Parent-delivered interventions used at home to improve eating, drinking and swallowing in children with neurodisability: the FEEDS mixed-methods study

Article history

The research reported in this issue of the journal was funded by the HTA programme as project number 15/156/02. The contractual start date was in July 2017. The draft report began editorial review in November 2019 and was accepted for publication in March 2020. The authors have been wholly responsible for all data collection, analysis and interpretation, and for writing up their work. The HTA editors and publisher have tried to ensure the accuracy of the authors’ report and would like to thank the reviewers for their constructive comments on the draft document. However, they do not accept liability for damages or losses arising from material published in this report.

Copyright statement

© Queen’s Printer and Controller of HMSO 2021. This work was produced by Parr et al. under the terms of a commissioning contract issued by the Secretary of State for Health and Social Care. This issue may be freely reproduced for the purposes of private research and study and extracts (or indeed, the full report) may be included in professional journals provided that suitable acknowledgement is made and the reproduction is not associated with any form of advertising. Applications for commercial reproduction should be addressed to: NIHR Journals Library, National Institute for Health Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK.

2021 Queen’s Printer and Controller of HMSO

Context

Long-term conditions affecting the brain, nerves and muscles are often grouped under the term ‘neurodisability’. ‘Neurodisability describes a group of congenital or acquired long-term conditions that are attributed to impairment of the brain and/or neuromuscular system and create functional limitations. A specific diagnosis may not be identified. Conditions may vary over time, occur alone or in combination, and include a broad range of severity and complexity. The impact may include difficulties with movement, cognition, hearing and vision, communication, emotion and behaviour’. (Reproduced with permission from Morris et al. ). 1

Eating, drinking and swallowing difficulties (EDSD) are common in young children with neurodisability. EDSD may lead to inadequate calorie intake, which affects a child’s nutrition, growth and general physical health. 2 There are three broad causes of EDSD: physical causes, which may affect control of the muscles of the lips, tongue, mouth and throat, and/or control of posture and movement, and/or impair the efficiency and safety of sucking, chewing and swallowing (e.g. children with cerebral palsy); non-physical causes, including sensory sensitivity that may lead to aversion, potential refusal of certain foods and rigidity or rituals associated with food or mealtimes [e.g. children with autism spectrum disorder (ASD), some of whom have avoidant/restrictive food intake disorder (ARFID)3]; and mixed, owing to both physical and non-physical causes. These three types of EDSD are used throughout the report to refer to these groups for ease of reference. Eating and drinking ability in children with EDSD of any cause is affected by their cognitive ability and their developmental age equivalent (rather than chronological age). Physical and non-physical EDSD frequently co-exist (e.g. children with cerebral palsy or Down syndrome). Both types of difficulties make mealtimes stressful for children and their families, and have negative impacts on quality of life and social participation.

The interventions that are available to address physical and non-physical EDSD are different;4–6 however, in practice, many children require judicious deployment of multiple interventions. 7 Descriptions of children with different types of EDSD and the interventions that they might receive are shown in Box 1.

Parents and primary carers of children with neurodisability and EDSD (referred to as parents for the remainder of the report) are usually supported in the NHS by multidisciplinary teams (MDTs) of health professionals (HPs);8 for example, paediatricians, speech and language therapists, occupational therapists, dietitians and, less frequently, clinical psychologists. HPs identify the cause(s) of a child’s EDSD by a combination of review of the child’s previous and current EDSD, understanding the origins of their condition, clinical observation and instrumental evaluation (e.g. videofluoroscopy). In the NHS, services to assess and support children with physical or mixed EDSD are more common than those to support children with non-physical EDSD.

Professionals take account of a range of parent and child factors, including parents’ views and their capacity to understand and implement interventions, and of the child’s cognitive ability. Individualised advice is then given on how and what to feed their child to improve the safety and efficiency of eating and drinking, improve the volume of oral solids and liquids if children can eat and drink safely, and how to manage behaviour so mealtimes are a positive experience. 7 This advice takes the form of individual interventions that are often delivered alongside each other; the actual intervention content received within each ‘intervention type’ varies with the child’s needs and neurodevelopmental profile. For children with either physical or non-physical EDSD, it has been unclear what advice is usually given, which interventions are commonly used, what constitutes ‘best clinical practice’ and whether or not there is robust evidence for such practice. 4–6 For instance, the National Institute for Health and Care Excellence (NICE) guidance for the treatment of both children with cerebral palsy and children with ASD recommends assessment and intervention as considered appropriate by clinicians, but does not specify how children should be assessed or which interventions should be provided (Box 2). 6,9

The interventions that HPs may advise families to adopt can be time-consuming to administer, may involve considerable changes to parents’ usual feeding plans and may sometimes be in contrast to parents’ beliefs about how their child should be fed. 7 To date, it has not been known which interventions are viewed as acceptable and feasible to be delivered by parents at home, and how intervention success is or should be measured by parents and/or HPs.

Why this research is needed?

Most children with neurodisability and EDSD are identified by the age of 6 years, and many from infancy. The need for evidence about the timing, duration, dosage and effectiveness of individual EDSD therapies was ranked as the number-one priority by parents of children with neurodisability and professionals in a James Lind Alliance research priority-setting exercise. 11 HPs believe that effective interventions should benefit children’s health, growth, nutrition, development and learning; to some extent these are inter-related, as adequate nutrition is required for brain growth and for optimal development. 7 In addition to improving a child’s physical health, interventions targeting EDSD may also have positive psychosocial and education outcomes; mealtimes may be shorter and/or more enjoyable to children and their families, which increases the social participation and quality of life of both the child and their family at home, and for the child at school. However, interventions may have unintended adverse outcomes, such as increased parental or family stress, if the interventions conflict with parents’ beliefs and wishes about how to feed their child. 7

Thus, studies are needed to establish the effectiveness of intervention(s) that parents can deliver at home. However, before such studies can be undertaken, information is needed on which groups of children to include; the range of interventions available; what parents and HPs think are the most relevant outcomes (e.g. medical outcomes, such as nutrition, weight and health, and outcomes related to the International Classification of Functioning,12 such as social participation); what outcome measurement tools are valid, reliable, responsive and acceptable; and what types of study design would be acceptable to children, parents and HPs, and feasible to deliver. In this context, the National Institute for Health Research (NIHR) commissioned research in 2016 to address the following research question and objectives.

Research aims

The overall purpose of this study was to answer the question: what interventions, which could be delivered at home by parents, are available to improve eating in young children with neurodisability and are suitable for investigation in pragmatic trials? The aims were to:

1. review the clinical practice and research evidence for interventions, outcomes assessed and the tools used to measure these outcomes

2. determine which parent-delivered interventions are currently recommended by NHS professionals, which interventions parents use at home and how parents and professionals evaluate whether or not an intervention is successful

3. construct one or more trial frameworks acceptable to children, young people, parents and professionals; or to specify the additional evidence about interventions, outcomes and tools that would be needed to support a future trial.

Scope of the study

The NIHR commissioning brief requested a focus on ‘young children with neurodisability’. Following Morris et al. ,1 we defined neurodisability as any condition that is attributed to impairment of the brain and/or the neuromuscular system and creates functional limitations. We included any non-progressive neurodisability condition that gives rise to physical, non-physical or mixed EDSD. Throughout the research, we gathered information relevant to children with physical and non-physical EDSD separately as we considered that interventions to address these specific types of difficulties may differ. However, as many children with primarily physical EDSD also have non-physical EDSD, we present information relating to children with physical and mixed EDSD together throughout this report.

As there was no prior definition of ‘young children’ specifically relating to EDSD and neurodisability, in our study we defined the age range covered by ‘young children’ during the project. The co-investigators agreed to start the project with a conservative working definition of ‘children as aged up to and including 8 years’. Participants at a consultation group prior to the start of the research agreed that this was an acceptable initial working definition. Broader age ranges were used in some elements of the research to ensure that we captured all of the evidence relating to children aged 0–8 years and enabled all parents and professionals with recent experience to contribute their views. Individual sections of this report state the age range of children with EDSD included in specific elements of the study, and describe why any extensions that were applied were deemed necessary.

The commissioning brief focused on interventions used by parents at home. However, children aged up to 8 years are also fed in education and other care settings using techniques similar to those used at home. Therefore, we included research that focused on children’s eating and drinking at home or at school or nursery. Studies of feeding interventions that were delivered solely on paediatric or neonatal units in a hospital were not included. Enteral feeding, which involves direct feeding by a tube into the stomach, was not included in this study because this intervention is used when oral feeding is not enabling a child to receive sufficient fluid or calories.

Design

To address the study’s aims we used an iterative mixed-methods design. The individual methods comprised systematic reviews (updating three published reviews of interventions and undertaking one review to assess measurement properties of published outcome measures), a mapping review, surveys, focus groups, a Delphi survey and stakeholder consultation workshops (Figure 1).

FIGURE 1.

Methods used to address each aim of the study. TAU, treatment as usual.

Delivery of the research

The research team was multidisciplinary and comprised clinical academics, clinicians, health services research methodologists and parents. The team members were from the north-east, south-east and south-west of England. The parent co-investigators (DG and JS) had experience of mixed and non-physical EDSD, were part of UK and international networks of parents of children with neurodisability and had previous experience of working in applied health research. The clinicians and clinical academics provided services to families of children with neurodisability who had EDSD: clinical psychology (HM and HT), community paediatrics (AC), gastroenterology (JT), neurodisability paediatrics (JP, JC and MA) and speech and language therapy (CB, LP and DS). The health services research methodologists had particular interests in childhood disability research (CM), clinical trials and health-related surveys (EM and CM) and evidence synthesis (DC).

The full research team met regularly to monitor the conduct and progress of the study and to consider the findings from each research activity. We discussed decisions on whether or not any interventions, outcomes or measurement tools lacked evidence of use in clinical practice in the UK or supporting research and, therefore, should not be taken forward. In the final months of the study, the research team met three times to discuss the main study findings and to formulate the recommendations for the design of future evaluation studies of interventions.

The research was directly informed by a parent advisory group (PAG) (see Patient and public involvement) that consisted of parents of children with neurodisability and physical, mixed and non-physical EDSD. An external study steering group that included members with clinical and research expertise and links to parent groups provided oversight and advice on the conduct and reporting of the research.

Patient and public involvement

The PAG advised on the methods, procedures, analysis and dissemination of each element of the research design. We recruited parents from the north-east of England to the PAG via social media and local networks known to parent co-investigators Deborah Garland and Johanna Smith. Deborah Garland is a local National Autistic Society (London, UK) representative. Johanna Smith is a parent of a child with mixed EDSD and additional sensory impairment. Five parents expressed an interest in taking part and three or four parents took part in each meeting, with one of those parents contributing their views via e-mail. Equal representation was achieved from parents of children with physical and mixed EDSD (n = 2) and parents of children with non-physical EDSD (n = 2). Their children ranged in age from 6 to 16 years. Meetings took place on four occasions over 10 months in Newcastle. PAG members agreed the dates and venues in advance to ensure convenience and maximise attendance. Any documents that required attention in the meetings were circulated in advance via e-mail to allow PAG members sufficient time to read them. The sessions were led by a minimum of two facilitators (HT, DG and JS) with at least one of the parent co-investigators present. The sessions lasted 2 hours and refreshments were provided. Parents received a £75 shopping voucher to thank them for their contribution.

The first meeting outlined the purpose of the group and agreed how parents would like to receive communication. Members agreed that face-to-face meetings were preferable and that any additional work would be undertaken via e-mail as necessary. The members did not wish to join a closed Facebook group (Facebook, Inc., Menlo Park, CA, USA; www.facebook.com) or use other social media in relation to group tasks. The group members agreed terms of reference for the PAG (see Appendix 1). In the first meeting, the PAG also considered a summary of the findings of the national survey of interventions recommended to and used by parents, and the important outcomes. Members discussed whether or not the findings were consistent with their experiences of supporting their child with EDSD and of service provision.

In the second meeting, the PAG received a summary of all the evidence gathered to date, which included findings from the systematic and mapping reviews, the first round of focus groups and the national survey. The discussion focused on how best to share this information with parents in the second round of focus groups. The PAG also reviewed the wording and layout of the Delphi survey and the associated information sheets. The discussions covered the use of appropriate and accessible language in documents, the presentation of visual information (colours, layout and clarity) and how best to present the information simply, without repetition or unnecessary jargon. The PAG were also presented with three parent-reported tools for outcome measurement that had relatively stronger evidence for supporting robust measurement properties. They commented on their wording, layout and ease of use.

In the third meeting, the PAG reviewed the amended Delphi survey and information sheets that had been modified in response to the discussion in the second round of focus groups. Discussions again focused on keeping information clear and accessible. The PAG also discussed how to order the statements relating to the interventions and outcomes in the Delphi survey and suggested providing examples of each intervention for clarity.

In the fourth meeting, the PAG discussed the findings of the Delphi survey and provided advice on how to present this information to parents at the stakeholder consultation workshops. The PAG reviewed the individual tasks for parents attending the workshops in terms of content, structure and timings. They also advised on how best to present information about the study and key findings in a short presentation, including simplifying language and presenting the study data in a pictorial format. They also suggested creating a document to send to all attendees prior to the workshops to provide a background to the study and clarity on what would happen on the day. The PAG were also asked to give their views via e-mail on the proposed dissemination plan during the final months of the study.

In each chapter of the report, we describe the patient and public involvement (PPI) in the planning and conduct of the individual stages of the study and the interpretation of their findings, along with the strengths and limitations of the individual research methods.

Ethics

The West Midlands and the Black Country Research Ethics Committee approved the study procedures (17/WM/0439). There were four amendments: one non-substantial amendment (to add new sites as participant identification centres), two planned substantial amendments to seek approval for documents developed for the Delphi survey, informed by the findings from earlier study stages, and one further substantial amendment (to change the recruitment target). Newcastle upon Tyne Hospitals NHS Foundation Trust was the research sponsor.

Complete and transparent reporting

The following chapters of the report describe the objectives, methods, results, strengths and limitations of each element of the research, using EQUATOR (Enhancing the Quality and Transparency of Health Research) reporting guidelines (www.equator-network.org/; accessed 11 November 2019), GRIPP2,15 Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA)16 and Strengthening the Reporting of Observational studies in Epidemiology (STROBE). 17

Objectives

The objective was to update three published systematic reviews of interventions to improve the EDSD of children with neurodisability that were published before our research began.

This chapter outlines the methods and findings of the update of two of these reviews. 5,6 The third review4 was updated by the original review team during the period of our study. The update review followed the original review protocol. The authors of that review provided pre-publication data and these have been included at the end of this chapter for completeness.

