were more children who were GFT fed in the special school. Maintaining skills is more difficult when the number of GFT-fed children is small.

Summary

Guidance at CS02 recommended that parents be assessed for their PSS needs and referred to a paediatric psychologist, yet pathways for referral were not always clear and there was no mention of assessment tools by staff. Referrals to the paediatric psychologist were not routine but made on an ‘as and when required’ basis, although there was a feeling expressed by staff that the children may fall through a gap because they were mainly non-verbal communicators. Parents appeared to value relationship continuity. Parental groups were less in evidence than at CS01, with some parents preferring to receive support from professionals. Barriers to parental peer support were professional and parental perception of need and usefulness, and the geographical spread of parents, which may leave some parents at a disadvantage in remote areas where services are less developed. Child care and transport were also mentioned, and professionals had some concerns about confidentiality issues in putting parents in contact with each other.

Overall description of the case

Case study site 3 is described as two contrasting services that refer to the same tertiary referral service. Therefore, we present CS03 as two subsites (CS03A and CS03B) and one tertiary centre (CS03C) (Figure 6).

FIGURE 6.

Configuration of care: CS03.

Both subsites can be described as district secondary care neurodisability services and were chosen to highlight comparative differences in resourcing, organisation and ways of working, although both services were employed by the same trust. CS03B did not have a fully integrated feeding clinic or access to a psychologist for support (a feature, until recently, of CS03A) and, therefore, relied on the lead professional to co-ordinate care involving a ‘virtual’ team. Of interest was a recently implemented surgical nurse assessment model at the tertiary centre that aimed to integrate, or link, community and tertiary health services and support families through the decision-making process. Differences between CS03A and CS03B were attributed to historical factors and leadership.

Staffing arrangement

At CS03A there was a dedicated feeding team and clinics. Feeding team meetings took place at schools or in the community and consisted of a paediatrician, dietitian, SLT, OT and, sometimes, school staff. If the feeding clinic was conducted in school(s), the teacher, teaching assistant and CCN attended. There were different feeding clinics:

• the CDC clinic (only for children who are seen by the MDT and aged 0–3 years; most children are aged 0–2 years)

• three school clinics for children attending special schools

• two additional outreach clinics, one in the city and one in a rural area.

Referrals came from SLTs and paediatricians. Criteria for referrals to the feeding clinic were in place.

At the core of the CDC was a therapy group where preschool children are assessed. With regards to feeding, children were seen together with the SLT, dietitian and paediatrician, who were always present in the clinic, and OT or clinical psychologist for certain groups of children and parents. Between feeding clinic appointments, individual members of the MDT may have seen the child on their own and communicated with the rest of the team by e-mail.

Conversely, CS03B did not have a fully integrated feeding clinic and access to a psychologist was not usual practice. The community paediatrician, SLT, OT, dietitian and CCN saw the parent/child in a range of locations including clinics, at home, at school or nursery. CS03B offered clinics, school visits or home visits of usually a single profession, although there were some joint visits with therapists. In the past there had been joint visits involving a SLT and a dietitian, but, because the dietitian was hospital-based and employed by another trust at the time of conducting the research, joint visits were rare. Referrals to the service came through the GP, health visitor, school nurses or other primary care providers. Children seen were between the ages of 0 and 19 years.

Integrating support

Psychosocial support

Parent peer support

There was no formalised parent support group at CS03. One parent mentioned a small support group that she ran every 3 weeks on a drop-in basis at CS03B, but it was unclear if this was widely known about. Staff across all sites acknowledged the importance of peer support and reported that they put individual parents in contact with others. Staff also talked about how parents who were linked with each other to provide support, especially through the school system, although they recognised that peer support would not suit all families:

I have to say special schools are brilliant at that as well, because obviously they’ve got the child every day and they are feeding them, so they’re very good at working with parents and identifying other parents that also provide peer support, which we have found hugely powerful actually . . . But whilst they’ve been really helpful, for some families, it doesn’t work for everyone. Some families are quite private.

CS03A, S1, FG

Maintaining confidentiality was at the forefront of the minds of the team and participants stressed the importance of asking permission from families before putting parents in contact with each other. This put the onus on the new family to proactively seek out those parents willing to talk about their experiences. In an observation of a clinical session, the paediatrician suggested that the mother (CS03A, P9, II) could ask the school nurse if she could facilitate contact with another parent to see what a GFT looked like on another child. The mother said she would like that. Caution about how relationships were managed was advised, in particular matching parents and the accuracy of information passed on and shared:

You’ve got to be really careful that we’re not opening a can of worms [when introducing parents to each other]. We need to make sure that those parents are well matched and their children, because I’ve had parents who’ve said, ‘My friend’s little boy, he used to eat and then he got his gastrostomy, and then his speech therapist said he couldn’t have any tasters any more, and now he’s lost his swallow and now he will never eat again’. And I’m thinking, ‘Well actually that’s not quite true’. So, some parents can pick up what they want to hear, so you’ve just got to make sure you know exactly, have all your ducks in a row.

CS03B, S7, IPI

Although there was no formal parent support group at the tertiary centre, staff reported that they used Facebook, having created a professional group, and could invite parents to join the group and put them in contact with others. Again, they emphasised the role of special schools as the place where parents were likely to meet and know each other:

So what about the role of parental peer support, it’s not a common practice?

I always ask, not in a formal way, but I do always ask, ‘Do you know any other?’, because they tend to run in circles, these families.

They all go to special needs schools together, and they’ll say, ‘Oh, I’ve seen someone at my son’s school that’s got one and I’ve spoken to their mum about it’. I would always ask that, but we don’t have a formal way of saying, ‘Make sure you go and see someone with a gastrostomy’.

We do also have a Facebook, we as a team are on Facebook as a professional group as such and we have, lots of our parents who have gastrostomies, or children have gastrostomies, are on there as a group, so if there is a parent that wants to, and doesn’t have access, we can signpost them to that group and add them to that group so they could ask questions of those families. It doesn’t tend to happen very often because most of our parents, as I’ve said, their kids, if they’ve got complex needs, are in a special needs school and have got, they talk to other parents about how things work and all this sort of thing.

CS03C, S1, S2, FG1

Peer contact may not be consistently offered, however. Whereas one mother at CS03B stated that she would have liked the opportunity to talk to other parents before the GFT had been placed, another parent did not:

The other parents, what type of support do you get from them because they are in the same situation as I am, so how can you expect from someone if they are in the same situation as you are? . . . And I’m a type of person, I don’t want to put my pressure onto somebody else.

CS03A, P2, II

The research suggests that schools can play an important role in facilitating parental networking and, hence, support.

Decision-making

Staff were asked about strategies for facilitating the decision-making process when recommending a GFT. A community dietitian referred to having a conversation with the parents, providing as much information as possible (face to face and by reference to written information), showing pictures of the procedure and identifying the advantages of the procedure and the positive impact it can have on the child’s life. The importance of having good relationships with families was stressed, alongside an experienced team that allowed them to proactively identify those children who might need a GFT:

One of the things that we’ve got, one of the bonuses we’ve got is we’ve got good relationships with these families and we’ve got lots of experience between us so we can almost predict and pre-empt which children are going to almost certainly need one.

CS03A, S1, FG

Other staff also talked about persuasion rather than coercion, which they felt was neither possible nor desirable, and the importance of setting goals around nutrition, which facilitated decision-making for parents:

So we cannot force parents, of course, to accept the solution, so we will, we will work on the feeding as, but it will not be myself working on the feeding, it will be the paediatrician and the other professionals, and they will make home visits and try to help the parents implement feeding plans. And we set very specific goals for nutrition and safety of swallow and we monitor the child’s progress, and the question of gastrostomy is mentioned, discussed with them throughout, and when the goals are not met for natural feeding, then usually the parents get closer to that decision. We also try to bring them in contact with other parents who have had, who have gone through the same and went for a gastrostomy.