Background

Three recently published systematic reviews relevant to interventions for young children with neurodisability and EDSD were identified at the proposal development stage. 4–6

Marshall et al. 5 reviewed evidence for interventions to improve feeding difficulties for children aged < 6 years with ASD that were published between January 2000 and October 2013. The review included 23 studies, which were a mix of single-case experimental design (n = 12) and small-group studies (n = 11), all with five or fewer participants who were aged 2–5 years. A total of 46 children participated, 37 of whom had ASD. The interventions were based on operant conditioning to ‘increase desirable eating behaviours or decrease undesirable eating behaviours’. 5 The risk of bias of each study was assessed using a tool developed for single-case experimental design by Horner et al. 18 The primary outcomes of interest were increased volume of food and variety of intake, which were considered to increase desirable mealtime behaviour. The secondary outcome of interest was a reduction of inappropriate mealtime behaviours. The authors concluded that there was a low level of evidence to support these types of interventions for children with ASD and that ‘favourable intervention outcomes were observed in terms of increasing volume, but not necessarily variety of foods consumed in young children with ASD and feeding difficulties’. 5

The NICE cerebral palsy guidance6 reviewed evidence generated by RCTs and observational studies of interventions for ‘management of eating, drinking and swallowing difficulties’ of children and young people with cerebral palsy aged < 25 years published before October 2014. The outcomes of interest in the review were:

• physiological function of the oropharyngeal mechanism (as determined by clinical evaluation, videofluoroscopic swallow studies or fibreoptic endoscopic evaluation of swallowing)

• changes in diet consistency that a child is able to consume (developmentally appropriate oral diet; texture and/or consistency of foods and fluids must be modified; supplementary feeding required)

• respiratory health – presence of a history of confirmed aspiration pneumonia or recurrent chest infection (with or without pneumonia with suspected prandial aspiration aetiology)

• nutritional status and/or changes in growth (weight and height percentiles)

• child and young person’s level of participation in mealtime routine/length of mealtimes (time taken to feed)

• psychological well-being of parents and/or carers

• acceptability of the programme

• survival.

The included publications comprised four RCTs and four cohort studies, which included a total of 235 participants who were aged 12 months to 21 years. The four RCTs all compared Oral sensorimotor therapy with routine therapy; each cohort evaluated a different intervention. One cohort considered the Innsbruck Sensorimotor Actuator and Regulator (ISMAR) compared with no ISMAR; one an Oral sensorimotor treatment; one a training programme delivered to children and caregivers; and one evaluated individual interventions delivered in combination (a multicomponent intervention), including carer training, behavioural interventions and Beckman Oral Motor Exercises. 19 The risk of bias was assessed using the Cochrane risk-of-bias tool. 20 Conclusions were not drawn from this review alone, but as part of the total guideline development process. This took account of stakeholder input to the evidence and consideration of the evidence by a committee made up of practitioners, professionals, care providers, commissioners, those who use services and family members or carers. Based solely on the published studies (n = 8) the Grading of Recommendations and Assessment, Development and Evaluation (GRADE) assessment rated the evidence to support these types of interventions in this population as being of very low to low quality. 6

Morgan et al. 4 considered interventions for children with any ‘neurologically based oropharyngeal dysphagia’. 4 The outcomes of interest were amount/variety of food and eating behaviours. Their review included two papers assessing sensorimotor treatment in 55 cerebral palsy patients aged 4–21 years; the studies were small and were rated as being at high risk of bias, as assessed by the Cochrane risk-of-bias tool. 20 Both of the papers were also included in the NICE6 review. Morgan et al. 4 concluded that there was ‘insufficient high-quality evidence from RCTs or quasi-RCTs to provide conclusive results about the effectiveness of any particular type of oral motor therapy for children with neurological impairment’. 4

Methods

We updated the review by Marshall et al. 5 and the NICE cerebral palsy review6 of EDSD interventions using the methods of the original reviews. Marshall et al. 5 provided the search strategy for the ASD review and the NICE review searches were based on the published search strategies. Updated searches were limited to 1 year before the date of the last searches undertaken for the original review, which allowed for delays in database updates. Protocols for the systematic reviews were registered on PROSPERO (www.crd.york.ac.uk/prospero/; accessed 11 November 2019). Full details of the search strategies are presented in Appendix 2.

Inclusion criteria

Studies were included in the update of Marshall et al. 5 if they met the following criteria:

1. Population – children aged 0–8 years with a diagnosis of ASD. (The original review included children aged 0–6 years, but we extended the age range to fit with our definition of young children with neurodisability.)

2. Intervention – non-pharmaceutical behavioural or environmental interventions.

3. Outcomes – amount of food and/or variety of foods consumed and/or desired or undesired eating behaviours.

4. Study design – an experimental design was used to investigate treatment outcomes, including the use of a control group, within-group designs or single-case-based experimental designs replicated across at least four participants (to give some indication of repeated effects of interventions). 18

5. Language – studies were published in English in peer-reviewed journals.

The update of the NICE review6 included studies that met the following criteria:

1. Population – children and young people aged 0–8 years with a diagnosis of cerebral palsy. (The original review considered young people aged < 25 years.)

2. Intervention – interventions that aimed to improve sucking/biting/chewing/swallowing of food, intake of food (amount and/or variety of food) and/or eating behaviours.

3. Outcomes – Co-ordination of chewing and swallowing, Increased amount and variety of intake, Duration of mealtimes, Nutritional status, Growth and Physical health (e.g. chest infections and mortality).

4. Study design – group experimental design.

5. Language – studies were published in English in peer-reviewed journals.

Screening/data extraction/quality assessment

For both review updates, two researchers (HT and LP) independently screened titles and abstracts to identify studies meeting the inclusion criteria. The full texts of potentially eligible articles were retrieved and assessed independently against inclusion criteria by two researchers (HT and LP or HM). Data extraction and quality assessment were conducted by one researcher (HT) and checked by a second researcher (LP).

We used the Cochrane risk-of-bias tool for randomised trials (Risk of Bias toolkit, version 2) to assess the quality of RCTs or quasi-RCTs that were included in either review update. 20 Critical judgements were made of the following domains: randomisation process, deviation from the intended intervention, missing outcome data, measurement of the outcome and selection of the reported results. Each included study was judged to be at ‘low risk of bias’, ‘high risk of bias’ or to give rise to ‘some concern of bias’ in each of these domains. An overall study risk of bias was established using the following criteria:

• low risk of bias – the study was judged to be at low risk of bias for all domains

• some concern of risk of bias – the study was judged to give rise to some concerns in at least one domain, but not judged to be at high risk of bias on any domain

• high risk of bias – the study was judged to be at high risk of bias in at least one domain, or the study was judged to give rise to some concerns for multiple domains in a way that substantially lowered confidence.

As in Marshall et al. ,5 we used the quality assessment tool developed by Horner et al. 18 to assess studies using single-case experimental design. This tool grades the presence of absence of 21 quality indicators within single-case experimental designs relating to (1) the description of participants and settings (three criteria), (2) dependent variable(s) (five criteria), (3) independent variable(s) (three criteria), (4) the establishment of a robust baseline measure and replicable description of the intervention (two criteria), (5) experimental control/internal validity (three criteria), (6) external validity (one criterion) and (7) social validity (four criteria). Each criterion is rated as met or unmet, giving a possible total of 21 per study.

Results

The numbers of references included and excluded at each stage of the study selection process for both review updates are shown in the PRISMA flow diagram (Figure 2).

Following de-duplication, 316 references remained for assessment against the two sets of review inclusion criteria. Of these, 286 references were excluded based on the information in the title and abstract. The remaining 30 references were then assessed based on the full-text publication. Of these, a further 24 were excluded from the update review. A total of six references, reporting six studies, met the inclusion criteria adapted from Marshall et al. 5 and NICE6 and were included: four references and two references, respectively (see Figure 2).

FIGURE 2.

Study selection PRISMA flow diagram. M, Marshall et al. 5: N, NICE.

Update of the Marshall et al.5 review focusing on children with autism spectrum disorder

Four studies were included in our update of the Marshall et al. 5 review (Box 3): two RCTs (one with a waiting list control21 and one with an active control22), one single-case experimental design23 (also referred to as a ‘n of 1’ study) replicated across participants and one pre–post pilot trial. 24 Nine single-case experimental design studies were excluded because they included fewer than five participants. These nine studies were included in the mapping review (see Chapter 4). Full descriptive details of the included studies are provided in the data extraction tables (see Appendix 3); however, a brief summary is provided here.

The total population across all four studies was 107 children aged 3–8 years with a diagnosis of ASD. Three of the four studies included training interventions delivered to parents to address their children’s food aversions, restricted diets or mealtime behaviour. 21,22,24 Peterson et al. 23 delivered caregiver training after the study, whereas Johnson et al. ,24 Marshall et al. 22 and Sharp et al. 21 incorporated training of the caregiver/parent by the therapist in a clinic setting, either from the beginning of the study or incrementally as the child/parents progressed. All of the studies included a behavioural intervention, using prompts (verbal and visual) and reinforcement of the child’s behaviour. Both Marshall et al. 22 and Peterson et al. 23 included a comparison intervention, with Graded exposure to food through modelling and play. In both studies the comparison intervention was delivered by the therapist; however, Marshall et al. 22 incorporated training of the caregiver/parent, whose involvement incrementally increased as the sessions progressed. The number and duration of sessions varied across the studies for both interventions and comparators, with one22 offering the choice of sessions delivered weekly for 10 weeks, or sessions delivered intensively within 1 week. The duration of follow-up ranged from 8 to 16 weeks. The most common target outcome was mealtime behaviours, both positive and disruptive. Other outcomes measured across the four studies comprised Dietary intake, Dietary variety, Food acceptance, Mouth clean, Grams consumed, Weight, Height, Body mass index (BMI), Behaviour outside mealtimes, Parent stress, Caregiver satisfaction and Feasibility.

The overall quality of the studies that were included in the update was rated as low. The two studies that used a single-case experimental design both scored highly on baseline and external validity criteria and poorly on social validity; both failed to meet two of the four elements of this criteria [(1) magnitude of change of dependent variable is socially important and (2) independent variable is practical and cost-effective]. In addition, Johnson et al. 24 scored poorly on the criteria description of participants/setting. The two RCTs21,22 were both considered to be at high risk of bias, with the main concerns being the measurement of outcomes and deviation from the intended interventions. Summaries of the quality assessments are presented in Table 1 and Figure 3 (see also Table 2).

FIGURE 3.

Quality assessment of RCTs using the Cochrane risk-of-bias tool, version 2. –, high risk of bias; +, low risk of bias; ?, unclear risk of bias.

Update of the National Institute for Health and Care Excellence cerebral palsy in under-25s review6

Two new studies were included in our update of the NICE review6 to inform the management of EDSD in children with cerebral palsy. 25,26 One was a RCT25 and the other was a small pilot study26 in preparation for a RCT. Full descriptive details of the included studies are provided in the data extraction tables (see Appendix 3); however, a brief summary is provided in Box 4.

The population of the two studies (combined, n = 100) was children who were aged 1.1–8 years with cerebral palsy. Serel Arslan et al. 25 evaluated a parent training intervention that was delivered by speech and language therapists, whereas Song et al. 26 piloted a therapist-delivered intervention. Both studies included interventions with multiple components that were carried out simultaneously and both included Positioning and Oral/sensory desensitisation.

Serel Arslan et al. 25 (n = 80 children) evaluated an intervention that comprised Functional chewing training, which was made up of five steps: positioning the child, positioning food, sensory stimulation, chewing exercises and adjustments to food consistency. This was carried out alongside Oral motor exercises: five sets per day, five days per week, for 12 weeks. In addition, parents were given a brochure on exercises.

Song et al. 26 compared Oral sensorimotor treatment (10 minutes) and neuromuscular electrical stimulation (20 minutes) twice weekly for 8 weeks with Oral sensorimotor treatment plus sham neuromuscular electrical stimulation in a pilot study. Oral sensorimotor treatment included various sensory stimuli that were applied to the cheeks, chin, lips, tongue and oral palate using human fingers, a vibrator and an ice stick.

Both studies measured Feeding behaviour as a primary outcome, but used different tools to evaluate progress. Serel Arslan et al. 25 also measured Chewing function, whereas Song et al. 26 measured Severity of dysphagia. The study by Song et al. 26 was a pilot study and as a consequence it was judged to be at high risk of bias, whereas the full study by Serel Arslan et al. 25 was considered to be at lower risk of bias. A summary of the quality assessments are presented in Table 2.

Update of the review of oropharyngeal dysphagia by Morgan et al.4

Searches and data extraction for an update of the review by Morgan et al. 4 were conducted by the original review team in 2018. An updated review is due to be published in 2020. Inclusion criteria for this review are:

1. Population – children and young people aged < 18 years with oropharyngeal dysphagia (i.e. difficulties in chewing or preparing food, moving food posteriorly with the tongue and swallowing food) diagnosed by a medical officer. Studies involving children with oesophageal dysphagia, including lower oesophageal sphincter dysfunction and gastro-oesophageal reflux, were excluded.

2. Intervention – any intervention that aimed to improve body functions underpinning eating/drinking, eating/drinking or participation in mealtimes.

3. Outcomes – Physiological Function, Aspiration and chest health, Diet consumed, Growth, Participation at mealtimes and Parental stress.

4. Study design – RCTs and quasi-RCTs.

5. Language – studies were published in English in peer-reviewed journals.

The review by Morgan et al. 4 included three studies (two involving children with cerebral palsy, both of which were included in the NICE review,6 and one additional study that focused on children with myotonic dystrophy). In their recent searches, Morgan et al. 4 identified one additional study: Sığan et al. 27 This study was a RCT and included 81 children with cerebral palsy who were aged 12–42 months. At the time of this report, Morgan et al. 4 had not extracted data from this paper or reviewed its quality. However, it was included in the original review by NICE6 and, therefore, has already been systematically appraised; it was rated as being low quality in the NICE review. 6

Strengths and limitations of systematic reviews

To make best use of these published systematic reviews of interventions we updated each using the methodology of the original review, with the exception of the age criterion. To align with our project aims we extended the age criterion to 8 years for all of the review updates. By following each of the review methods different risk-of-bias and assessment tools were used across the updates to appraise the specific quality markers of RCTs and single-case experimental designs. Although the tools for assessment differ, resulting in different presentations, the criteria against which risk of bias and quality are being assessed are generally the same. We have assumed that the original searches and processes were robust enough to identify all relevant studies, as each of the reviews followed established systematic review processes. Each review includes and represented the best available evidence (with the inclusion of experimental designs) around effective management of EDSD in children with cerebral palsy or ASD. Our updates have ensured that the findings and recommendations of these reviews remain up to date.

Patient and public involvement in systematic reviews

The PAG considered the summaries of the findings from the updated systematic reviews of interventions alongside findings from the mapping review and national survey. The PAG advised on creating a pictorial summary of the identified interventions and outcomes to aid discussion in the second focus groups and the stakeholder consultation workshops (see Figures 9 and 10).

How did the systematic review findings inform the next step?

The interventions and outcomes identified in the updates of the published systematic reviews of interventions were considered by parents and professionals in the first focus groups with regard to their use in the UK (see Chapter 5). They also directly informed the design of the national survey of current practice (see Chapter 7) and the outcome measures used in studies included in the updates of the three published systematic reviews of interventions were listed for inclusion in the systematic review of measurement properties of tools (see Chapter 6).

Objectives

• To review the clinical practice and research evidence for the interventions, outcomes measured and tools used to measure these outcomes for EDSD in young children with neurodisability.

• To identify the subgroups of children for whom there is the most robust evidence on intervention success/failure.

• To investigate the extent to which interventions have been defined and manualised to facilitate replication.

Methods

We searched for literature pertaining to any intervention that aimed to improve EDSD for children with neurodisability. This was a mapping review rather than a systematic review to establish an estimate of the effectiveness or assess the quality of the evidence. Nonetheless, the approach taken to searching and screening was rigorous and consistent with that used in a systematic review.

Inclusion criteria

Literature was included in the mapping review if it met the following criteria:

• Population – children (aged 0–8 years) with any type of non-progressive neurodisability who had EDSD. The following conditions were excluded: cystic fibrosis, gastro-oesophageal reflux and structural abnormalities [e.g. cleft lip and palate, and CHARGE (coloboma, heart defects, choanal atresia, growth retardation, genital abnormalities and ear abnormalities) syndrome]. Children who had rumination (i.e. persistent regurgitation, re-chewing, re-swallowing or vomiting of previously eaten foods), eating disorders (unless specifically about food avoidance/restrictions not related to a desire for thinness) or problem behaviour at mealtimes that was not related to eating were also excluded. Studies were included if any of the participants were aged 0–8 years.

• Intervention – any intervention to improve eating, drinking and swallowing that can be delivered by parents to their children aged 0–8 years. The following interventions were excluded: Pharmacological, Dietary or Nutritional interventions, Gastrostomy and Oral appliances. Interventions that focused on speech development or improvement and the swallowing of tablets were also excluded.

• Comparator – any other intervention for eating, drinking and swallowing or mealtime behaviour, any intervention described as ‘treatment as usual’ or no intervention.

• Outcome – any outcome pertaining to food intake, behaviour, health, well-being or acceptability.

• Study design – systematic reviews of interventions and any controlled or non-controlled study of intervention effects or acceptability. Editorial/commentary/opinion articles were excluded.

• Limitations – manuscripts written in English and published from January 1985 to October 2017.

Searches were designed by an information specialist in collaboration with the project team. The search strategy was designed on MEDLINE [via Ovid^Ⓡ (Wolters Kluwer, Alphen aan den Rijn, the Netherlands)] using thesaurus headings and title and abstract keywords, and translated as appropriate to the following databases: Cumulative Index to Nursing and Allied Health Literature (CINAHL) database [via EBSCOhost (EBSCO Information Services, Ipswich, MA, USA)], PsycINFO (via Ovid), Web of Science™ (WoS; Clarivate Analytics, Philadelphia, PA, USA), EMBASE™ (Elsevier, Amsterdam, the Netherlands) (via Ovid), Education Resources Information Center (ERIC) (via EBSCOhost), Cochrane Database of Systematic Reviews [via Wiley Online Library (John Wiley & Sons, Inc., Hoboken, NJ, USA)], Cochrane Central Register of Controlled Trials (CENTRAL) (via Wiley Online Library), The Speech Pathology Database for Best Interventions and Treatment Efficacy (speechBITE; The University of Sydney Lidcombe, NSW, Australia) (www.speechbite.com) and Occupational Therapy Systematic Evaluation of Evidence (OTseeker; www.otseeker.com). This search was run between 5 October and 17 October 2017. Full details of the search strategies are presented in Appendix 4. Two researchers (HT and LP) independently screened titles and abstracts to identify studies meeting the inclusion criteria. The full texts of potentially eligible articles were retrieved and assessed independently against inclusion criteria by two researchers (HT and LP or HM). Where there were discrepancies in these processes, a third person from the review team was consulted and a consensus was reached. One researcher (HT) extracted the data and classified each study; LP checked the data extraction and coding.

Results

Our searches identified 5790 references; following sifting on title and abstract, we retrieved 492 full texts, of which 147 fitted the inclusion criteria (Figure 4). Fifteen of the papers identified through the updates of the published systematic reviews of interventions (those from the Marshall et al. 5 and NICE6 updates) were also found in the mapping review, including nine single-case experimental design studies replicated across fewer than four participants.

FIGURE 4.

The PRISMA flow chart of mapping review.

Study participants ranged in age from < 1 year to 31 years, with many of the studies including participants outside the age range that we defined as ‘young children’ (i.e. ≥ 9 years). In most cases, the results for our target group of young children (aged 0–8 years) could not be disaggregated. The interventions reported across the studies were grouped as addressing physical, mixed or non-physical factors affecting eating, drinking and swallowing: 27 studies addressed physical EDSD, 53 non-physical EDSD and 66 mixed EDSD. Most interventions directly targeted EDSD, such as Modifications (Environment, Equipment, Food or drink, Placement of food and Positioning), improved mealtime communication (Enhancing communication strategies, Responding to the child’s cues for feeding and Pace of feeding) and desensitisation strategies (Graded exposure to foods or textures, and Oral and sensory desensitisation). Other interventions that did not directly target EDSD included Psychological support for child and parent and Self-feeding. Teaching techniques (Prompting and Reinforcement) were referred to frequently in the teaching of any of these interventions. There was a range of outcomes measured across these studies including Swallowing function, Chest health, Amount of food eaten, Eating efficiency, Oral motor function, Number (percentage) of bites, Variety of food consumed, Mealtime behaviour, Self-feeding, Food acceptance and Amount of liquid consumed. Further details of the included studies are presented in Table 3.

The totals for the number of studies and included participants do not include the systematic or literature reviews to prevent double counting and due to the reviews including a large number of studies that did not meet the criteria for inclusion.

The majority of studies described multicomponent interventions; for example, an intervention might ensure that the children were in a safe position to eat and drink (Positioning), were fed textures that they could swallow easily (Modifying food or drink) and received praise for swallowing (Reinforcement). The frequency of individual interventions studies included in the mapping review is shown in Figure 5. The mapping process enabled us to disaggregate multicomponent interventions to explore a number of questions, including (1) which individual interventions were more frequently provided together as a multicomponent intervention; (2) the difference in the frequency of interventions between participants with physical and mixed EDSD and participants with non-physical EDSD; and (3) the number of participants in whom each intervention had been assessed.

FIGURE 5.

Number of studies and included participants by intervention and type of EDSD.

We identified 18 individual interventions, most of which had been assessed within a multicomponent intervention that included participants with physical, mixed and non-physical EDSD (see Figure 5). In Figure 5, we have presented the frequency of assessment of each intervention, based on the number of studies, alongside the total number of participants in those studies. The frequency count is based on the number of studies reporting the primary outcome. Based on the number of studies, the most common individual interventions considered across the populations were Reinforcement (109 studies, 554 participants) and Prompting (97 studies, 393 participants). However, these individual interventions are teaching techniques to support the delivery of specific EDSD interventions. Beyond the teaching interventions/techniques, the most commonly assessed interventions for children with physical or mixed EDSD were Modification of food or drink (33 studies, 519 participants), Positioning (22 studies, 456 participants), Modifying equipment (19 studies, 194 participants) and Oral motor exercises (17 studies, 498 participants). The most commonly assessed interventions for children with non-physical EDSD were Modification of food or drink (26 studies, 104 participants), followed by Visual supports (19 studies, 120 participants), Food desensitisation (18 studies, 124 participants) and Scheduling of meals (17 studies, 139 participants). Psychological support for parents (three studies, 44 participants) and Responding to a child’s cues for feeding (three studies, 72 participants) were assessed only for participants with physical or mixed EDSD; however, Psychological support for the child (one study, 11 participants) was found in an intervention assessing only children with non-physical EDSD. Although Psychological support for parents and the child and Responding to a child’s cues for feeding were included in studies infrequently (i.e. evaluated in fewer than five studies), a large number of individual interventions were seen in more than 10 studies of both children with physical or mixed EDSD and children with non-physical EDSD. Figure 5 illustrates the significant overlap in the individual interventions being considered for children with physical or mixed and non-physical EDSD. Only three interventions were considered in only one of the populations: Responding to a child’s cues for feedings (three studies) and Psychological support for the parents (three studies) were considered in only a non-physical population, and Psychological support for the child (one study) was considered in only children with physical and mixed EDSD. These three individual interventions were also the least frequently considered.

A range of study designs, from those providing the highest level of evidence (systematic reviews of RCTs) to those providing the lowest (case studies), were used to evaluate the interventions (Figure 6 and Table 3). Appendix 5 shows the study designs used to evaluate each intervention. Figure 6 shows that a large number of studies had designs that are widely considered to be less robust and, therefore, more prone to bias, such as case studies and before-and-after studies. In total we identified 147 studies, 121 of which were before-and-after studies, case studies, literature reviews or single-case experimental designs. We also identified 12 RCT/quasi-experimental design studies and 14 systematic reviews. The amount of evidence included in these reviews was variable and they provided no robust conclusions regarding the optimal multicomponent intervention.

FIGURE 6.

Types of study design and numbers of participants by causes of EDSD.

Outcomes

The studies in the review measured 24 different outcomes (as shown in Table 3), with most studies measuring multiple outcomes.

Measures

The studies used 33 published protocols/measures to assess change in the outcomes, as shown in Table 3. A total of 25 studies used published protocols/measures, with the remaining studies using bespoke measures that the authors had developed specifically for use in their study. These bespoke measures lacked evidence of reliability, validity or responsiveness to change.

Summary of mapping review

The mapping review collated a wide range of research evidence. The scope and purpose of the mapping review was not to assess the quality of the individual studies but rather to explore and understand the frequency and level of evidence for each of the individual interventions. The aim was to present a full picture of the interventions that have already been developed and/or evaluated, to ensure that all of the potential interventions were considered in the later stages of this work. The level of evidence found suggests that there are several studies evaluating some of the individual interventions identified; however, owing to the design of the studies it is likely to be low-level evidence, given that there is a lack of RCTs. The mapping review also highlighted the significant overlaps in the interventions delivered to children with physical or mixed and non-physical EDSD in these studies.

Strengths and limitations of the mapping review

The mapping review has elicited the number of published studies evaluating each individual intervention type, the study designs used and the number of participants with physical or mixed and non-physical EDSD included. Considering the number of studies identified, it was necessary to take a pragmatic approach to data extraction, ensuring that only the necessary information was retrieved. Furthermore, we have not explored the data beyond the scope of the question that we set out to address. There are many other questions that this literature base might support answering; however, these were out of scope of this research.

Patient and public involvement in the mapping review

Parent co-investigators checked the intervention and condition terms that were included in the searches for the mapping review. They also discussed the search findings with members of the research team, to help ensure that all relevant studies were being retrieved. The PAG considered the summaries of the findings from the mapping review alongside findings from the updates of the three published systematic reviews of interventions and national survey. The PAG commented on a pictorial summary of the identified interventions and outcomes to aid discussion in the second round of focus groups (see Chapter 9) and the stakeholder consultation workshops (see Chapter 11) (see Figures 9 and 10).

How did the mapping review inform the next step?

The mapping review provided information regarding the evidence base for interventions aimed at improving EDSD in children with neurodisability, the outcomes measured and the tools used to measure those outcomes. This information informed the list of interventions and outcomes included in the national survey (see Chapter 7), the discussions within the second round of focus groups (see Chapter 9) and the searches for papers examining relevant outcome measurement tools for the measurement properties review (see Chapter 6).

Objective

The objective was to gain an understanding of the interventions offered through NHS services to parents of children with neurodisability who experience EDSD by consulting with parents and HPs to inform the development of a national survey of current practice.

Methods

Results

Interventions, outcomes and measures used by parents and health professionals

Parents reported that they currently used or had used a wide range of interventions, often in combination. Similarly, HPs reported that they or their colleagues recommended use of a range of interventions, sometimes simultaneously or in an additive approach (Box 5). Parents and HPs discussed the potentially stressful experience of mealtimes, and the importance of understanding the nature of both the children’s difficulties and their communication skills to implement any changes at mealtimes. Parents also stressed the variability in children’s eating and drinking from day to day. Both parents and HPs reported that children’s physical health and developmental progress were important outcomes of EDSD interventions, but also highlighted the social nature of mealtimes and outcomes related to enjoyment of food and eating with others. Although participants identified a substantial list of individual areas in which to measure progress or outcome, few participants reported a formal measurement that was used to evaluate progress. Tools that were mentioned were food diaries, weight, number of spoonfuls eaten in a meal and Therapy Outcome Measures (TOMs),64 which includes a Dysphagia scale.

Summary of findings

Parents and HPs reported using a wide range of interventions to enable children to use their current skills to eat and drink safely, or to teach new skills. They often used interventions in combination. They viewed children’s physical health and developmental progress, children’s enjoyment of meals and participation in meals as social activities as outcomes to target through intervention. HPs seldom used formal outcome measures to evaluate intervention success.

Strengths and limitations of the first round of focus groups

The first round of focus groups comprised a small sample of parents and HPs from one region of England served by numerous secondary-level services and one tertiary-level service. In the small sample, most of the parents had children with non-physical EDSD. The sample and the service organisations may not be representative of families receiving EDSD services or service providers across the UK. The children of some parents included in the groups were older than the target age range of the study. This meant that parents were recalling previous interventions and outcomes. However, their children had ongoing EDSD and parents were able to recall differences in interventions by school age, for example preschool and primary school, which was important for our study. The small size of the groups meant that there was time for all participants to relate their experiences.

Patient and public involvement in the first focus groups

The parent co-investigators recommended that separate groups be held for parents of children with physical or mixed EDSD and for parents of children with non-physical EDSD, as similar shared experiences may help the groups to gel and encourage discussion. The parent co-investigators reviewed the topic guide and agreed that the questions should initially be open, without examples of interventions and outcomes. The PAG reviewed the summary lists of the interventions and outcomes reported in the focus groups. They agreed that each intervention and outcome identified should be included in the survey.

How did the first focus groups inform the next step?

Parent and HPs in the focus groups endorsed all of the interventions and outcomes identified in the reviews; therefore, all were included in the national survey of UK parents’ and HPs’ use of EDSD interventions and evaluation of their outcomes (see Chapter 7).

Objective

To examine the psychometric robustness of tools used to measure change in EDSD in young children with EDSD.

Methods

Outcome measurement tools were primarily identified through the updates of the three published systematic reviews of interventions and mapping review, as well as the first round of focus groups and the survey (see Chapters 3–5 and 7). The list of 43 named tools focused on the evaluation of EDSD-related outcomes of interventions (see Appendix 7). Tools mentioned in the literature, or by participants in the focus groups and survey, that measured other important outcomes (e.g. Parent stress and Child quality of life) were not included in the listing and review of measurement properties.