CS03A, S5, IPI

And some parents find the idea of having a gastrostomy and their child having to undergo surgery so abhorrent that it’s quite traumatic for them, so we just have to have repeated gentle conversations really and reassurance that what they need to focus on is what is in the best interests of their child.

CS03B, S2, IPI

Others felt that parents often came to their own conclusions on the need for a GFT. One parent (CS03A, P1, IPI) suggested that the decision had been very challenging because she was unable to admit to anybody how difficult it was ‘getting fluids into’ the child. The decision was helped by talking with other mothers whose children had the same condition and being ‘open and honest’ about how hard she was finding it in a feeding clinic appointment a few weeks later. When the team suggested a GFT, the mother felt relief because she had wanted the team to suggest it rather than her ask for it, which may have been interpreted as an admission of an inability to cope.

Another parent discussed the inevitability of the decision (drawing on military metaphors, e.g. ‘losing the battle’ and ‘biting the bullet’), and reluctance to accept the decision that they nevertheless later suggested, which proved very worthwhile:

So we started getting things in place, such as talking with the doctor about the feed and the gastrostomy, and we had the dietitian and all, lots of people involved, and the speech and language [therapist] because of the tongue movements trying to move the food round the tongue, all these people were involved. But we started losing the battle really, because we couldn’t get the volume of food we needed into her and she was losing, she was looking dreadful. So, we bit the bullet. I’m glad we did but at the time you think ‘What am I doing to her, poor thing?’. But at the time, you think ‘Yeah, I don’t want to do this’, but it was worth it, looking back it was worth it.

CS03B, P4, II

Decisional conflict

When making decisions about whether or not their child should have a GFT, parents typically described how it was often quite a lengthy process. Reasons given for the need for a GFT included the child not putting on weight or becoming ‘dangerously underweight’, not being able to take enough food orally or having problems with choking. Where disagreements arose, staff suggested that it was a matter of weighing up the risk to the child of not going ahead with the procedure. In terms of concerns about feeding, other professionals would be involved and, in extreme cases, the team would be prepared to refer the family to other agencies, such as social care (CS03B, S7, IPI).

A paediatrician summarised the process whereby parents would be encouraged to make a decision for their child to have a GFT. Again, a multidisciplinary approach appeared to be key in managing situations where there were safeguarding concerns:

We look first of all at do we have any safeguarding concerns and then look at whether we need to support the family down that route, but usually it’s taking a multidisciplinary approach involving the specialist from the hospital who would, if we’ve got to the point where the child is referred but then they don’t want to go ahead with it, and to see then what, how we can support the decision-making. If it hasn’t got to that, is usually, depending on urgency we try a softly approach, so that parents have time to get used to the decision, to be able to make it in a supported way.

CS03B, S8, IPI

Staff were asked if there were ever disagreements in the team about a child’s need for a GFT, and all suggested that it was rare for other staff involved with the family not to agree with the decision, although there was some recognition that differences in opinion could arise between staff in the community and acute sectors:

We in the community are good at looking at that bigger picture and saying, do you know what, don’t worry about that for the moment, or let’s just focus on this, or what is needed here, whereas the acute teams just see one problem and fix that one problem.

CS03A, S1, FG

A paediatrician (CS03B, S2, IPI) felt that conflict was more likely in families who were resistant to the child having a GFT placed. These conflicts can impose additional burdens on mothers, who are usually the ones to attend clinical appointments, in terms of managing information, emotions and relationships with the wider family, and may suggest a need for greater support.

Interviews with staff at the tertiary centre suggested that it was rare for safeguarding issues to affect the decision-making process and that the concerns and conflicts that did arise were similar to those reported at other case study sites. Strategies employed were to guide families by outlining the pros and cons of GFT feeding and not putting undue pressure on them. The nursing team were aware, however, of a few cases where issues of safeguarding had arisen, and they worked closely with the surgeons and the community paediatrician in these examples. In general, when safeguarding was raised, feeding was usually part of a bigger issue. In the community services there were procedures in place for safeguarding where the team could refer to the safeguarding nursing team, but, given that the teams had developed relationships with families, they felt that they were more likely to resolve the issue without recourse to a referral to another team. Procedures were also being formalised.

Blended diets

As at other sites, BDs occupied a grey area of practice. A service document outlined the tension: ‘although current policy does not recommend blenderised tube feeding due to lack of scientifically supporting evidence, providing balanced information that is currently available from a health professional is essential and good practice’ (source: service document, CS03). Staff adopted a pragmatic approach, given that parents were already adopting the practice, but would offer support only following a risk assessment in line with trust guidance (CS3A, S16), as was common across all case study sites. Resourcing support was a major concern. Staff made the distinction between ‘supporting’ and ‘recommending’ BDs (CS03A, S6).

Staff training

Apart from the psychologist at CS03A, none of the staff members identified any specific training to support the emotional and psychosocial aspects of families’ decision-making, though most drew on their initial professional training and some had backgrounds in learning disability or autism. Training mentioned by one or more participants included a counselling module, generic team training around how to have difficult conversations, statutory and mandatory training, continuing professional development (CPD) courses and a paediatric module provided by the BDA (not feeding specific). Informal learning from senior and experienced colleagues and through observations in feeding clinics was also mentioned. Similarly, although the paediatric surgical nurses were all paediatric trained, extensive formal training for complex feeding as part of nurse training had not been undertaken, only the occasional study day. Nevertheless, the nurses discussed ways in which they gained their knowledge, the main being ‘on the job’ or through ‘hands-on training’. However, training in PSS was not part of this training.

A view that offering support was ‘common sense’ was also suggested, but this perhaps underestimates the skills involved in supporting parents making difficult decisions. For example, one parent recounted that a clinician had offered to put her in touch with the school to ‘teach her’ how to feed her child. Although the suggestion was made with good intent, it was perceived by the parent as undermining her parenting skills.

Summary

Historically, CS03A has had input into the feeding team from a psychologist, but only a limited number of children were able to access this support through the CDC. Moreover, it was noted that these children and families may not be those most in need of support, illustrating ways in which a resource can create inequities within the service. Support was generally provided by the MDT and there were mixed views about the need for a psychologist for all families and how a psychologist could be best embedded in the MDT in ways that did not add to the appointment burden of parents. Relationship continuity provided by professionals known to families was mentioned as an important factor by staff and parents, as was the supportive role of schools. A recently implemented nursing assessment model at the tertiary centre responsible for placing gastrostomies, initially designed to reduce waiting times, ensured that the first point of contact was the nursing team, rather than surgeon, which may have allowed a space to provide support to families. The team also provided support to the local community nursing team and, indeed, when there were shortages of CCNs they were sometimes able to provide support, particularly at follow-up, which was formalised. Some surgical staff adopted strategies to persuade families to accept a GFT that would suggest a more utilitarian approach to food – that is, a child can or cannot eat and that technically a GFT offers a different way of delivering nutrition – which contrasts with the social meaning of food and feeding that many parents valued and community staff were aware of at the other case study sites. These different understandings may also reflect professional roles and location (that is, the type of advice offered by the surgical team in the acute sector versus that offered by professionals based in the community).

Overall description of the case

Case study site 4 provides a district disability, hospital-based service, in line with the BACD’s Quality Principles for Paediatric Disability Services. 70 It was described as a consultant-led team with expertise in paediatric disability. Although not a CDC, the service model drew on some aspects of a CDC but saw children aged 0–18 years. Professional expertise in the trust and in the community was drawn on as and when required. This site was selected because the composition of teams could vary and members were bought together in a flexible way depending on need and the expertise required. Housed under one roof, it offered a one-stop shop, particularly as, until recently, gastrostomies were placed on-site. Figure 7 illustrates the configuration of care.

FIGURE 7.

Configuration of care: CS04. BF, breastfeeding; VFSS, video-fluoroscopic swallow study.

Staffing arrangement

There was a three-tiered feeding pathway for children with complex feeding needs who could be seen in the neonatal clinic, neurodisability clinic or feeding clinic. There was no formal feeding care pathway. Any professional could refer to the paediatric disability service, although most referrals came from within the hospital because most children were already under the care of a consultant with a special interest in the field of practice. In addition, referrals could be received from primary care practitioners (e.g. health visitors, GPs) and special educational needs co-ordinators. Historically, gastrostomies were placed in-house by a consultant with gastroenterology expertise. Gastrostomies are now done in a regional paediatric surgical centre, although post-surgery support is devolved back to the local CCN team and the dietitian.

A feeding clinic was held monthly in the children and young person’s service based in the hospital, which usually involved a range of HCPs, including a paediatrician with gastroenterology expertise, a CCN, a dietitian and a SLT with expertise in dysphagia. Appointments were either unidisciplinary or multidisciplinary, with any of the above professionals participating. There were monthly operational meetings that included the paediatric disability team, specialist support team, disabled children’s social work team, CCNs and allied HCPs to facilitate communication and problem-solving. There was also a local specialist support team for children and young people with physical and medical needs in mainstream schools, comprising a senior specialist teacher, specialist paediatric OT and nurses. The paediatricians also ran clinics at several special schools that had specialist school nurses employed by the trust. Community nurses supported children post surgery and there was input from a dietitian. Gastroenterology nurses also provided support prior to gastrostomy. Community nurses played a key role in decision-making.

Clinical psychology formed part of children and young people’s services [Child and Adolescent Mental Health Service (CAMHS)]. In theory, support could be accessed through referral as required, including specialist clinical psychology support where children and young people were identified as having a learning disability. There was no psychologist in the paediatric disability service.

All of the above professionals provided support for families, either individually or together. This was on an ‘as and when required’ basis, tailored to the needs of the individual child, young person and family/carers. Some families were offered more frequent outpatient clinic appointments where the need was greater. Children with complex disability also often had an emergency health-care plan that highlighted what to do in an emergency and when to seek help and advice from professionals at that time. Telephone support was offered to families and they could contact any of the team members should they need further advice.

A local group run by a physiotherapist, OT and SLT supported children and their families/carers with complex needs from an early age, although this is not feeding specific. Referrals were made by consultants. The group, which ran for 10–12 weeks, provided an opportunity to assess children and families and identify additional needs. The group ran in term time and was limited to six places because space constraints limited numbers. In a feedback session it was felt that this group could provide a space to further embed emotional support for families considering a GFT for their child.

Generally, if a family came under the catchment area of the service, they were likely to receive care from the service from a very early age until the child transitioned into adult services at the age of 18 years. This would be a different model from a CDC, which would see children only at a very early age. Professionals therefore commented that they knew their families very well and all knew each other as professionals. There was a sense of relationship continuity and care provided by this service model. However, inequity in care provision was identified for referrals originating from outside the region. These families could not access the community nursing team or the SLT because these services were the responsibility of the local providers. This affected a small group of families annually and the reasons for referrals out of patch tended to be parental choice and changes in local teams.

All appointments with families were underpinned by the use of a traffic light tool71 that aimed to facilitate communication in the consultation. Families indicated the issues that they would like to focus on by ticking a pre-formed list of issues, including feeding, or by noting any concerns in a free-text box. The tool was also designed to celebrate positive aspects of children’s development. The tool, developed with the input of disabled young people, parents and other experts, was widely available to all neurodisability services. Although PSS was not included, it could be adapted to include this, thus enabling parents to assert their right to PSS. There was also a podcast explaining the use of the tool available on YouTube (YouTube, LLC, San Bruno, CA, USA; www.youtube.com). 72

Speech and language therapists (SLTs) played a key role in assessing children. Referrals to the SLT team were triaged through a referral form in addition to letters that clearly stated that the referral was for an assessment around ‘eating, drinking or swallowing’. The SLT interviewed stated that the SLT team tended to see children only where there were specific concerns about choking or swallowing. Children with behavioural/sensory feeding issues (e.g. children with autism) would be referred to the autism team. SLTs would also see children from the neonatal ward if there were concerns about swallow safety and there was need for support around oral feeding.

One parent reflected on the support offered from various professionals in relation to feeding:

We were in touch with the dietitian and the consultant sees her every 6 months. If I need the community nurse, I’ve got them, you know, on my phone, I’d just ring up for advice . . . When we first introduced food it was a battle, and I used to get myself really, really upset about it, and the speech therapist used to come out and she would say, ‘Don’t make mealtimes a battle, put it out, if she wants it she’ll eat it, if she doesn’t, just take it away and try at the next sitting down’, which is what we did, and as I say it’s just gradually built up from that.

CS04, P1, II

Observations of a multidisciplinary nutrition meeting prior to the feeding clinic, and of the feeding clinic itself, evidenced multidisciplinary working. This involved a senior specialist dietitian and a paediatric consultant along with the parents. At one meeting, the dietitian offered to send the mother some information on playful eating and provided a few tips on how to introduce new food and textures into the child’s diet. She advised the parent that the child needed to explore different textures by touching and playing with the food, which was viewed to be as important as eating, and parents were advised not to worry about mess.

Integrating support

Psychosocial support

Although in theory there was access to psychological support through CAMHS (as at all case study sites), this was not routine practice and for many children they would be too young to access this service. There was no psychologist at the paediatric disability service because this was not commissioned by the CCG. This contrasts with the availability of support for children with other long-term conditions and disability (e.g. chronic fatigue and diabetes) – a familiar pattern across all case study sites. Families and caregivers had to approach their GP for parental mental health issues. The following extract illustrates, to some extent, the complexity of pathways into mental health services and how children with neurodisability, and their families, fall through the gaps:

So when a parent is in distress about issues relating to feeding, what referrals are available to you? Is there a psychologist available, or any other professional?

Not really, I mean that’s, there isn’t, I mean we have CAMHS, children and young peoples’ psychological service, but you really have to be accessing, you have to, well, I mean a lot of our children don’t even meet the criteria for accepting referrals. So, we can make referrals, but they often get rejected, and it’s normally just about the team trying to support that family. Health visitors can be, some of the health visitors can be really helpful in supporting the little ones with feeding, and sometimes community nurses again, because they’re going in a bit more regularly. They can be very good in supporting with feeding. And then, so we do have some children’s nurses who go out to those who’ve got very specific medical needs, and sometimes we use them as well with feeding. So, we just, there isn’t, well there is something that we can refer onto, but often the children don’t meet its criteria unfortunately, so there’s a gap there.

CS04, S11, IPI

Professionals had different views on the added value of having a psychologist in the team. Concerns about including a psychologist included cost, recruitment difficulties, level of expertise and how they would be integrated into the wider team. There was also a feeling that other members of the workforce had those skills:

So, now I don’t, so yes, it would be wonderful to have a psychologist in the team and to be involved with every family. Having worked in teams where psychologists have been there it’s been very valuable and very useful; however, psychologists are extremely expensive resources and, you know, we have general recruitment difficulties across mental health teams as well as paediatric teams in our area. I don’t think our families lose out because they haven’t got a psychologist in the team, I’m sure that, you know, value could be added for some cases particularly the ones where there’s that tricky conversation, but they are very few and far between, I would say only one every, maybe one in 4 or 5 years, so you couldn’t justify a psychologist based on that.