Searches were conducted to specifically identify papers that examined the measurement properties of the named tools. The search strategy included terms for neurodisability or feeding disorders and children, and included a COnsensus-based Standards for the Selection of health status Measurement INstruments (COSMIN) filter (www.cosmin.nl; accessed 11 November 2019) (see Appendix 8). The COSMIN system was developed by an international group of experts to standardise assessment of the methodological quality of measurement studies. Papers identified from searching are examined in terms of the quality of the research study examining a tool’s measurement properties (e.g. whether the study had sufficient numbers of participants, clear hypotheses stated and a robust approach to the conduct of factor analysis); the paper is rated as ‘inadequate’, ‘doubtful’, ‘adequate’ or ‘very good’ on each property. Next, the data presented in each paper on reliability, validity and responsiveness to change are extracted and judged on COSMIN criteria (e.g. a cut-off point for a good reported level of inter-rater reliability) as being of a ‘sufficient’, ‘insufficient’ or ‘indeterminate’ level. Finally, the evidence is synthesised, following standard COSMIN criteria, to determine the strength of the evidence on each measurement property across all papers that examine any one particular measurement tool.

The following databases were searched: MEDLINE, CINAHL, PsycINFO and WoS. Following the initial searches of MEDLINE and PsycINFO on the 43 listed tools, further named tools of potential relevance were identified in the texts of the measurement properties papers, for example as criterion reference tools or revised versions (n = 21) [e.g. a paper on the Parent Mealtime Action Scale (PMAS) concerned development of a revised scale, the Parent Mealtime Action Scale – Revised (PMAS–R)]. The searches were updated to include the new tool names; thus, in total, we searched for papers on the measurement properties of 64 named tools.

Two reviewers (HM, a clinical psychologist who is experienced in systematic reviewing including use of the COSMIN approach, and CU, systematic reviewer in health research) sifted search results by title and abstract for likely relevance, using definitions and criteria agreed with the research team (see Appendix 9); uncertainty was resolved by requesting the full text. Papers were examined for inclusion at the full-text stage separately by both reviewers, with any disagreement settled by referral to a third reviewer (LP).

At the full-text stage, and before data extraction, a strategy was developed to focus on the most promising outcome measurement tools for application in a future trial. The criteria for exclusion comprised the age range covered (e.g. excluded the Neonatal Oral Motor Assessment Scale); not being about EDSD once further information was available from papers (e.g. Motivation Assessment Scale); cost of training and/or poor availability of training (e.g. Dysphagia Disorders Survey); precision of measurement [e.g. excluding the TOMs and the Eating and Drinking Ability Classification System for individuals with cerebral palsy (EDACS) and other similar classifications of function on 4- to 7-point scales]; or having been poorly rated on measurement properties in a published systematic review of measures of oropharyngeal dysphagia for preschool children with cerebral palsy and neurodevelopmental disabilities by Benfer et al. 65 Based on these criteria, 20 tools were not considered further.

Data extraction followed the COSMIN risk-of-bias checklist on reliability and validity of the evidence,66 with additional information noted on acceptability, feasibility, precision and interpretability from Fitzpatrick et al. 67 The COSMIN criteria for judgement of good measurement properties were taken from Prinsen. 66 The two reviewers trained together on papers and established reliability, before proceeding with data extraction (in addition, HM checked all data extracted by CU). Data were not extracted from papers with a sample of < 10 participants, with a non-relevant sample (e.g. a feeding clinic sample not further described) or when the paper was not about a measurement property.

Results

Results of searches

Papers from the first searches of PsycINFO and MEDLINE were sifted by title and abstract; 51 out of 560 papers and 18 out of 196 papers, respectively, were taken forward to sifting at full text. The reviewers checked 11% of articles (i.e. 88 articles) with 92% agreement on ‘get full text’/exclusion.

In the second search, CINAHL and WoS were added, as well as searching for the 21 additional named tools identified in the previous search. The searches of all four databases yielded 888 references, from which a total of 111 went forward to be sifted at full text. The two reviewers double checked 17.3% of articles, with 94% agreement on ‘get full text’/exclusion.

After de-duplication, a total of 127 papers were sifted as full text from the first and second searches. Of these, 86 papers were excluded and, therefore, 41 papers were included for data extraction (Figure 7).

FIGURE 7.

The PRISMA flow chart.

Conclusions

The strongest evidence for robust measurement properties of a tool measuring child behaviours was for the Paediatric Eating Assessment Tool (PediEAT),71,72 a 78-item parent questionnaire that is used with children aged 6 months to 7 years (see Tables 5–7). Evidence for the measurement properties of the Brief Autism Mealtime Behaviour Inventory (BAMBI),50 an 18-item parent questionnaire (or 15 items51) for children aged 2–11 years, is more mixed. The BAMBI was subsequently reworked for all children with feeding problems as the Brief Assessment of Mealtime Behaviour in Children (BAMBIC),68 a 10-item parent-reported scale relating to children aged 18 months to 17 years, which also had mixed evidence of robustness of its measurement properties. The evidence relating to seven further tools measuring child behaviours was limited and poor; therefore, these tools will not be considered further.

In addition, there were two types of tool used to measure children’s intake in this category (i.e. food frequency and food preferences) (see Tables 5–7). The Food Frequency Questionnaire (FFQ)61 evaluated was based on the youth/adolescent version of a list developed at Harvard University. 86,87 It has 131 items, and parents indicate foods refused and the number of times per day foods are eaten. Evidence was found only in relation to hypothesis testing of convergent validity and discrimination between groups.

In regard to parent strategies tools, the evidence of measurement property robustness is sparse and poor (see Tables 5–7). Among the child behaviours tools, the Behavioural Paediatric Feeding Assessment Scale (BPFAS),33 Child’s Eating Behaviour Inventory35 and Meals in Our Household70 also have parent domains. The BPFAS has a little more evidence of robustness for use with this population. The BPFAS has 25 items on child behaviours and 10 items on parent attitudes and strategies, relevant for those children aged 2–6 years.

The systematic reviews of parent-reported measures of feeding difficulties by Sanchez et al. 88 and Jaafar et al. 89 supported the BPFAS as the most robust measurement tool for children with feeding problems aged 2–5 years. In studies identified by our measurement properties review, it was used with children with a range of conditions, including ASD. 90,91

Regarding oral motor skills, the strongest evidence is for the Schedule of Oral Motor Assessment (SOMA). 45,92 The SOMA involves a structured assessment of children’s ability with a range of food textures and trained observers to rate the video-taped session. The strength of this tool was also concluded by the systematic review by Benfer et al. ,65 although more recent reviews by Barton et al. 93 and Speyer et al. 94 more cautiously reported the difficulty of reaching any conclusion on the basis of the patchy evidence.

Consultation

As the evidence from the review became available, three of the scales (PediEAT, BAMBIC and BPFAS) with the more robust properties were presented to the PAG in September 2018. For child behaviours, parents commented that the PediEAT was long. The BAMBIC was thought to be relatively easy to fill in, but short. The parents in the advisory group considered that some child behaviour items of the BPFAS were difficult to answer for particular situations, for example ‘eats junky snack foods but will not eat at mealtime’ as a child may eat junky snack foods as a meal at mealtime. The parent co-investigators also commented on the FFQ, with reservations expressed about the affordability of some of the range of foods included.

Summary of findings

The review of the papers on measurement properties of tools relevant to EDSD revealed the patchiness of the available evidence. A similar conclusion was reported by other recent reviews examining parts of this topic. 95,96 The review enabled the recommendation of candidate tools to be included in the design of outcome measurement in any future trial of interventions for EDSD in children with neurodisability. The PediEAT is a recently developed parent-report tool that measures child feeding difficulties and mealtime behaviours, and had the most evidence of robust measurement properties. For child intake, a FFQ might be acceptable. The BPFAS combines child and parent subscales, and had marginally stronger measurement properties than other parent strategy scales. The SOMA is the strongest measure of oral motor skills, and requires training of the assessors and observers.

Strengths and limitations of the measurement properties review

The identification of tools to be included in the review was comprehensive as it drew on a range of sources, including the mapping review. The inclusion of further tools identified within papers of measurement properties increased the breadth of the review. The pragmatic decision to focus data extraction on tools that were most likely to be candidates for use in evaluation in any future trial of interventions for EDSD was a limitation, but also an expedient best use of resources. The two reviewers worked closely together, to a detailed set of definitions, with access to another experienced reviewer to resolve discrepancies. Conclusions about the robustness of tools were checked against similar systematic reviews. The COSMIN approach to evaluate measurement properties is not applicable to the way in which some of the most valued outcomes are measured (e.g. Growth and Nutrition), which implies that a further review of approaches to their measurement will be required.

Patient and public involvement in the measurement properties review

Members of the PAG were presented with three parent-reported tools with the strongest evidence of their measurement properties, and commented on wording, layout and ease of use.

How did the measurement properties review inform the next step?

The three parent-reported tools that captured child behaviours, child intake of food and parent strategies were presented to groups at the stakeholder consultation workshops for further discussion (see Chapter 11). The process of the review also informed the thinking of the research team around how to conceptualise categories of outcomes valued by parents and professionals as elicited in the focus groups (see Chapter 9) and in the surveys (see Chapters 7 and 10). The tools reviewed for their measurement properties belong mostly to the intermediate category of child behaviours and parent strategies relating to EDSD, with child oral motor skills being grouped with proximal outcomes, such as Nutrition and Growth (see Chapter 9).

Objectives

To establish current UK clinical practice in relation to the interventions recommended and used for EDSD in young children with neurodisability and the outcomes measured, from the perspective of parents and health and education professionals.

Methods

Questionnaire development: UK clinical practice in managing eating, drinking and swallowing difficulties in young children with neurodisability

A questionnaire was developed to ascertain the interventions recommended and used for EDSD in young children with neurodisability and the outcomes measured, from the perspective of parents, HPs and education professionals. Lists of interventions and outcomes to be included within the questionnaire were generated from the updates of the three published systematic reviews of interventions (see Chapter 3), the mapping review (see Chapter 4) and the first focus groups (see Chapter 5). An additional four interventions were added by the research team based on their knowledge and expertise: Sensory stimulation, Medication, Sensory aids and Modifying social eating and drinking opportunities. An additional four outcomes were added by the research team based on their knowledge and expertise: Fewer breathing changes, More involvement in family activities, Fewer abnormal/unusual movements and More opportunity to talk to others about feelings regarding the child’s EDSD. The research team developed the questionnaires, drawing on clinical expertise, experience of survey design and best practice in the design and conduct of survey questionnaires. 97 The final online surveys and paper versions were piloted by a small number of parents, HPs, education professionals and researchers.

The final questionnaire had three sections: (1) demographic characteristics; (2) items about interventions, including usage, effectiveness, acceptability (i.e. was it acceptable to deliver at home or school), timescales for change and training; and (3) important outcomes. A total of 25 interventions and 32 outcomes were listed. The interventions were compiled from those identified through the updates of the three published systematic reviews of interventions (n = 13; see Chapter 3), the mapping review (n = 19; see Chapter 4) and the first round of focus groups (n = 21; see Chapter 5). The outcomes were compiled from those identified through the updates of the three published systematic reviews of interventions (n = 10; see Chapter 3), the mapping review (n = 24; see Chapter 4) and the first round of focus groups (n = 25; see Chapter 5). A number of the interventions and outcomes appeared in more than one research activity so duplicates were taken out. Respondents had the option to add additional interventions or outcomes. HPs were also asked about the tools that they used to measure outcomes. Most of the questions offered fixed-choice responses, with some opportunities for free-text responses. A small proportion of the questions were compulsory, such as those relating demographic information and whether or not participants had used a particular intervention. Questions asking for further detail about each intervention (e.g. whether or not it was acceptable to deliver) were answered only if respondents had experience of using that intervention. Respondents could use a ‘back’ button to review or change their answers as required, and the survey could be saved and completed at a later time or date. A ‘completion bar’ showed the respondents progress through the survey (see Report Supplementary Material 1 for the national survey questionnaire).

Procedure

Convenience samples of parents, HPs and education professionals were recruited. Recruitment was UK-wide, and took place between March and September 2018.

Parents were recruited via national and regional parent networks, parent support organisations and charities. These comprised:

• parent carer forums

• special needs networks

• the Council for Disabled Children (London, UK)

• Cerebra (Carmarthen, UK)

• Contact (London, UK)

• the National Autistic Society (London, UK)

• the Down’s Syndrome Association (London, UK)

• Down’s Syndrome North East (Newton Aycliffe, UK)

• Cerebral Palsy UK

• Action Cerebral Palsy (Bicester, UK)

• SCOPE (London, UK)

• Skills for People (Newcastle upon Tyne, UK)

• the National Network of Parent Carer Forums (London, UK)

• the Toby Henderson Trust (Bedlington, UK).

Parents were also recruited through two research databases: Autism Spectrum Database – UK (ASD-UK) and the Database of Children with Autism Spectrum Disorder Living in the North East (Daslⁿe). Further sources of parent recruitment were 24 NHS trusts across England that joined as participant identification centres (PICs), mainstream and specialist schools across the UK, participants in the first round of focus groups and social media linked to relevant organisations, charities and parent forums.

Health professionals were approached through relevant professional bodies. These comprised:

• the British Association of Community Child Health (London, UK)

• the Royal College of Speech and Language Therapists (London, UK)

• the British Dietetic Association (London, UK)

• the British Academy of Childhood Disability (through the UK Child Development Team database) (London, UK)

• the British Society of Paediatric Gastroenterology, Hepatology and Nutrition (London, UK)

• the College of Occupational Therapy (London, UK)

• the Chartered Society of Physiotherapists (London, UK).

Health professionals were also recruited through neurodisability and community paediatric networks, such as regional dysphagia clinical excellence networks, special interest dietetic groups, and local and national nursing and health visitor networks. In addition, some HPs were recruited through clinical services in the north-east and south-east of England (areas in which members of the research team were based) and the 24 PICs. Finally, HPs were also recruited through social media linked to relevant organisations and professional forums, and from participants who took part in the first round of focus groups (see Chapter 5).

Education professionals were recruited through independent and local authority schools across the UK, which were identified from websites such as Special Needs UK (URL: www.specialneedsuk.org, accessed 17 December 2020) and local authority websites. Education professionals were also recruited through local special education needs co-ordinator and Sure Start networks in the north-east of England. Further sources of education professional recruitment were via PICs and through relevant professional bodies, networks and voluntary organisations via social media, including:

• the Department for Education

• SEN magazine (Clitheroe, UK)

• National Association of Head Teachers (Haywards Heath, UK)

• Autism Education Trust (London, UK),

• National Day Nurseries Association (Huddersfield, UK),

• Early Education: The British Association for Early Childhood Education (St Albans, UK)

• Teach Early Years magazine (London, UK)

• Nursery World News (London, UK)

• Percy Hedley Foundation (Newcastle upon Tyne, UK)

• Childcare News (Milton Keynes, UK).

Separate, yet similar, versions of the questionnaire were developed for each of the three study groups (parents, HPs and education professionals). Electronic, web-accessible versions were hosted by Newcastle University, Newcastle, using Qualtrics (Provo, UT, USA), with paper versions available on request. E-mail and web-based flyers were sent to potential participants with a link to the appropriate version of the questionnaire alongside information on how to request a paper copy if this was preferred. Paper versions of the questionnaire were also distributed to PICs on request. At the end of the questionnaire, respondents were asked to provide their contact details if they would like to enter a prize draw to win one of five £100 vouchers for each of the three study groups, to be contacted with information about the Delphi survey (see Chapter 10) later in the study or to receive a summary newsletter about the results of the study. Reminders were sent out to parents, HPs and education professionals through the same sources to encourage non-respondents to participate.

Results

Participants

Figure 8 shows participant recruitment and flow through the study.

FIGURE 8.

Participant recruitment and flow through study.

Use of interventions

Table 13 shows that parents reported using a wide range of interventions. The most commonly reported interventions used by parents were Food or drink modification (57%), Desensitisation programme for food avoidance (47%), Modification of utensils (41%), Enhancing parent–child communication strategies at mealtimes (41%) and Positioning (40%). Food or drink modification was the most frequently reported intervention used with children with physical and mixed EDSD (69%) and with children with non-physical EDSD (48%). Parents of children with physical and mixed EDSD also reported frequently using Positioning (62%), whereas parents of children with non-physical EDSD frequently reported the use of strategies to Enhance parent–child communication at mealtimes (46%). Few parents reported using Manoeuvres (11%), Modelling (10%), having Counselling (5%) or using Sensorimotor therapy (3%).

Health professionals also reported using a wide range of interventions, with the majority using multiple interventions (median 11). The most frequently used interventions by HPs were Positioning (77%); Food or drink modification, such as texture or consistency (56%); Modification of environment (52%); Information on the impact of sensory (51%) or movement (49%) difficulties on eating and drinking, and Desensitisation programme for food avoidance (49%). Some interventions were used by a minority of HPs, with over half reporting that they did not use them: Sensory stimulation (60%), Modelling (60%), Sensory aids (64%) and Sensorimotor therapy (71%).

The majority of education professionals had been involved in delivering strategies aimed at improving EDSD (82%). The most commonly delivered interventions were Enhancing parent–child communication strategies at mealtimes (71%), Hand-over-hand prompting (71%), Food or drink modification (68%) and Modification of utensils (68%). Education professionals working with children with physical and mixed EDSD reported Positioning as their most frequently used intervention (77%), alongside those listed above. Education professionals working with children with non-physical EDSD also frequently reported using Modification of environment (50%), Desensitisation programme for food avoidance (50%), Modification of utensils (50%), Visual supports (50%), Schedule of meals (50%), Strategies/programmes aimed at changing behaviour at mealtimes (50%) and Modifying social eating and drinking opportunities (50%). However, as only a small number of education professionals (n = 10) completing this survey worked solely with children with non-physical EDSD (16%), these findings should be interpreted with caution. Some interventions were rarely delivered by education professionals, with over half of the respondents reporting that they had not been involved with Sensorimotor therapy (60%) or Counselling (65%). There was considerable overlap in professionals’ use of interventions. Both HPs and education professionals frequently used a range of interventions, including Positioning, Modifications (to Food and drink, Utensils and the Environment), Information about the impact of the child’s sensory difficulties on EDSD and Food desensitisation.

Delivery of interventions

Health professionals delivered interventions across a range of settings: families’ homes; school and preschool settings; NHS settings including hospitals and community provision; respite services including short break services, residential care and hospices; and other settings, such as independent services. The majority of HPs reported offering ongoing support with individual interventions, although a proportion offered these only as part of a time-limited programme and a small minority provided advice around each intervention on a single occasion only. All interventions, with the exception of Sensorimotor therapy, were recommended for both children with physical and mixed EDSD and children with non-physical EDSD, indicating a common approach to working with children with neurodisability and EDSD regardless of the cause of their difficulty. All interventions were used across the age range that was included in the survey (0–18 years). The majority of HPs worked with parents (87.9%) and education professionals (68.6%) to deliver interventions. HPs reported offering training on delivering interventions to parents (mean across interventions 91.1%, range 73.7–97.0%) and education professionals (mean across interventions 71.6%, range 25.9–84.4%). Similar levels of training were reported as having been received from HPs by parents (mean 91.1%, range 74.1–97.0%) and education professionals (mean 68.6%, range 38.9–93.3%).

Acceptability of interventions

Table 14 shows that all of the interventions in use were acceptable to parents to deliver at home (mean 94%, range 80–100%) and to education professionals to deliver in school (mean 95.1%, range 75–100%).

Effectiveness of interventions

Table 15 shows the numbers and percentages of parents, HPs and education professionals who reported interventions as effective (based on the respondents who had used them). The interventions most frequently rated as effective by parents of children with physical and mixed EDSD were Energy supplements (64%), Hand-over-hand prompting (64%), Modification of utensils (63%), Modification of environment (63%), Food or drink modification (63%), Medication (62%) and Pace of feeding at mealtimes (60%). For parents of children with non-physical EDSD, Sensorimotor therapy (100%), Sensory aids (64%), Energy supplements (61%) and Hand-over-hand prompting (59%) were the most frequently rated effective by those who had used them. The interventions that were least frequently rated as effective by parents who had used them were Sensory stimulation (35%), Schedule of meals (35%), Oral motor exercises (34%) and Strategies/programmes aimed at changing behaviour at mealtimes (30%). The majority of HPs reported that the interventions they used were effective (mean 99%, range 91–100%). Parents’ views showed greater variability and differed in accordance with the nature of the child’s difficulties (i.e. whether the child’s EDSD were because of physical and mixed difficulties or non-physical difficulties). Education professionals’ views also showed considerable variation. Overall, the interventions most frequently rated as effective by the education professionals who had used them were Modification of environment (90%), Strategies/programmes aimed at changing behaviour at mealtimes (89%), Food or drink modification (86%), Visual supports (85%), Pacing of food at mealtimes (83%), Positioning (83%), Training to wait for child’s cues for feeding (83%), Enhancing parent/child communication strategies at mealtimes (82%) and Hand-over-hand prompting (82%).

Time taken to produce change

Parents were asked about the duration over which they used each intervention. Parents reported using the majority of interventions for over 1 year. These interventions included using Information given about the child’s sensory (88%) and movement difficulties (78%), Modification of the environment (88%), Modifying social eating and drinking opportunities (86%), Sensory aids (84%) and Strategies to enhance communication between the child and the feeder at mealtimes (80%). In contrast, < 60% of parents reported using Scheduling of meals (58%), Oral motor exercises (56%), Sensory stimulation (48%) and Manoeuvres (44%) for over 1 year. Parents were not asked about the time taken for interventions to produce change, as it seemed likely that they would abandon interventions that they felt were not/no longer working.

Health professionals reported their perception that a large number of the interventions produced change quickly (0–3 months); these interventions included Manoeuvres (71%), Pacing of food at mealtimes (70%), Medication (66%), Modification of utensils (65%), Positioning (63%), Training to wait for a child’s cues for feeding (61%), Sensory aids (60%), Modification of environment (60%) and Food and drink modification (58%). For some of the interventions listed, such as Desensitisation programme for oral sensations, Oral motor exercises and Desensitisation programme for food avoidance, there was a relatively equal spread of responses from HPs across the different categories (0–3 months, 4–6 months, 7–9 months, 10–12 months and > 1 year), indicating that there was less consensus on how long these interventions took to produce change.

Education professionals agreed with HPs on most of the interventions that were thought to produce change quickly, including Modification of environment (76%), Modification of utensils (74%), Food and drink modification (67%), Medication (64%), Pacing of food at mealtimes (62%) and Training to wait for a child’s cues for feeding (59%). Education professionals also reported that Modifying social eating and drinking opportunities (62%), Energy supplements (60%) and Visual supports (60%) were quick to produce change (0–3 months). They also showed a spread of responses across the different categories (0–3 months, 4–6 months, 7–9 months, 10–12 months and > 1 year) for some of the interventions listed. These included Desensitisation programmes for oral sensations and food avoidance, Oral motor exercises, Sensory stimulation and Information about the impact of sensory or movement difficulties on a child’s EDSD, indicating there was less consensus among education professionals on how long these interventions took to produce change.

Potential benefits of interventions for eating, drinking and swallowing difficulties

Table 16 shows the numbers and percentages of parents, HPs and education professionals who viewed each potential benefit (referred to for remainder of report as ‘outcome’) as ‘important’. HPs, parents and education professionals all reported that the most important outcomes of interventions were Improved nutrition (parents, 40%; HPs, 31%; education professionals, 39%) and Better general health (parents, 31%; HPs, 32%; educational professionals, 48%). Parents also rated Weight gain (21%) and Increased growth (18%) as important, whereas HPs rated Fewer or shorter hospital admissions as important (17%). Outcomes related to the child’s and family’s overall well-being were also highly valued by professionals, including Better quality of life for the child (HPs, 26%; educational professionals, 36%), Less parental/carer stress (HPs, 17%), Child enjoying mealtimes more (education professionals, 23%) and Child being better able to communicate (education professionals, 19%).

Use of tools to measure outcomes

Health professionals showed great variation when asked about whether or not they formally measured outcomes, with 18% reporting that they ‘usually’ formally measured progress following intervention, 29% reporting they ‘sometimes’ did and 25% reporting that they ‘never’ did (28% respondents did not answer this question). The most commonly used outcome measurement tools were published measures that focused on the child’s body structure, functioning and activity, such as TOMs (13%), the Oral Motor Assessment Scale (3%) and the SOMA (3%). HPs also reported using goal-based outcome measures (8%); published functional classification systems to describe functional ability (5%), such as the EDACS and the Gross Motor Function Classification System (GMFCS); adapted or self-developed non-standardised measures (4%); anthropometric measures (3%), such as Weight, Height, BMI, Proportion of food and liquid taken orally versus by tube feeding and whether the child was meeting their nutritional requirements; and parent-related measures (2%), such as measures of Depression, Anxiety, Stress and parent feedback questionnaires.

Summary of national survey

The survey provided insights into current UK-based clinical practice to treat children with EDSD based on the experiences of parents of children with neurodisability who experience EDSD, HPs and education professionals. Questions that were addressed included which interventions are used, how acceptable interventions are to deliver, the perceived effectiveness of interventions, how and where interventions are delivered or implemented, and which outcomes are important. The survey found that a wide range of interventions were used by parents, HPs and education professionals in the management of EDSD in children with neurodisability, despite limited research evidence to demonstrate clinical effectiveness. The majority of the interventions recommended by HPs were used with children with physical, non-physical and mixed EDSD, although the way these are implemented may differ based on the individual needs of the child and family. All interventions were viewed as acceptable to deliver in home and in school. The frequency of use did not reflect views on effectiveness; some less frequently used interventions were viewed as effective by parents and professionals in managing EDSD, such as the use of Sensorimotor therapy with children with non-physical EDSD. The most highly valued outcomes included those relating to the child’s physical health alongside those relating to the child’s and family’s overall well-being. The survey also highlighted low levels of formal outcome measurement by HPs as part of their clinical practice.

A small number of the outcomes listed in the survey were not taken forward into the second round of focus groups because < 5% of respondents rated them as important. These were Less drooling, Fewer abnormal or unusual movements, the Child being able to communicate better, Less food waste or reduced cost of food and More opportunity for parents to talk to others about their feelings about their child’s EDSD.

Strengths and limitations of national survey

To maximise recruitment to the survey, a large number of relevant parent and professional networks were contacted via e-mail and social media. As a result, we do not know how many eligible people received an invitation and the opportunity to take part in the survey; therefore, we cannot calculate a denominator for the response or examine the extent of any potential response bias. Only a small sample of education professionals responded to the survey and within this sample only 10 worked specifically with children with non-physical EDSD, which makes it difficult to draw conclusions about the use of interventions within schools and the outcomes valued by education professionals. We also have limited information about the frame of reference used by professionals responding to the study, especially in relation to the child’s age, with a large number of respondents reporting that they worked across the age range 0–18 years. Professionals were required to share only the nature of the EDSD of the child with whom they worked (i.e. whether the children had physical, non-physical or mixed EDSD) and, therefore, we cannot further explore any differences there might be in how professionals work with children within these groups. Although the same interventions were used with children with physical, non-physical and mixed EDSD, the delivery and specific details/content of the individual intervention will be guided by the needs of the child and family. Therefore, the delivery and details of each intervention are likely to vary across individual children and groups. For example, the intervention ‘Modifying equipment’ could, depending on the individual child’s needs, mean using a spoon with a bigger handle to facilitate a better grip for self-feeding or using a plastic spoon because of a heightened bite reflex. Although the number of respondents to the survey was sufficiently large to address the aim of identifying the interventions used, the survey was not sufficiently powered to allow statistical analysis of subgroup data, such as how different types of professionals deliver the interventions. The time taken to produce change from the interventions was unclear for a number of the interventions, making it difficult to draw conclusions regarding the duration of interventions and outcome measurement points for future studies.

Patient and public involvement in national survey

Parents and HPs were consulted on the list of interventions and outcomes to be included within the questionnaire to ensure that it was as inclusive as possible, through a discussion group and the first round of focus groups. Their views were also sought on supplementary questions, including the acceptability and effectiveness of interventions and the time taken to achieve change. The parent co-investigators were also involved in designing the questionnaires and advised on the use of simple language and clear examples of interventions to maximise participants understanding. The final online questionnaire and paper versions were piloted by a small number of parents, HPs, education professionals and researchers. The PAG considered the summaries of the findings of the national survey alongside the findings from the updates of the three published systematic reviews of interventions (see Chapter 3) and the mapping review (see Chapter 4). They suggested some amendments to the name of the interventions to improve clarity and accuracy, which are outlined in Chapter 8.

How did the national survey inform the next steps?