CS4, S2, IPI

However, views on the benefits of including a psychologist in the team were influenced by the difficulties they had experienced in resourcing other posts. For example, one consultant reflected on a failed attempt to persuade commissioners to provide additional support for CCNs. Although CCNs were valued highly by parents (and other staff), their role, it was suggested, was poorly understood and undervalued by commissioners. They also had concerns that, to pay for a psychologist, cuts would have to be made elsewhere because trusts were not looking for ‘new business’ and there was ‘no money to pay for more staff’; instead, the resource would have to be paid from efficiency savings such as ‘more joined-up working’, ‘effective use of the feeding clinic’ and ‘reducing the number of appointments with different professionals’.

It was highlighted that, as support is not always available in the community, particularly at weekends, families may need to go to accident and emergency (A&E):

I think we need a few more community nurses as well. So, if a child has a tube and they have a problem at the weekend they need to go into A&E, so looking at kind of the weekend support around tube feeding. I think sometimes some families can just go straight up, like phone ahead and go up to one of the paediatric wards but that depends on if the ward’s full or not, I would have thought, you know, it’s a trip to A&E at the weekend for families if the tube’s come out for example. And our community nurses, they’ve got less time now as well, so I think they’re fairly stretched.

CS04, S9, IPI

The pressure on the CCN team, and the possible under-resourcing of the team, was emphasised at CS03 and contrasts, perhaps, with CS02, where resource constraints were not foregrounded in staff narratives. The CCN team at CS02 was provided by a Health Board rather than the CCGs typical of the English system, although we cannot say if these perceived differences were due to differences of commissioning arrangements, caseload or factors relating to the locality of each case.

The need for PSS early in the feeding journey was identified by a nurse:

Yeah, ah-ha. I suppose it would be helpful mainly for parents who are thinking about going down the road for gastrostomy; it’s quite hard, thinking of coming to terms with, that your child is probably, is not going to eat. You know, so I think support that way. I mean at the moment we do have a child who has an NG tube in, and she doesn’t have anything orally, but parents don’t want to have a gastrostomy fitted, because they believe she’s going to eat in the future, and I think it’s just, you know, it’s going to take a lot of time for them to realise that their child is never, probably is going to eat, or they’re never going to eat an efficient diet and in the meantime, you know, the child has an NG tube in, and pulling it out regular and, you know, all the things that come with having an NG tube. So, I think if there was more support around that, then maybe that may, that may help.

CS4, S7, IPI

The importance of early intervention was stressed by another member of the team (CS04, S9, II) who felt that a drop-in feeding group for young children with swallowing difficulties being weaned on to solids, a model adopted in a London children’s hospital, would be helpful. However, barriers to establishing such a group at CS04 were the problem of travelling with children on public transport and a recognition that funding for transport for families had been cut.

Similarly, a consultant felt that, with more time, the service could proactively identify problems:

I suppose maybe having a bit more time to do a bit more with the families would be good, if we had, you know . . . we could do more support, possibly, if we had more time to dedicate to them. That might be useful, we could do more frequent reviews then and pick up on problems maybe early or possibly if there were some. Because, as I say, at the minute we rely sometimes on the families contacting us if they’re concerned.

CS04, S6, II

Peer parental support

Professionals relied on informal contact and support between parents rather than having formalised referrals. Moreover, it was felt that parent-to-parent contact was happening organically around the clinic. One consultant reflected on the growth of social media, which, they thought, had, perhaps, taken over the role of clinicians in facilitating parental networking:

When I first started working in community 20-odd years ago there was no social media whereas now they all know each other because of Facebook and Twitter and things like that and they meet at the outpatients appointment, they swap, they’ll friend each other and before you know it you’ve got a little support group going on social media. I think some families definitely do that because social media [has] taken over from us a little bit now.

CS04, S3, IPI

However, one parent (CS04, P1, II) identified the need for a support group to share experiences and to discuss coping strategies with other parents, which challenged staff assumptions that peer support had developed organically. A staff member (CS04, S6, IPI) mentioned that in MDT meetings there was often discussion about ways of stimulating more contact between parents and families, and they thought that nurses could do more to suggest this. Another member of staff noted the benefits of peer parental contact:

As a team we’ll maybe identify that our parent might benefit from speaking to another parent about, if they were going to try a blended diet for example or around a PEG but I think that’s done fairly carefully in that the community nurse might identify a family and ask if they would be OK, you would be checking permission with both sides before introducing. More commonly, it’s actually parents just get in touch with other parents through various networks, be it social media, Facebook groups or Portage [www.portage.org.uk/about/what-portage; accessed August 2020].

CS04, S9, IPI

Transitions into adult services were identified as a potential area for improvement, although this was contested by another member of staff who felt that there was joint working and a slow transfer to help facilitate the move:

A disability liaison nurse and social worker look at the transition care at 16 years old; at 18 years old they leave the special school. They also leave the paediatric neuro team and transition the adult team, so the paediatric team do home visits up to the age of 18.

CS04, S3, II

Are there particular issues for older children and transitions in terms of referral pathways and support?

There is room for improvement in referral pathways into adulthood. District community nurses don’t take them on after they transition into adult care, so there is less in the adult arena. Homeward take over and disability nurses could get involved and [their] patients tend to go into hospital for any issues that may arise.

CS04, S1, II

The need for parent information about GFT feeding was identified, suggesting that there may not be collective knowledge about the resources available developed by commercial companies, or concerns about their use.

Decision-making

Feeding problems were monitored and, when it became clear that an alternative form of feeding was going to be needed, the community nurses played a role in supporting families. There were monthly meetings with the MDT and community nurses. As at other sites, staff reflected on the emotional impact of the suggestion of a GFT, which parents viewed as artificial, and the impact on family mealtimes, and feelings of guilt at not being able to feed a child:

They may resent or resist suggestions of what are often seen as artificial methods of feeding and I think a lot of what we try to do is keep that territory as neutral as we can from as an earlier stage as possible so that families don’t feel they’re failing by having to resort to those things but those are things that are there to try and help them make the next step in terms of supporting their child’s development and feeding, where that’s possible.

CS04, S5, II

Most HCPs interviewed agreed that families and carers needed to be actively involved in decision-making about a GFT. There was a feeling that parents received a lot of information before the procedure, including about the use of toys with a button or PEG to see what a GFT looked like.

The decision-making process was also described as quite complex owing to the involvement of multiple family members (parents are often accompanied by other family members, such as grandparents) who may or may not be involved in children’s care. Therefore, HCPs had to understand how the family as a whole made a decision, as portrayed in the following quotation:

Generally the demographic of the local population is that people don’t really move away so they have quite complex family networks, and grandparents, other relatives, often come to appointments with the family, and decision-making’s interesting in that you obviously can provide all the information to families but sometimes, not always, a lot of families will want you to try and guide them as to the right decision and they don’t always like it if you just say these are the options, these are the pros and cons, what do you want to do?

CS04, S4, IPI

With the expectation that families should take a lead role in decision-making, the observation that some families tried to hand back responsibility for the decision to the consultant was considered undesirable as professionals wanted families to make an informed choice and become well versed in the pros and cons of gastrostomy and GFT feeding:

There’s this temptation for a lot of families to sort of hand over the decision to you to make as the professional, which isn’t really how you would normally do a consultation, you know, you’d really want the family to be paramount in terms of having information and then being able to make that decision.