Following our iterative process, the national survey findings were used to inform the topic guide for the second round of focus groups (see Chapter 9), the Delphi survey questions on interventions and outcomes (see Chapter 10) and the stakeholder consultation workshops (see Chapter 11). For example, the survey showed that there was great variability in the interventions recommended by HPs; therefore, it was important to gather further information at the stakeholder consultation workshops about what comprises treatment as usual and how services are configured to facilitate the design of future trials. The survey, alongside the mapping review (see Chapter 4) and the first round of focus groups (see Chapter 5), showed that multiple interventions were being used by parents and recommended by HPs for both children with physical and mixed EDSD and children with non-physical EDSD. This information was considered as part of the evidence synthesis (see Chapter 8) and informed the discussions about interventions at the second round of focus groups (see Chapter 9). A large number of outcomes were valued by parents, HPs and education professionals and, therefore, further information was sought through the second round of focus groups (see Chapter 9), Delphi survey (see Chapter 10) and stakeholder consultation workshops (see Chapter 11) building towards agreement on the key areas to measure within future studies/clinical trials. Respondents to the survey had provided contact details if they wished to take part in the Delphi survey (see Chapter 10) and, therefore, the national survey acted as a vital part of sampling frame construction and recruitment for later stages of the study.

Objectives

The objectives were to draw together information from preceding research activities on the support for individual EDSD interventions and their outcomes, and to inform a Delphi survey on the most important interventions and outcomes for future research evaluation.

Methods

Across the study, we collated evidence on the effects of EDSD interventions from the updates of three published systematic review of interventions (see Chapter 3), identified further interventions in a broader mapping review (see Chapter 4) and reviewed the measurement properties of published outcome measures (see Chapter 6). We also obtained the opinions of parents, HPs and education professionals on the current use and potential effectiveness of the EDSD interventions, and how they are evaluated in the UK, during the first round of focus groups (see Chapter 5) and from a national survey (see Chapter 7).

The previous chapters of this report have described the methods that we used in the individual activities and their results. Given our overall aim to identify interventions that could be delivered at home by parents to improve eating, drinking and swallowing in young children with neurodisability, we took an iterative, additive approach to synthesise/collate data regarding interventions and outcomes. To create full lists of all interventions and outcomes that have support from either research and/or current practice, we charted the data from each of the individual activities. We added the information obtained from each data set to the next data set, and iteratively created a full picture of potential interventions and outcomes. Each individual activity/data set was given equal weighting, as our aim was to develop a full picture rather than assess effectiveness.

Results of the evidence synthesis

Tables 17 and 18 show the interventions and outcomes that were identified and supported in the individual research activities.

We reviewed the interventions and outcomes identified through the updates of the three published systematic reviews of interventions (see Chapter 3), the mapping review (see Chapter 4), the first round of focus groups (see Chapter 5) and the national survey (see Chapter 7). Given that individual interventions were rarely used on their own, the research team decided that the individual interventions were usually delivered in series or in combination, depending on the needs of the child and parent. We conceptualised this as a ‘toolkit of interventions’, and compiled a separate version for children with physical or mixed EDSD and children with non-physical EDSD (Figures 9 and 10), including the associated outcomes.

FIGURE 9.

Visual summary of the interventions and outcomes for children with physical or mixed EDSD from the evidence review and national survey.

FIGURE 10.

Visual summary of the interventions and outcomes for children with non-physical EDSD from the evidence review and national survey. CBT, cognitive–behavioural therapy.

When compiling the first versions of the toolkit, the following decisions were taken by the research team on the basis of the evidence synthesis and feedback from the PAG.

Strengths and limitations of the evidence synthesis

The sequential approach to gathering data on interventions and outcomes that are supported by research and/or current practice in the UK enabled us to build on a robust foundation underpinned by the relevant evidence. We have been pragmatic and forward focused; therefore, we have not revisited or updated each stage of the work beyond the point at which it was used. For example, we did not revisit and update the search for the mapping review; although it is feasible that we would have identified new studies, findings could not have been incorporated into our survey of current practice or consensus seeking in the Delphi survey. Owing to the pragmatic nature of the work, we searched for and included only papers that were published in the English language.

How did the evidence synthesis inform the next step?

The toolkit of interventions and associated outcomes was shown to parents and HPs in the second round of focus groups (see Chapter 9) to capture their views on the idea of a toolkit and to identify any additional interventions or outcomes that were not found through the research activities to date.

Objectives

We aimed to gain an understanding of parents’ and HPs’ views on which interventions identified in the previous study stages should be tested in research, on which outcomes identified in previous stages are important to measure and on which measurement tools could be used.

We also wanted to explore parents’ and HPs’ views on the future research priorities for interventions and choosing appropriate outcome measures for the interventions researched.

Methods

Results

Nine focus groups were held in September and October 2018: four in Newcastle (two for HPs and two for parents), two in London (for HPs), one in Sussex (for HPs) and two in Exeter (for parents). Three parents due to take part in a focus group in Exeter were unable to do so because of family circumstances. Subsequently, these parents continued to express a desire to participate and were interviewed individually by telephone.

Summary of the second focus groups

The focus groups highlighted that all interventions and outcomes identified in previous stages of the research should be taken forward into the Delphi survey (see Chapter 10). Multiple interventions are often provided concurrently or sequentially, but there are several issues affecting their implementation. There is a need to build trust between parents and HPs for shared decision-making. There is significant variation in service organisation and available personnel, and the skills and experience available affect what is offered to children and their families. ‘Treatment as usual’ varies widely across the UK. A consistent feature that was highlighted by all groups was the general lack of psychology input for children with non-physical EDSD.

Participants universally agreed that further research is required, but the complexities of measuring outcomes, both clinically and for research, were acknowledged.

Strengths and limitations of the second round of focus groups

The focus groups were held in multiple UK locations, which reduced the chance of bias from regional practice opinion. However, as participants volunteered to take part, they may have had particularly strong views about certain issues discussed that were not representative of other parents and HPs. Where parents were unable to attend, their views were included through interviews. Terms such as ‘stress’ and ‘anxiety’ were not well defined, and there was not an opportunity to explore this in detail during groups in which a range of topics were discussed. Analysis was challenging owing to the wide range of topics discussed, as well as variation in opinion, practice and expertise. Themes contained useful views of relevance to future research.

Patient and public involvement in the second round of focus groups

The PAG advised on how best to share information about the interventions and outcomes that were identified through the updates of the three published systematic reviews of interventions (see Chapter 3), mapping review (see Chapter 4) and national survey (see Chapter 7). They advised on creating a pictorial summary of the interventions and outcomes to aid discussion in the groups, and using the term ‘strategies’ to describe the interventions (see Figures 9 and 10). Given the overlap between interventions identified for physical and mixed EDSD and for non-physical EDSD, they advised that it might be beneficial to run groups including parents of children with physical, mixed or non-physical EDSD together to capture the commonalities and differences within and between these groups.

How did the second round of focus groups inform the next steps?

The focus groups highlighted that all interventions and outcomes identified in previous stages of the research should be taken forward into the Delphi survey (see Chapter 10).

Presentation of the interventions in the visual format (see Figures 9 and 10) validated the research team’s concept of a single toolkit for any child with neurodisability and EDSD, which was made up of individual interventions to be selected in tandem or sequentially by parents and HPs. This concept evolved further at co-investigator meetings and was discussed at the stakeholder workshops (see Chapter 11), in which the themes about future research were also discussed.

The research team further reviewed the interventions and outcomes included in the proposed toolkit following the second round of focus groups in developing the Delphi survey. The PAG also offered further advice regarding the terms used within the toolkit, which resulted in a number of changes.

Changes to interventions

For ease of understanding, we renamed the following interventions based on feedback from the second round of focus groups and the PAG:

• Modification of utensils changed to Modifying equipment.

• Reading child’s feeding cues changed to Responding to a child’s cues for feeding.

• Raising awareness changed to Sharing information.

Following feedback, for accuracy and clarity we renamed the following interventions to ensure that they were more clearly relevant to parents of children with physical or mixed EDSD and non-physical EDSD:

• Food and drink modification into Modifying the consistency of food or drink and Modifying other aspects of the food.

• Desensitisation programme for food avoidance into Graded exposure to new textures and Graded exposure to new food.

In response to feedback, we added the following intervention:

• Vitamin or nutritional supplements.

Changes to outcomes

We renamed the following outcome following feedback from the PAG and the views of the second focus groups and research team:

• Less parental/carer stress changed to Mental health of parent/caregiver.

In response to feedback, the following outcome was added:

• Efficiency (to incorporate Duration of mealtimes).

One outcome was not taken forward, as it was not considered specific or measurable:

• Prevention of further difficulties.

One further outcome was not taken forward, as we considered it had been covered by an outcome already included:

• Vomiting was covered by General health.

A table describing all of the original intervention and outcome terms used in the national survey and the revised terms used in the Delphi survey is included in Appendix 13.

Objectives

The objectives were to seek agreement between parents of children with neurodisability and HPs on which interventions and which outcomes are considered to be the most appropriate for young people with neurodisability and EDSD.

Methods

Study design

As the aim was to establish consensus, an iterative online Delphi survey was employed. A questionnaire was sent to parents and HPs in two rounds; parents and HPs received the same questionnaire in each round. Both survey rounds were open for 3 weeks, with a week between the two rounds to allow for data analysis. In the first round, respondents were asked to rate the importance of individual intervention categories and outcomes for children with neurodisability who have EDSD. In the second round, respondents were shown the results of the first round in the form of bar charts of both parent and HP ratings of the importance of each intervention to be included as part of a package for children with EDSD, and the importance of each outcome. Respondents were then asked to re-rate the importance of each intervention and outcome in the light of the results from round 1. No items were removed from the survey between round 1 and round 2.

As an incentive, respondents were informed that at the end of the second round of the Delphi survey they would be entered into a prize draw to win one of five £100 vouchers available for each stakeholder group (parents and HPs).

Questionnaire development

The round 1 questionnaire was developed by listing the interventions and outcomes identified from the synthesis of evidence gathered through the systematic and mapping reviews (see Chapters 3 and 4, respectively), the national survey (see Chapter 7) and the second round of focus groups (see Chapter 9). The research team developed the structure and format of the questionnaire, drawing on previous experience of Delphi surveys and with reference to methodological recommendations. 102

The questionnaire comprised (1) demographic characteristics, (2) questions about outcomes that could be used to measure progress in eating, drinking and swallowing and whether or not interventions were effective, and (3) questions about the inclusion of specific interventions that could be delivered at home by parents of young children with neurodisability and EDSD. The majority of questions used fixed-choice rating options, although participants could suggest any additional outcomes or interventions not listed and/or comment through free-text boxes. Respondents were invited to rate how important they thought it was to include each of the 25 discrete interventions as part of a package of treatment for EDSD and their perception of how important they thought each of the 22 outcomes were. Ratings were made on a 9-point scale in which categories were labelled 0–3, ‘not important’; 4–6, ‘important but not essential’; and 7–9, ‘essential’. Respondents were able to tick ‘unable to score’ if they were not able to comment based on their knowledge or experience, or if the item was not relevant or applicable to them. Electronic, web-accessible versions were hosted by Newcastle University using Qualtrics (Provo, UT, USA).

The PAG reviewed the draft survey documents and offered advice about wording and layout. The final online questionnaire was piloted by a small number of parents, HPs and researchers. Respondents were able to use a ‘back’ button to review or change their answers as required and a progress bar told them how far through the questionnaire they were (see Report Supplementary Material 2 for the first round and Report Supplementary Material 3 for the second round).

Procedure

Parents and HPs who took part in the national survey (see Chapter 7) and who had expressed an interest in completing another survey were contacted via e-mail with an invitation to take part in the Delphi survey, and a hyperlink to the online questionnaire. To maximise participation, both respondents and non-respondents from round 1 were invited to take part in round 2.

Analysis

Consensus was conservatively defined as ≥ 67% of both stakeholder groups separately rating that it was essential (rated 7–9 at round 2) to assess an outcome domain or to incorporate intervention in the treatment package. 102

Results

Response

Invitations to participate were sent to 196 parents and 175 HPs (Figure 11). Of these responses, in each round there were:

• parents –

  • round 1: n = 81/196 (41%)

  • round 2: n = 61/196 (31%)

  • with 52 parents taking part in both rounds.

• HPs –

  • round 1: n = 76/175 (43%)

  • round 2: n = 61/175 (35%)

  • with 51 HPs taking part in both rounds.

FIGURE 11.

Flow diagram of Delphi survey recruitment.

The round 1 HPs comprised:

• speech and language therapists (n = 29)

• dietitians (n = 5)

• occupational therapists (n = 20)

• physiotherapists (n = 5)

• clinical psychologist (n = 1)

• paediatricians (n = 12)

• gastroenterologist (n = 1)

• nurses (n = 2)

• other (n = 1).

The round 2 HPs comprised:

• speech and language therapists (n = 28)

• dietitians (n = 3)

• occupational therapists (n = 14)

• physiotherapists (n = 2)

• clinical psychologist (n = 1)

• paediatricians (n = 10)

• gastroenterologist (n = 1)

• nurses (n = 2).

Sample characteristics

Table 20 shows the characteristics of the respondents who completed the first and second rounds of the survey, and the non-respondents. In the first round, the respondents comprised a similar proportion of parents and HPs (49% and 51%, respectively). Respondents were predominantly from England. The majority of respondents were aged 41–50 years (49% of parents; 33% of HPs) and most respondents were female (94% of parents; 92% of HPs). The majority of respondents identified as white British (96% of parents; 92% of HPs). Half of the parents had a child with non-physical EDSD (49%) and half had a child with mixed or physical EDSD (51%). The majority of HPs worked with children with mixed EDSD (75%).

In the second round, a similar proportion overall of parents and HPs responded (50% and 50%, respectively). Respondents were predominantly from England. The majority of parents were aged 41–50 years (53%) and the majority of HPs were aged 51–60 years (34%). The majority of respondents were female (95% of parents; 94% of HPs) and most identified as white British (97% of parents; 90% of HPs). Around half of the parents had a child with non-physical EDSD (52%) and half had a child with mixed or physical EDSD (48%). Most HPs worked with children with mixed EDSD (74%).

The characteristics of respondents who completed both rounds of the Delphi survey and those who completed the second round only were very similar apart from age, for which the majority of those completing both rounds were aged 41–50 years (46%), whereas the majority of those completing the second round only were aged 31–40 years (63%). See Appendix 14 for full details of the characteristics of respondents who completed both rounds of the Delphi survey and those who completed round 2 only.

Interventions

Table 21 shows the proportion of parents and HPs who rated interventions as essential in the first and second round of the Delphi survey. The percentage of respondents giving a rating of ‘essential’ (score of 7–9) increased from round 1 to round 2 for most interventions, for both parents and HPs. Consensus was achieved for 17 of the 25 interventions at round 1, increasing to 19 out of the 25 interventions at round 2. There was substantial agreement between the ratings of participants who completed both rounds of the survey and the ratings of participants who completed round 2 only (data not shown).