CS04, S4, IPI

Decisional conflict

Disagreements between professionals regarding GFT placement were not reported by staff because of a culture of ‘open discussion’. Disagreements with families were usually managed by going at the families, own pace:

It’s usually, that’s usually where there’s a safety issue, where the swallow clearly isn’t safe on the video, fluoroscopy and so the family want to continue to feed the child. Those are the only situations where we’ve run into difficulties, but we’ve managed to work those through with families and it’s not been a matter of interprofessional disagreement, it’s more been a matter of being able to help the family to see what the safety issues are.

CS04, S2, II

Although the tertiary centre had a role in influencing parental decisions by offering a surgical opinion, it was generally felt that the bulk of the work of supporting families came from the local referring team. In exceptional cases, children with very complex needs requiring additional support could be referred to the feeding/neurodisability clinic at the tertiary centre.

Blended diets

Another potential area for conflict surrounded the use of BDs. As at other sites, staff at CS04 highlighted the tensions between a lack of official support for BDs and increasing use by families: ‘We have certainly got a few on the caseload, yes, and managed without any national guidelines for it, and it’s not actively encouraged from a professional point of view’ (CS04, S3, IPI). This will be discussed in the final chapter.

Staff training

Staff were trained in their clinical areas and were competent to provide clinical care but training very much focused on these aspects:

So, they [the feeding clinic team members] have all had training, so all our nurses have postgraduate degrees, they’ve got their clinical skills course that they’ve completed, they’re prescribers as well. The speech and language therapists that are involved in children with feeding issues are dysphagia competent in line with their respective colleges and they’ve had relevant training in communication skills, et cetera. So, we’re very, we’re lucky that we’ve got a team that is, you know, been well trained and is practising regularly.

CS04, S2, II

Staff were also involved in training each other on the clinical and technical aspects of feeding (e.g. in the case of premature or other newborns, SLTs provide extra support to NICU staff to establish whether or not oral feeding is appropriate, and CCNs are also involved in training school staff in the management of gastrostomies). Specialist school nurses also reported in-house training on nutrition and feeding pumps/GFT management by gastrostomy specialist nurses (CS04, S7, IPI). SLTs reported training health visitors to enable them to provide more general feeding support and advice (e.g. weening process), to avoid unnecessary referrals and to enable the SLTs to better focus on children with an increased risk of poor growth (CS04, S9, IPI), but there was little formal training in the psychosocial aspects of feeding and support, although generic CPD courses may have relevance and transferable learning. Nurses may have had some generic training in mental health, but some felt that additional training in counselling skills could be useful (CS04, S1, II).

Summary

Case study site 4 provided an example of a three-tiered feeding pathway housed under one roof where the composition of teams was flexible. Input of team members was dictated by children’s needs and, therefore, similar to teamworking at CS03B. Care was co-ordinated by the lead professional. However, there was no written care pathway. The general view was that additional support from a psychologist may be beneficial but not for all parents and that a range of staff, with the right training, could provide PSS. Cost considerations were considered a barrier. Staff felt that they would like to offer more proactive support in detecting feeding difficulties sooner and develop parental peer support further. The role of the CCN appeared key in supporting families, although there was a feeling that the team needed strengthening, as at CS03.

It was difficult to recruit parents into this study; hence, the views of families were not well represented. Therefore, we were not able to triangulate views expressed by staff with those expressed by parents, although observations were used. The issue of recruitment is discussed in Chapter 6.

Satisfaction with care

When asked about their satisfaction with the support received the last time they had a worry or anxiety about their child’s feeding, 16 reported being fairly or very satisfied, three reported being partially satisfied, one reported being not very satisfied and one reported being not at all satisfied. When asked about their overall satisfaction with the care related to their child’s GFT or feeding problems, eight reported being fairly or very satisfied, six reported being partly satisfied, four reported being not very satisfied and one reported being not at all satisfied.

Health-care professionals’ confidence and training in providing psychosocial support

A total of 31 out of 109 questionnaires were received from HCPs (response rate 28%), who were asked about how confident they were in providing PSS to families using a five-point Likert scale as well as about what training, if any, they had received to do this. Responses (including type of professional) are provided in Tables 5 and 6.

Few of the HCP respondents (7/31) had received specialist training for PSS. Among these, specialist training for PSS was usually part of medical or formal professional training. Only one nurse (the only one to report being very or fairly confident) had received training and this was provided by the NMCN CEN (online and DVD training). There was a tendency for those with more confidence in providing PSS to have received relevant training. These results support the interview data from phase 2, during which a lack of specific training was identified.

Willingness to pay

A total of 168 respondents started the survey. Among these, 148 consented to their data being used for the purpose of research. A total of 103 completed the rating of service A (usual care) and 96 completed the rating of service B (enhanced support involving some provision for access to a psychologist and a more formalised approach to peer parental support) (see Chapter 3). Respondents were asked how much they would value a package representing the current provision and one representing the enhanced provision. Because parents would not be asked to pay for this PSS, and we did not wish to imply that they would, we used the preferred term ‘value’. Table 7 illustrates the types of respondent – professional or parent – who completed the survey. The majority of respondents were HCPs (72%).

Table 8 provides information about respondents’ household composition. Report Supplementary Material 7 provides additional demographic detail including sex, age, education level, employment status, annual household income, country of residence and ethnicity.

Respondents were predominantly female (96%), aged between 25 and 54 years (85%), residing in England (80%), living with a partner (70%) and in paid employment (81%); 86% had a degree at either bachelor’s or master’s level and the majority were from the higher-income brackets. This reflects that the survey was completed mainly by HCPs.

Table 9 sets out the frequency of values, expressed in Great British pounds, attached to service A, which represents usual care for PSS, and service B, which represents the enhanced package.

Figure 8 represents the change scores in the values respondents assigned to services A and B and demonstrates that most people’s scores changed between 0 and 300 when valuing the two different services in favour of service B.

FIGURE 8.

Change scores assigned to services A and B.

Table 10 sets out the means, standard deviation (SD) and 95% confidence intervals (CIs) of values assigned to services A and B. Because a preliminary analysis indicated that the data were skewed, a non-parametric Wilcoxon signed-rank test was used to measure the difference in values between the two services. The median ranks for service B were significantly higher than those for service A, demonstrating a preference for enhanced support with involvement of a psychologist and parental peers, which was statistically significant (n = 96; p < 0.001).

A two-sample t-test (unequal variances assumed) was used to assess whether or not there was a significant difference in WTP between parents and HCPs. Mean WTP for service A (carers, £252.17; HCPs, £262.33; p = 0.8090) and WTP for service B (carers, £306.52; professionals, £395.89; p = 0.0519) showed no significant difference in WTP between them. There was also no significant difference in WTP by household income group for service A (p = 0.5550) and B (p = 0.6852) using a one-way analysis of variance.

Conclusion on use of resources for psychosocial care

The number of parents or carers responding and reporting discussion on psychosocial issues during routine appointments was small and, as a result, the evidence on service costs to the NHS, social services and educational services is not strong. This reflects the challenge of researching a relatively small population. Those who responded may have been motivated to respond if they had experienced more discussion around PSS and so may have had higher than the average contact. The average estimate of costs is £76.42 per child per year at 2017 prices,73 with the highest single estimate being £317 for one child for 1 year. These figures suggest that the implementation of PSS into regular appointments would possibly add a percentage point to overall costs. In a previous study,28 we reported the overall net costs of GFT placement compared with pre-gastrostomy costs to be £147.96 per week per child at 2002 prices. 28 This is equivalent to £10,500 per year at 2017 prices. 73

We were not able to estimate value in terms of quality of life (to estimate opportunity cost to services) but instead provided WTP estimates, which were intended to assess the societal value of the services. When the cost estimates were compared with the estimates of WTP, which were £259.00 (95% CI £227.73 to £292.05) for scenario A (n = 96) and £374.00 (95% CI £344.14 to £404.82) for scenario B, it becomes clear that these values far exceed the costs estimated from our sample. However, a limitation of these values is that they can be biased by the respondents’ ability to pay. This was analysed in the analysis of variance, which showed no significant difference in WTP by household income.

Psychosocial support during appointments was provided by a range of HCPs, including doctors, nurses, teachers, dietitians, OTs, SLTs and health visitors. Appointments took place in hospitals, at GP practices, at community health centres and at schools. The professionals involved received salaries from different budget sources, there being no central budget for this care. Costs of support were therefore spread between a number of budgets. Parental peer support was valued but not offered consistently. Apart from our previous study on costs of gastrostomy,28 there is very little information on resource use and none for PSS for this cohort of children and families. Future studies may be required.

Case study site 1

Case study site 1 was a specialist neurodisability service with dedicated feeding clinics and a large number of enabling features, including a high level of integration and a relatively high level of PSS achieved through integration with a co-located school, and task-sharing between health and educational staff. Care was co-ordinated by a lead paediatrician. In addition, there was a high level of parental involvement, including representation on feeding committees and parent-led use of social media (e.g. Facebook). This level of integration had been achieved through strong leadership, policy underpinning the partnership and the funding model, which included pooled budgets (recently implemented). In addition, a joint surgical clinic held in the community health services, involving a surgeon from the tertiary centre at which gastrostomies are placed, served to integrate care in line with research recommendations. 29 This example of shared care contrasted with models in which tertiary service involvement was largely limited to a consultation with families for the placement of a GFT (e.g. CS04).

Although parental access to psychologists for support was not usual practice, members of the MDT had access to psychologists and psychiatrists for additional support as required. Although there was no written care pathway, the right to emotional support for children more generally was leveraged through the shared clinical services and school’s children’s charter, informed by a rights-based discourse and developed with children and families. Competency in providing PSS was strengthened through shared learning among team members. Although the model was highly specialised, and not necessarily replicable, it showcased elements of best practice that could be explored for adoption in other services subject to the availability of funding.

Case study site 2

Case study site 2 is an example of a generalist service for children with exceptional health-care needs tailored to a rural context and staffed by a MDT. The provision of PSS was enabled through a best practice statement that specifically mentioned the need to provide emotional support based on research evidence. Resources to support children and families could be leveraged through MACs. The model offered a different approach for achieving high levels of support for a small number of children, with assistance from a national managed network (NMCN CEN). The network disseminated best practice, including a DVD for families considering a GFT, produced with parental involvement. There was an educational module on the provision of PSS available through e-learning and developed in conjunction with a local university and the NMCN CEN. However, this training was not mandatory and was available only to individual members of the NMCN CEN, raising access and implementation issues.

Despite the challenges of care provision in rural areas, care appeared to be person- and relationship-centred and, hence, PSS was achieved in a relatively informal way. Relationship continuity across the life course was particularly strong with the CCN team. Problems following surgery were likely to be identified by the CCNs or in the 6-monthly reviews of children in the therapist/nurse-led, home enteral feeding clinic. This was a strong feature of the model given that research suggests that dedicated enteral feeding teams can reduce hospital re-admissions for GFT-related complications from 23% to 2% post surgery. 78 The enteral feeding clinic also specialised in supporting oral feeding following GFT insertion, which parents, more generally, valued.

Case study site 3

Case study site 3 was a specialist neurodisability service and our case highlighted uneven provision both within and between services, with risk of gaps in community settings. Whereas CS03A had a feeding clinic, CS03B did not. Teams were co-ordinated by a named paediatrician; however, the model of teamworking at CS03B was described as serial, rather than multidisciplinary, with some members of the MDT employed by different trusts, attesting to the fragmentation of health services delivery. Staff saw families individually and the team communicated by telephone, e-mail or face to face at MDT liaison meetings. It was unclear if or how this affected families. Although there is no research to suggest which models of care work best in terms of child health outcomes, parents may have preferred the ‘one-stop shop’ approach afforded by the feeding clinic model. National Institute for Health and Care Excellence (NICE) guidance on the care of children with cerebral palsy (not feeding specific) recommends that children should be cared for by a core multidisciplinary team. 79 The model of service delivery at CS03B appeared to rely more heavily on the lead paediatrician to co-ordinate and integrate care involving professionals across different trusts. There was a written care pathway but PSS was not explicitly stated. Of interest was the availability of the services of a psychologist at CS03A, although only particular groups of children were able to access the services through the CDC, based on the criterion of age, reflecting inequity in access within and across services. No similar resource existed at CS03B. Moreover, additional restrictions to accessing the services of the psychologist at CS03A were implemented during our study.

A recently implemented surgical nurse assessment model at the tertiary centre at which gastrostomies were placed provided an alternative model of integrating the community and tertiary services and supported families to make a decision about the need for a GFT. The surgical nursing team based at the tertiary centre at CS03 conducted an initial assessment of children’s need for a GFT and provided support to families before and after surgery, a model, as at CS01, that could be considered an example of best practice given that our provider survey (phase 1) identified gaps in communication between community and tertiary centres. Follow-up post surgery, at specified time points, allowed any problems arising from the placement of a GFT to be identified and actioned. The tertiary centre’s use of social media (e.g. Facebook) provided an opportunity for the provision of information to families.

Case study site 4

Case study site 4 was a district, hospital-based service where teams were co-located. The composition of teams varied according to need and, hence, there was a fluidity to how teams functioned. As at CS03B, the co-ordination of care was reliant on the lead professional. However, co-location is not a proxy for integration or a ‘unitary construct’ but a ‘complex set of relationships, organisational structures, and other features meant to help practices deliver care’. 75 The difficulty in capturing this complexity was reflected in the service map at CS04, which has a three-tier feeding pathway (although unwritten), a further indication of the high level of specialisation. This level of specialisation was reflected in the fact that, until recently, gastrostomies were also placed within the service. This function then transferred to the tertiary centre, which had minimal involvement in supporting the majority of children referred, which contrasted with the model of care at CS03. There were strong links with the CCN team, which was viewed as the ‘eyes of the service in the community’, although reported to be under pressure. An important aspect of care was the traffic light tool (a communication aid developed with the input of parents) widely adopted by clinicians, which aimed to empower parents in clinical consultations. The tool could be adapted to help guide conversations around PSS issues related to children’s feeding and GFT placement.

Processes and mechanisms of care

Processes of care include the interactions of staff with parents as opposed to the clinical tasks that are performed. We identified three analytical constructs governing processes of care: hidden work, expressing emotional vulnerability and negotiations around risk.

Outcomes

Although this was not a systematic study of outcomes following gastrostomy, parents generally viewed gastrostomy favourably, as documented in the research literature,95 although many recalled problems in the immediate aftermath of surgery and being faced with having to learn new feeding practices and nursing skills. Side effects of the procedure resulted in families having to seek health care, involving travel and sometimes admission to hospital, or required parents to develop expertise in managing the nursing care of their children themselves, particularly as support from services waned over time. Research has documented the benefits of gastrostomy feeding, including reductions in psychological distress over time (e.g. at 12 and 18 months post surgery). 96,97 However, problems that children and families experience when using home enteral nutrition overnight include sleep disturbance, dislodgement and blocking of GFTs, and problems with the pumps delivering feeds are also reported. 80 Models of care that support families in the immediate aftermath of surgery, such as the CCN team and the home enteral clinic at CS02, and the tertiary nursing assessment model at CS03, have the potential to improve care post surgery by minimising the side effects of treatment and preventing readmissions to hospital. 78,98 Models of care that better prepare families for surgery, as at CS03, where children were assessed by nurses at the tertiary centre where the GFT was placed prior to appointments with surgeons, may also provide important spaces for families to discuss issues of concern. In this example, the surgical nurses additionally provided an important link between families and the CCNs, who also provided support, although there was a perception that these services were under-resourced. Schools also play an important role, and there was evidence of significant task-sharing between health-care and educational staff at CS01, a service characterised by high levels of integration between health-care and education. At other case study sites (e.g. CS02), school staff would not necessarily engage in nursing activities, which would be the role of the CCN team.