Figure 12 shows the interventions that were viewed as essential by ≥ 67% of parents and HPs in the second round of the Delphi survey (see Appendix 15 for the interventions viewed as essential by ≥ 67% of parents and HPs in the first round). In the second round, parents and HPs identified that a large number of interventions (n = 19) were an ‘essential’ part of an intervention package for young children with neurodisability and EDSD. These comprised interventions directly targeting EDSD, such as Modifications (Environment, Equipment, Food or drink, Placement of food and Positioning), improved mealtime communication (Enhancing communication strategies, Responding to the child’s cues for feeding and Pace of feeding) and desensitisation strategies (Graded exposure to foods or textures and Oral and sensory desensitisation). Other interventions not specifically targeting the child’s EDSD were also viewed as essential, such as Sharing information about the nature of the child’s difficulties, Medication and Parental support. None of the 25 interventions was viewed as ‘not important’ to include in an intervention package by either parents or HPs. See Appendices 16 and 17 for ratings of ‘not important’, ‘important but not essential’ and ‘essential’ for interventions by parents and HPs in the first and second rounds of the Delphi survey.

FIGURE 12.

Interventions viewed as essential by ≥ 67% of parents and HPs in round 2 of the Delphi survey.

Outcomes

Table 22 shows parents’ and HPs’ agreement on outcomes rated as essential in the first and second rounds of the Delphi survey. In keeping with the findings about interventions, the percentage of respondents rating the outcome as ‘essential’ typically increased between the first and the second rounds. Figure 13 shows the outcomes viewed as essential by ≥ 67% of both parents and HPs in the first and second rounds of the Delphi survey. These are presented together given that the items for which there was consensus did not change between rounds. The 10 outcomes viewed as essential included child-focused outcomes, such as Physical health, Safety, Oral motor control and Quality of life. They also included family-focused outcomes, such as Quality of life of family, Parents’ understanding of the child’s difficulties and Parents’ mental health. None of the 22 outcomes was viewed as ‘not important’ by parents or HPs. See Appendices 18 and 19 for ratings of ‘not important’, ‘important but not essential’ and ‘essential’ on outcomes by parents and HPs in the first and second rounds of the Delphi survey.

FIGURE 13.

Outcomes viewed as essential by over 67% of parents and HPs in round 2 of the Delphi survey.

Summary of the Delphi survey findings

The Delphi survey established consensus between parents and HPs on the ‘essential’ interventions to include in an intervention package for young children with neurodisability and EDSD, and the important outcomes. The Delphi survey showed that for both parents and HPs 19 interventions were viewed as ‘essential’ to include in an intervention package and 10 outcomes were viewed as ‘essential’; these focused on both the child and the wider family. Schedule of meals and Visual supports were viewed as ‘essential’ interventions for inclusion in an intervention package by HPs only, and Oral motor exercises and Psychological support for children were viewed as ‘essential’ interventions to include by parents only. Training to self-feed and Changing behaviour at mealtimes were not viewed as ‘essential’ to include in an intervention package. None of the interventions or outcomes included in the Delphi survey was viewed as ‘not important’ to include in an intervention package, reflecting the complexity of neurodisability and highlighting the need to tailor interventions to meet the needs of individual children and their families.

Strengths and limitations of the Delphi survey

We used a wide range of recruitment sources that were also used in the national survey (see Chapter 7), and a reasonable number of respondents to that survey showed willingness to contribute further. This meant that we had information to enable a comparison of characteristics of those who participated and those who did not, and, therefore, the Delphi survey invitation was sent only to previous survey respondents. The overall response for the surveys was typical and acceptable (≈ 40%) for the Delphi approach, and there was little difference in the characteristics of respondents between round 1 and round 2 (see Appendix 14). The response ratings about ‘essential’ interventions and outcomes were very high; therefore, a higher response would have been unlikely to lead to different results. Our consensus definition of 67% was conservative; however, most interventions and outcomes were rated ‘essential’ by a much higher proportion than this. Nonetheless, if we had used a higher percentage to define consensus, our findings would have differed slightly. Contacting non-respondents from round 1 in round 2 increased the responses in round 2 and, therefore, improved precision; although this is not always carried out in Delphi surveys, there was substantial agreement between the ratings from participants who completed both rounds of the survey and those who completed round 2 only.

Patient and public involvement in the Delphi survey

The Delphi survey questionnaires and information sheets were developed by the research team, including the parent co-investigators. These draft documents were reviewed by the PAG who offered advice around content, wording and layout. These documents were further amended to take account of the discussions in the second round of focus groups and these amended versions were reviewed again by the PAG. The PAG advised on the ordering of statements relating to interventions and outcomes for the Delphi survey to ensure that these were presented in an order that made sense to the reader.

How did the Delphi survey findings inform the next study stage?

The Delphi survey findings informed decisions around which interventions and outcomes to take forward for further discussion at the stakeholder consultation workshops (see Chapter 11), and for consideration when designing future research and pragmatic trials.

The Delphi survey identified that a large number of interventions were viewed as essential to consider for improving EDSD in young children with neurodisability. The research team used this information and that gathered previously to make pragmatic decisions about which interventions to include in the toolkit so that they could be selected for use depending on the child and family’s individual needs.

The Delphi survey also showed that a large number of outcomes were valued by parents and HPs, focusing both on the child and the wider family. These essential outcomes were presented at the stakeholder consultation workshops (see Chapter 11) to facilitate further discussion regarding the important primary and secondary outcomes for use in future research and trials.

Objectives

The objectives were to gauge consensus between HPs and parents of children with neurodisability on the interventions for EDSD, and their outcomes, that should be evaluated in future research, and to agree on frameworks for such research in the NHS.

Methods

Data processing

Flip chart feedback compiled during the workshop discussions and the notes made at each group formed the data for analysis. Morag Andrew and Lindsay Pennington reviewed all data, identified key themes and presented findings to the full research team for discussion.

Results

A total of 15 parents and 19 HPs took part in the workshops. Nine of the parents had children with physical EDSD, two had children with non-physical EDSD, two had children with mixed EDSD and two had one child with physical EDSD and one child with non-physical EDSD. HP participants comprised speech and language therapists (n = 5), dietitians (n = 4), paediatricians (n = 4), occupational therapists (n = 3), clinical psychologists (n = 2) and a physiotherapist. One member of the NIHR-appointed external steering committee (speech and language therapist) participated in the Newcastle workshop and one (nurse) in the London workshop.

Use of eating, drinking and swallowing difficulty interventions in current clinical practice

Participants reported that multiple EDSD interventions are used in current clinical practice, but in an unstructured and unco-ordinated manner.

Toolkit of interventions

Participants agreed that no single intervention would be suitable for all children with EDSD; for many children several interventions may be delivered concurrently or sequentially. Participants liked the concept of an intervention ‘toolkit’ that parents and HPs could use together to identify the most appropriate interventions for individual children and their families.

Participants agreed that the toolkit should be represented visually and available in digital and hard copy, with interactive properties to aid communication with HPs. They thought that it should be flexible to allow families and teams to individualise intervention selection. Parents thought that some parents would want to have ownership of the toolkit and to be integral to decisions on which interventions were selected for their child. In that context, parents and HPs agreed that thorough information on each available intervention would allow families to share decision-making with HPs.

Support for families using the toolkit

Health professionals thought that toolkit use should be supported by a lead HP (e.g. a speech and language therapist) and MDT. Professional support would be required throughout toolkit use and may include psychological input; however, the nature of the support that was required would vary between families.

Participants raised several issues about the delivery of an intervention toolkit:

• how to deliver the toolkit to meet the needs of a heterogeneous population with diverse EDSD

• how to deliver the toolkit in geographical areas in which multidisciplinary EDSD teams are unavailable or under-resourced

• how to avoid/overcome problems caused by delays in obtaining appropriate equipment (e.g. optimising positioning for feeding) to ensure efficient use of the toolkit

• how best to deliver the toolkit for children with non-physical EDSD who are not currently linked to HP teams.

Ways to measure outcomes

The focus group and Delphi survey stages identified the important outcomes of any EDSD intervention (see Chapters 9 and 10). These outcomes were organised into three categories for presentation at the workshops:

1. category 1 – improvement in physical aspects of a child’s EDSD

   • Safety

   • General health

   • Nutrition

   • Growth

   • Oral motor control

2. category 2 – changes in behaviour of child or parent

   • Child’s behaviour at mealtimes

   • Parental understanding of child’s eating and drinking difficulties

3. category 3 – changes in child or family’s well-being

   • Quality of life of child

   • Quality of life of family

   • Parental mental health

   • Participation.

The groups discussed potential measures for each EDSD outcome and their strengths and weaknesses in clinical practice. For implementation in research, measures must be acceptable, valid and reliable. Not all of the measures that were suggested by participants fit all three of these criteria. A full list of measures generated in the stakeholder consultation workshop is provided in Appendix 20.

No acceptable measures that have evidence of reliability and validity were identified for Safety (measures discussed were videofluoroscopy, frequency of chest infections, frequency of choking episodes and observation of meals) or General health (use of a structured questionnaire capturing general health information, school attendance, bowel function including medications needed, skin pallor, hair thickness, dental health, hospital admissions or outpatient attendances, energy levels, concentration and sleep). For Nutrition, participants suggested 3-day food diaries; measuring blood micronutrient levels; food frequency questionnaires; body fat percentage using smart scales and the The Eatwell Guide [Public Health England, UK, URL: www.gov.uk/government/publications/the-eatwell-guide (accessed 23 December 2020)] (see Appendix 20); photographs of food at the beginning and end of a meal, including photographs of the floor to assess spillage; and the need for dietary supplements. Participants identified several measures of Growth including skinfold thickness, bioelectrical impedance, weight and height, and upper arm circumference (see Appendix 20 for further information). Several of the measures suggested for Growth and Nutrition have evidence of reliability and validity, but each would require further investigation in terms of their acceptability and validity in children with neurodisability.

As these outcomes had not been included in the measurement properties review, we undertook a brief review of their properties (Box 6).

For Oral motor control, participants identified the SOMA, which was included in the measurement properties review.

The measurement properties review showed that the PediEAT and the BPFAS had evidence of validity and reliability as measures of the Child’s enjoyment of meals and Parents’ understanding of child’s eating and drinking difficulties. In the consultation workshop, groups of parents considered these two measures in detail. Parents liked that the PediEAT has clear, specific questions, which feel quick to answer, and that the wording is factual and does not feel judgemental. The tool was deemed likely to identify the main issues and provide sufficient detail to help inform joint goal setting and identify comorbidities. The front page was considered off-putting owing to its length, as it included an outline of intended use, disclaimers and references relevant to the tool. However, the questionnaire was judged as lengthy for use during clinic appointments and better completed during a separate session to enable collection of the most detailed information. Although it is not a measure of participation, parents thought that some items were relevant to participation.

Parents liked the specificity of the BPFAS, but disliked its length. They felt that parents would need an explanation of why it was being used and why it was so long, so that they felt it would be worth the time taken to complete. However, they thought that it would be useful to consider results taken at different time points to review progress using this measure.

Participants did not identify measures of Participation, Quality of life of children or family members or Mental Health; however, generic measures have been validated, such as the CAPE117 and KIDSCREEN,118 that measure children’s Participation and Quality of life, respectively.

Designing future research (including randomisation and recruitment selection)

Potential barriers to recruitment

Summary

The stakeholder consultation workshops showed consistent support from parents and HPs for the concept of a toolkit of interventions that could be worked through by HPs and parents. There was debate about how a toolkit might be best presented. The parents and HPs in the consultation workshops agreed with most of the outcomes that were deemed essential by the Delphi survey participants: Safety, General health, Nutrition, Oral motor control, Growth, Children’s enjoyment of mealtimes (incorporating reduced frustration and distress and associated behaviours), Parental understanding of child’s eating and drinking difficulties, Quality of life of child, Quality of life of family, Parental mental health and Children’s social participation. Ways of measuring most of these outcomes were proposed. Parents also endorsed the Pedi-EAT tool, which was found to have the strongest measurement properties (see Chapter 6). Parents and HPs felt that a trial of a toolkit could be achieved and supported a cluster design in which services were allocated to implement the toolkit or treatment as usual. However, treatment as usual would need defining in comparator group services, as this varies considerably. Participants also identified challenges to the identification and recruitment of participants for future NHS research, including increased referrals to services if the trial was advertised within trusts.

Patient and public involvement in the stakeholder consultation workshops

The parent co-investigators were involved in designing and delivering the stakeholder consultation workshops. The PAG reviewed the materials to be shown to parents and professionals, and commented on the structure of the workshops and the timings of the individual tasks. Changes to the layout and wording were subsequently made to the slide presentation summarising the study and key findings, and a document was created to send out to all attendees prior to the workshops to help provide a background to the study and clarity on what would happen on the day.

Strengths and limitations of the stakeholder consultation workshops

Consultation workshop strengths include participation of both parents and HPs across two diverse geographical areas. Parent/carers of young people with physical, non-physical and mixed EDSD participated, representing a broad range of EDSD experiences. Parent/carers had accessed secondary or secondary and tertiary-level EDSD services. Multidisciplinary professional representation was achieved during both workshops, with participation from HPs working in secondary and tertiary feeding services. The iterative nature of the workshops facilitated collection of detailed information on the topics discussed. We decided not to include young people with EDSD in the consultation workshops because of the abstract nature for older respondents of interventions for young children. This could be considered a limitation of the workshops; however, young people gave their opinion about outcomes during separate young people’s focus groups.

How did the stakeholder consultation workshops inform the next study stage?

The opinions of young people with EDSD on the outcomes identified as important to parents and HPs during the two stakeholder consultation workshops were sought during two young people’s focus groups (see Chapter 12).

Output from the stakeholder consultation workshops was used to generate recommendations, specifically those concerning the further development of an intervention toolkit, outcome measures and the design of future research to establish the effectiveness of an EDSD intervention toolkit.

Objective

The objective was to establish which of the outcomes of interventions for EDSD that were identified as important by parents and professionals were most meaningful to young people with neurodisability.

Methods

Two focus groups with young people with neurodisability who had EDSD, or had EDSD previously, were conducted. There was one focus group for young people with physical or mixed EDSD and one focus group for young people with non-physical EDSD.

Participant recruitment and selection

For focus group 1, seven young people with physical EDSD who were cognitively able to participate in the focus group were identified by a senior member of their local speech and language therapy team. Invitation letters were sent out to parents, followed by a telephone call to non-respondent parents to ascertain their willingness to participate. A familiar communication partner for each young person was also invited to attend the group.

For focus group 2, the focus group was advertised via a social group for young people with autism. Individuals who were aged 12–16 years with non-physical EDSD were invited to attend. The leader of the social group confirmed that participants were cognitively able to participate in the focus group. Each young person was accompanied by a parent.

Parental informed consent and participant assent was obtained for participants aged < 16 years. Informed consent was obtained from participants aged ≥ 16 years.

Procedure

The procedure for both focus groups was identical. One member of the research team (MA) led a 1-hour workshop in a room familiar to the participants. She presented images representing the outcomes identified as essential in the Delphi survey, printed on A4 paper. Figure 14 shows an example.

FIGURE 14.