Research on health outcomes for paediatric neurodisability has demonstrated that parents value emotional well-being as an aspect of health. 99 In our study, little was recorded in the children’s notes that could be utilised to evaluate this aspect. Recording of satisfaction with the procedure, or the PSS provided, was generally absent, as were any quality-of-life measures for children and families. Data on the impact of parental peer support, given that this was an informal arrangement, were not captured, nor was the effect of training on team performance. A systematic review of core outcomes reported that ‘life impact and resource use’ were under-represented in research studies. 100 The authors of the review suggest that these outcomes are either undervalued or not measured owing to a lack of standardised tools. This also presented difficulties with phase 3 of this research on costing PSS. A recent study analysing children’s medical records in Sweden101 demonstrated that clinicians emphasised biological and nutritional outcomes post gastrostomy, whereas information on psychosocial aspects was only sparsely documented. The authors concluded that information relating to medical intervention goals dominated and was prioritised over psychological aspects, despite the involvement of psychologists and social workers in setting these goals – findings that were supported in this study.

The lack of documented outcomes is not surprising given the hidden nature of PSS work. Professionals reported that they spent considerable time supporting families. If such investment of time is to be valued then this work needs to be rendered visible through appropriate documentation and evaluation to show the potential to improve the quality and experience of decisions about children’s feeding, parental enablement and the PSS provided, including the overall impact across the life course. It may also help to obtain resources.

Equally, there were no mechanisms in place to record or evaluate the impact of specialist training on the team. If training was focused specifically on assessing and meeting the PSS needs of parents, or staff with specialist skills were involved in supporting parents or teams, this could prove an effective use of resources. This is important given that austerity has led not only to services being cut but also to professionals having less time with children. 102 Team training, which strengthens the ‘collective competence’ of the team, could ensure that PSS is consistently embedded103,104 rather than left to individual interest, and could also draw on the expertise of parents as trainers. 105

Integrating psychosocial support

In interviews, HCPs report spending significant time supporting children and families, but because PSS is integrated into appointments it is undocumented and therefore not recognised or valued, and provision may therefore be inconsistent.

The challenge for services is how to better integrate PSS into clinical ways of working by creating spaces dedicated to PSS initiatives that can also be documented, audited and evaluated. The different mechanisms for integrating care identified in this study could serve as foci for strengthening PSS initiatives in these enabling structures.

Because there were differences in parental need for PSS, a personalised approach using appropriate assessments by a named staff member, with training and with continuity of the professional involved, could also ensure effective use of resources. Given that parents and children may not be able to assert their need for PSS in time-limited appointments dominated by clinical aspects, appointments where the primary focus is PSS or longer appointments with dedicated time may enable parents to discuss their concerns. Flexibility will be important because parents’ need for PSS may not coincide with appointments and parents may find it difficult to talk about their feelings in the presence of their children. PSS could be formalised as part of standard care, which would help to normalise emotional vulnerability, and standards of support could be developed and shared with children and families (see Report Supplementary Material 8). Because professionals had different ideas about parents’ PSS needs, ranging from a discussion about the risks involved in placing a GFT to an understanding of the emotional impact of the decision (e.g. a grief journey), we recommend the term ‘psychosocial emotional support’ to ensure that appointments focus on values and emotions in addition to clinical risks and tasks. Service providers could offer home visits, which have been shown to be valued, although this would incur costs for travel and additional time.

Parents also expressed a preference for receiving support from someone they knew, which suggests the importance of relationship continuity, which may be an important factor when considering how to integrate support from a designated member of staff (e.g. psychologist or equivalent), where available. School staff also have an important role to play in wrapping support around the child and family and could be more proactively involved in supporting families through team training. Lead professionals have a role in co-ordinating this support, which also needs to be recognised.

Where access to a psychologist or equivalent professional with specialist training is available, it would be helpful to parents and staff to know how to obtain this access, should they choose, and what to expect through clear pathways. Careful attention to the use of language (terminology), as well as how the issue of PSS and the opportunity to see a specialist is raised, will be needed to avoid the suggestion that parents cannot cope and, hence, reinforce the sense of failure that many parents may already feel.

The provision of child- and family-friendly information using social stories106 could also be better integrated into care as a standard and entitlement to ensure equity of access. There was evidence of a DVD for parents at CS02, which could be shared, and online resources made available through professional and parental networks.

Mechanisms for recording and auditing PSS may help to make the case for additional resources and could provide useful costings information. National guidance on how to code and capture data will be necessary107 to minimise discrepancies in coding to compare service data. Coded and structured data could then be exploited for secondary purposes. 108

Methodological limitations

We conducted a qualitative case study with a focus on four service configurations chosen, in part, from the findings of a WBS that produced a low yield of responses. Therefore, we cannot claim that these services are representative of all services. However, we chose sites based on what they could tell us about how PSS was provided and integrated into children’s care and the factors that enabled services to provide the support they offered based on the findings of the WBS and additional theoretical criteria. A study on the organisation of midwifery units51 similarly chose four sites, and generalisation in that study was seen to work on a ‘conceptual and theoretical level’. We believe that our analytical constructs (hidden work, expressing emotional vulnerability and negotiations around risk) are theoretically generalisable and reflect processes of care for families regardless of the different characteristics of the services we chose to study. We also recognise that the mechanisms for the integration of care that we have presented may not be exhaustive, and that our findings are provisional, given that services are often in a state of change. Indeed, we witnessed changes in the organisation of care at two sites over the course of our study – a finding reported elsewhere. 113 It is of interest that a survey of 100 parents conducted in New South Wales, Australia, reported overlapping concerns with those reported by some families in this study: families expressed dissatisfaction with their care and the support they received both before (42%) and after (36%) surgery – themes that resonate with some parents’ experiences in our study (although not all), albeit in a different context. 114

It proved extremely difficult to disseminate a national survey (phase 1) outside a professional database of leads of child development teams. Dissemination through the R&D networks of different trusts required a considerable amount of administrative work given that each R&D department must assess their capacity and capability to participate in research studies. We had little control over the dissemination of the questionnaire. The survey was disseminated during a transition period for the HRA in England and Wales, which led to considerable delays and inconsistency in the process of obtaining permissions. This undoubtedly led to a poorer response rate than anticipated, and we have discussed further methodological limitations of surveys as tools to capture organisational practice in the context of complex service configurations in Chapter 1. There were also differences between responses reported in the WBS and actual practice. For example, 15 out of 35 respondents stated that PSS was formalised, and 19 out of 23 respondents stated that referrals to other parents were formalised, yet our field work (phase 2) suggested this was not the case. These differences may be due, in part, to sampling or an artefact of the methods used. We were better able to probe and tease out these differences in interviews.

Interviews can also have their limitations when used to identify everyday practices that remain hidden. 39 Ethnographic observations of care, and reflective interviews with teams and parents, following clinical consultations may prove a more effective approach to understanding the delivery of PSS in real time. This may prove challenging to organise where facilities and time are limited.

The system of permissions

The permissions process for the case study research was also administratively onerous given the fragmentation of services (community services coming under one trust and tertiary services another) and where members of the MDT were employed by different trusts. Each case required at least two sets of permissions. In one case an additional set of permissions would have been required to interview one member of the MDT employed by a different trust. Because this was not an efficient use of research time, some members of the MDT were not interviewed. Assessments of capacity and capability often took several months, particularly in the second half of the study, owing to concerns about the General Data Protection Regulation (GDPR). 115 One R&D department made an application to the Caldicott Guardian because our research involved children, demonstrating an additional level of governance not required elsewhere. Therefore, the model of research governance proved challenging for a project requiring a more iterative and flexible approach – an important characteristic of progressive focusing. Regional and cross-sector approvals would to help minimise the administrative burden for studies of this type and complexity.

Parent and child participation

It proved difficult in some cases to engage parents and children in the research. The research team did not have direct access to parents and relied on research nurses to disseminate information about the project. Before sending information by post, the services contacted parents by telephone to seek their permission to participate and send out the information. However, the research team, concerned that this might affect response rates, felt that the first contact messaging should be nuanced in a way that encouraged participation and consent on receipt of the information. We also asked services to advertise the project through their social media and via posters in clinics (which needed additional approvals). In addition, our lay chairperson contacted local parent groups informally, where these existed, to inform them about our study. It is also possible that families were dealing with more pressing issues that took priority in the face of cuts to local services,102 and some of our case study sites were drawn from areas characterised by higher than average levels of deprivation, which may have affected participation rates. One service cautioned that, historically, parents did not participate in qualitative studies, suggesting a culture that is biomedical research dominated. Poor parental recruitment in palliative care research as a result of professional gatekeeping has been reported. 116

It can also take time to access children because of the need to work with gatekeepers, including parents, who provide consent to interview children. 117,118 An audit of consent forms across two case study sites revealed that just over half of parents did not consent to their child participating in research. Most parents when asked to consent left the box blank, or actively refused, or put ‘not applicable’. Reasons for refusal included that the child was ‘non-verbal’ and that the child had global developmental delay and could not speak. One parent agreed providing they were present to assist with communication. There are particular challenges to interviewing children in this population (approximately 42–60% are estimated to have communication impairments)10,60 due to the different modalities that they use to communicate and how these interactions are interpreted. 119 For this reason we planned to video-record interviews. However, one service would not allow us to video the interviews of children on their premises, although the research had received ethics approval. Moreover, the service was not able to provide rooms for interviews owing to space limitations. For those parents who did provide consent, it proved logistically difficult to arrange interviews owing to children’s schooling and the need for permission to video-record on school premises, as well as illness that prevented participation. Because some parents lived a considerable distance away from health centres, with little public transport, travel to families’ homes made it difficult for researchers conducting field work at a distance and Skype interviews would not be suitable.

It also took time to establish a relationship of trust with children and learn their preferred way of communicating, which would have required repeated visits, with the potential to affect children’s schooling and family life. Gaining children’s assent to participate could take up to 30 minutes, and in the case of one child 20 minutes was their maximum attention span. One interview had to be conducted through the child’s teaching assistant, but researchers have questioned the validity of children’s views expressed through intermediaries. 120 Therefore, we have not presented data from children in this report in any substantive way. However, our PPI work involving children provides a particularly powerful example of children’s views on the right to support.

Timescapes and experiences

Parents’ accounts of their experiences were often historical and reflected diverse timescapes. Hence, issues took on different significance depending on where parents were in their child’s feeding journey. For example, in some cases decisions about gastrostomy were made, and the surgery undertaken, many years ago, and the children were now in their teens, whereas other experiences were much more recent. In some instances, decisions had yet to be made. Thus, parents’ narratives were located in the past and present. However, the accounts of staff almost invariably described their experiences as a provider of care in a service at the case study site under investigation and in relation to current practice, which often served to support the views expressed by families. Moreover, parents of older children provided data on the experiences of GFT feeding across the life course, not previously reported, which adds to our understanding of support issues beyond the decision-making phase.

Moreover, owing to the complexity of children’s needs and the severity of their neurodisability, children often received care from multiple health providers in different locations. Parents sometimes discussed their experiences in relation to services within and outside the boundaries of the case study sites. Parental narratives therefore traversed these complex timescapes and landscapes of care.

Resource use, costings and willingness to pay

Response rates for the costings questionnaire from both parents (20%) and professionals (28%), although comparable to that reported in a study costing key worker schemes (30%),121 warrant further discussion. An audit of consent forms at two case study sites revealed that just under one-quarter of parents did not consent to participate in one, or both, of the phase 3 studies. It is of interest that 30% of staff did not consent to participate either, although at this stage the costings questionnaire had not been included in the information packs. We later changed the stage at which the questionnaire was disseminated so that it was included with the consent forms for fear of sample attrition. For example, owing to a change of sponsor, the project was paused for 1 year at the time that the questionnaires were sent to CS01 for dissemination; however, owing to indemnity issues, they were not sent out until after the pause. This inevitably led to a loss of momentum and continuity. There was much discussion in the PAG about whether or not sending the questionnaires with the consent forms contributed to information overload. We asked the local research nurses to advise parents that they did not need to complete the costings questionnaire at the same time as the consent forms and that they could receive assistance to complete the forms from the research team to maximise consent.

Higher response rates (e.g. 71%) were reported in a study on the cost-effectiveness of gastrostomy,28 but that study took place at a tertiary centre with a higher volume of children than the community services in our study. Parents were recruited directly in clinics by an onsite researcher who had formed a relationship with the families because the research design, involving multiple data collection points, necessitated frequent contact with the families, before and after the placement of a GFT. The researcher was therefore known to the families. The researcher was able to assist families with the completion of forms at the hospital or by telephone. Gatekeeping was also minimised. However, in our study researchers were very much ‘outsiders’ to the services and not allowed to approach families directly. Future costing studies may therefore need to be conducted at the regional or tertiary centres to capitalise on the volume of patients, although much of the support will have taken place in the community, sometimes over many months or years.

Low response rates are not surprising given that we were looking for a particular cohort of children. Moreover, parents may be wary of participating in research if they feel that this could result in the withdrawal of services that are already perceived to be under threat. 87,102 Indeed, this was confirmed by a professional at one case study in conversation with a parent. Professionals may have similar misgivings about costings research in a climate of cuts and limited resources, particularly where services may be under review, as was the case at one of our sites. One clinician, for example, did not agree to the WTP aspect of the study and refused to advertise the study to parents because it might be misconstrued as requiring payment for health care.

Our estimates of cost were based on the responses of eight parents, five of whom reported that they had received PSS in the previous 12-month period; therefore, the results have to be interpreted with caution, as should the estimates of time provided, given that self-reporting of time spent in activities may be over- or underestimated. 13 Other parents either indicated that they had not attended appointments in the previous 12-month period or left this section blank. There was a perception in our case study that parents had to learn to manage what could arguably be described as nursing tasks post gastrostomy. They may have resolved their issues themselves or through other means, for example by telephone, and not received appointments per se. Again, this reflects the problem of researching small populations with health disparities122 where data are lacking or simply not recorded because PSS is not recognised or valued as a distinct activity by services and commissioners. However, this is an important finding.

Notwithstanding the limitations, a key strength of this study lay in the use of a mixed-methods approach, gathering data from a range of perspectives where possible. This meant that professional reports of if and how PSS was provided, for example, could be balanced by the views and reported experiences of parents and carers, and a range of professional experiences were captured. In addition, the use of observation, although only limited data are reported directly here, provided a third ‘observer’ perspective that added to our capacity to triangulate the findings. The case study has also showcased several initiatives in providing PSS that will help services to further develop and embed resources. The case study has also highlighted gaps in service delivery and the training needs of staff.