Growth outcome image.

Morag Andrew presented two practice examples (friends and screen time) before the proposed outcome images; young people confirmed that they understood the process. Along with each of the outcome images, Morag Andrew provided an explanation of each of the nine outcomes (outcomes were described rather than named); Parent mental health was not considered appropriate to be discussed. One outcome at a time, Morag Andrew gave individual participants A5 images that matched each A4 outcome image. She placed three large A1 poster sheets headed ‘Very important’, ‘In the middle’ and ‘Less important’ on the wall in clear view of the participants. Morag Andrew repeated the description of the outcome, then asked the participants, in turn, to indicate whether they considered the outcome ‘Very important’, ‘In the middle’ or ‘Less important’. Morag Andrew presented the outcomes in the following order, using the given descriptor:

• Growth – ‘a change in a child’s growth, including how much they weigh/how heavy they are and how tall they are’.

• Health – ‘children are more healthy, feel ill less often’.

• Safety – ‘children are able to eat and drink without choking or food going down the wrong way’.

• Nutrition – ‘making sure that the foods children eat can give them enough energy and vitamins so that children can grow and stay healthy’.

• Oral motor control – ‘children are able to control the movement of the mouth, jaw, tongue or lips, and swallow’.

• Parental understanding of EDSD – ‘parents’ understanding of why eating and drinking may be hard for their child’.

• Enjoyment of mealtimes – ‘child’s enjoyment of mealtimes at home and school’.

• Quality of life of young person – ‘how happy a child is with their life’.

• Quality of life of family – ‘how happy other members of the family are with their lives’.

Once each outcome had been assigned to an importance category, Morag Andrew took the poster sheets down and replaced them with new poster sheets entitled ‘Most important’ and ‘Very important’. Morag Andrew displayed all of the outcomes previously identified by participants as ‘Very important’ on the relevant poster sheet. She then asked participants to consider which of the ‘Very important’ outcomes were ‘Most important’. Morag Andrew moved the A5 images of the outcomes selected as ‘Most important’ to the ‘Most important’ poster sheet. She audio-recorded the focus groups and photographed the poster sheets so that the information collected could be checked or verified. Participants received a £50 shopping voucher as a thank you for their contribution.

Results

In focus group 1, four female participants aged 14, 15, 16 and 18 years agreed to participate. Three of the participants used Voice Output Communication Aid communication systems and one participant used verbal communication. All of the participants were supported by a familiar communication partner. Three of the participants regularly participated in small group work together. All participants had physical EDSD; none had mixed EDSD. All four young people had EDSD and fed orally; two participants had enteral feeding tubes to support intake but maintained partial oral feeding. All participants had current EDSD.

In focus group 2, six young people with autism and non-physical EDSD agreed to take part: four males and two females aged 12–15 years. Two participants also had selective mutism and gave their responses by writing on individual white boards. All young people had or previously had non-physical EDSD; none had mixed EDSD. All participants fed orally, without enteral feeding tube support. All participants were recruited on the criterion of recent or current EDSD, but we did not receive information on the current EDSD status.

Table 23 shows how young people from the focus groups rated outcome importance.

Young people with physical EDSD unanimously agreed that Safety was the ‘Most important’ outcome, closely followed by Nutrition. Participants also selected Oral motor control, Quality of life of young person and Health as being additional ‘Most important’ outcomes, in the order given. One young person with non-physical EDSD chose not to rate the most important outcome. The remaining five young people with non-physical EDSD selected Quality of life of young person (n = 2), Safety (n = 2) and Health (n = 1) as the ‘Most important’ outcomes.

Summary of the young people’s focus groups

For young people with physical EDSD, Safety and Nutrition were the most important outcomes. For young people with non-physical EDSD, Quality of life of the young person, Safety and Health were the most important outcomes.

Strengths and limitations of the young people’s focus groups

The inclusion of young people with physical and non-physical EDSD was a strength of the focus groups. Insufficient time to fully explore the rationale driving participant outcome selection was a limitation. The age range of the two focus groups differed slightly: participants in focus group 1 were slightly older than participants in focus group 2, which may have affected participant responses. Focus group 1 comprised females only; sex may have contributed to the responses obtained. The young people’s focus groups were all held in north-east England; additional data from focus groups in other regions of England would be valuable to establish any differences of opinion of young people living in socially and economically diverse areas of the country.

Patient and public involvement in young people’s focus groups

The parent co-investigators helped liaise with schools and local community support groups to recruit the young people.

How did the young people’s focus groups inform the next study stage?

The ratings of the importance of outcomes from the young people’s focus groups were considered by the research team alongside the information gathered in the stakeholder consultation workshops (see Chapter 11), and informed the recommendations of future research and trial design.

Outcomes and measurement

We identified substantial agreement between parents and professionals in relation to the most important outcomes that should be measured in trials; young people also agreed with many of these. We do not think that there is a clear outcome area or measurement tool that could be currently recommended for use as a singular primary outcome. The ‘medical outcomes’, such as Safety and Growth, and those relating to the International Classification of Functioning Disability and Health, such as Social participation of the child, were widely endorsed and could be measured in future trials. 12

Families of children with neurodisability and the HPs who work with them seek interventions that are effective. They require evidence from research to inform decisions about which treatments to choose to invest in. As with most areas of health care, ‘evidence’ about effective interventions for children with neurodisability has emerged largely from clinical practice rather than from high-quality research. The James Lind Alliance Priority Setting Partnership conceptualised and funded by the British Academy of Childhood Disability11 identified priority areas that require higher-quality evidence; this influenced the commissioning of the FEEDS study. Our findings about provision, interventions and outcomes are directly relevant to current clinical care, and may influence current UK and international clinical practice and UK NICE guidelines. 6,9,10

Strengths of our study

The strengths of our component studies have been described in their individual chapters and are not repeated here. We consider that the main strengths were as follows.

The project used an iterative research process, with each phase of the work informing subsequent data collection and interpretation. We were able to use the findings from recently published systematic reviews of interventions to guide our research. 4–6 The published systematic reviews of interventions and the updates we undertook, and the mapping and measurement properties reviews, comprehensively identified the evidence base and found how limited the evidence is.

We recruited > 900 parents, young people and health and education professionals from across the UK. This meant that a wide range of views were identified from people with lived experience and expertise in physical and non-physical EDSD.

The focus groups, workshops and discussions with young people all gave face validity to the findings from the survey stages and overall results, and participants in those elements of the research expressed strikingly similar views about the direction for clinical development and future research. Thus, the results are likely to be widely applicable beyond the UK in countries with similar health service resources.

Limitations of our study

The limitations of our study components have been described in the individual chapters and are not repeated here. We consider that the main broader limitations were as follows.

Participants were not representative of the UK population as a whole. Evidence from children with disorders causing progressive neurodisability were not included in the study. Most study participants were female, white British and from England. The views of parents from an ethnic minority and the views of fathers were not well represented. We did not investigate the socioeconomic status of participants.

Despite the project being about young children, few parents of children aged < 5 years participated, with most being parents of children of primary school age. Recall bias may, therefore, have distorted some parents’ views, as expressed in the surveys and focus groups. On the other hand, the lived experience throughout their child’s early life lends validity to parental perspectives. Owing to the nature of the sampling frame for the national survey, it was not possible to investigate the extent or nature of non-response bias.

Conclusions

Parents and HPs reached consensus on 19 interventions and 10 outcomes as being ‘essential’. Across all strands of the research, we established that no single, standalone intervention is likely to be appropriate, effective or acceptable to parents and professionals in supporting children with neurodisability and EDSD. Therefore, evaluation of a specific intervention as a discrete entity in a RCT with a large sample of children with neurodisability is unlikely to be useful. Multiple interventions need to be used in combination, taking into account the underlying causes of the child’s EDSD, their individual needs and intervention goals. HPs and parents appear enthusiastic about the idea of the proposed FEEDS toolkit of interventions, which professionals could use in partnership with parents to identify and agree priority areas to address for a particular EDSD and to tailor the choice of interventions. Both stakeholder groups made useful suggestions for the development of the toolkit, including creation of a web-based version that could become part of the clinical notes and have interactive elements to facilitate recording. We believe that development and optimisation of the FEEDS toolkit is a prerequisite to any future deployment and evaluation thereof in, for example, pragmatic trials. This development work should be operationalised as a complex intervention, taking account of constituent content, delivery strategies considering fidelity of delivery and acceptability, sustainability of implementation and manualisation. Use of a toolkit approach in clinical practice needs to be informed by theories and models of behaviour change. A possible barrier to delivery of a novel intervention (and indeed standard care) is limited therapist and clinical psychologist capacity in terms of both the staff–child ratio and the skill base of professionals.

Our findings suggest that conducting a RCT at this stage may be challenging. We conclude that a development study should first be undertaken in which feasibility and acceptability of the toolkit, and primary and secondary outcomes and their measures, are investigated further. Subsequently, a clinical implementation study or RCT would be appropriate and achievable and lead to rigorous evaluation of the effectiveness of the toolkit.

Recommendations

• Future research should evaluate whether or not a combination of interventions can be delivered effectively (sequentially or in parallel). Some of the interventions we identified are standard practices for many HPs. These already have clinical evidence of effectiveness (e.g. Positioning and Food modification). Therefore, if other discrete interventions were to be evaluated (e.g. Oral motor exercises and Psychological support for the child), they would have to be delivered in combination with standard practices.

• Our proposed FEEDS toolkit requires further development in the context of guidance for complex interventions, optimisation and manualisation, including delivery strategies and media used (paper or electronic/web-based versions). The toolkit will include a menu of potential strategies and interventions, a brief description of what they mean to enable shared decision-making and signposting to more information or the appropriate local professional to contact.

• Use of the FEEDS toolkit of interventions in clinical practice needs to be informed by theories and models of behaviour change, including identification of barriers to and facilitators of different care pathways and MDT provision. This would need to draw on expertise of health psychology colleagues and implementation scientists, who also draw on sociological and organisational theories of change.

• Evaluation of the acceptability and feasibility of using the FEEDS toolkit in clinical practice for shared decision-making about selecting interventions collaboratively with professionals and parents is required. Acceptability and adherence in the home, school and other environments by parents and carers requires investigation.

• Consideration should be given to a trial or other evaluative study of the FEEDS toolkit. Development work would be needed to operationalise the toolkit and investigate whether or not it can be delivered with fidelity and acceptability. If evaluation of intervention with a toolkit was considered possible in a trial, it would be challenging to determine and operationalise the control/comparator group, given the variability of what constitutes treatment as usual. Thus, in addition to a RCT, other evaluative research methods should be considered. A development study, followed by a clinical implementation or effectiveness/implementation hybrid design study, may be appropriate.

• Further research is needed to identify the most appropriate tools to be used to measure the most valued outcomes. On the basis of work so far, we do not yet recommend a single primary outcome for any study: the outcomes would depend on individualised focus of change. The most appropriate and robust broad parent-reported outcome measurement tool to assess child feeding difficulties and mealtime behaviours for children with EDSD is the PediEAT questionnaire.

• To underpin further evaluation research in this area, it would be desirable to have a consensus-agreed core outcomes set for young children with neurodisability and EDSD. Core Outcome Measures in Effectiveness Trials (COMET) methodology would provide the obvious methodology to follow for this process building incrementally on the work already achieved in the FEEDS study (URL: www.comet-initiative.org, accessed 17 December 2020).

Contributions of authors

Jeremy Parr (https://orcid.org/0000-0002-2507-7878) (Professor, Paediatric Neurodisability, Consultant, Paediatric Neurodisability and Study Lead Applicant) was chief investigator, co-led the design and delivery of the project; participated in all project stages; led on the survey and Delphi survey; supervised quantitative data analysis; wrote sections of the report; and finalised the report.

Lindsay Pennington (https://orcid.org/0000-0002-4540-2586) (Reader, Communication Disorders, Speech and Language Therapist and Co-Investigator) co-led the project with Jeremy Parr; participated in all project stages; led the systematic review; co-led the mapping review; co-led evidence synthesis and led on the young people’s focus groups; wrote sections of the report; and, with Jeremy Parr, finalised the report.

Helen Taylor (https://orcid.org/0000-0002-2847-9661) (Clinical Psychologist and Clinical Research Associate) developed the survey and Delphi survey materials; ran the survey and Delphi survey and analysed the data; organised and co-facilitated the first and second round of focus groups; analysed the data from the second round of focus groups; organised and co-facilitated stakeholder consultation workshops; produced materials for the PAG; and wrote sections of the report.

Dawn Craig (https://orcid.org/0000-0002-5808-0096) (Principal Scientist, Evidence Synthesis and Co-investigator) co-led on the mapping review, led on evidence synthesis for the study and wrote sections of the report.

Christopher Morris (https://orcid.org/0000-0002-9916-507X) (Associate Professor in Child Health Research and Co-Investigator) contributed to the design and delivery of the project, particularly the survey and Delphi survey, second round parent focus group and stakeholder consultation, and edited the final report.

Helen McConachie (https://orcid.org/0000-0002-0713-3987) (Professor, Clinical Psychology and Co-investigator) led on the measurement properties review and wrote sections of the report.

Jill Cadwgan (https://orcid.org/0000-0002-1198-8400) (Consultant, Paediatric Neurodisability, Associate Clinical Researcher and Co-Investigator) contributed to the design of the survey and Delphi survey, co-facilitated the second round of professional focus groups and wrote sections of the report.

Diane Sellers (https://orcid.org/0000-0002-3090-2107) (Clinical Specialist and Lead Speech and Language Therapist and Co-Investigator) co-facilitated the second round of professional focus groups, contributed to the design of the survey and Delphi survey, and wrote sections of the report.

Morag Andrew (https://orcid.org/0000-0003-2532-1655) (Consultant, Community Paediatrics) facilitated the young people’s focus groups, conducted the literature review on measures of nutrition and wrote sections of the report.

Johanna Smith (https://orcid.org/0000-0003-1549-0279) (Parent and Co-Investigator) led on the organisation of the PAG; recruited to and co-facilitated the first and second rounds of parent focus groups and stakeholder consultation workshops; contributed to the design of the survey and Delphi survey; and co-wrote sections of the report.

Deborah Garland (https://orcid.org/0000-0002-3708-4186) (Parent, Resource Centre Manager and Co-Investigator) led on the organisation of the PAG; recruited to and co-facilitated the first and second rounds of parent focus groups and stakeholder consultation workshops; contributed to the design of the survey and Delphi survey; and co-wrote sections of the report.

Elaine McColl (https://orcid.org/0000-0001-8300-3204) (Professor, Health Services Research and Co-Investigator) contributed to the design of the survey and Delphi survey, and wrote and reviewed sections of the report.

Charlotte Buswell (https://orcid.org/0000-0002-7530-7694) (Speech and Language Therapist and Co-Investigator) contributed to the design of the survey and Delphi survey, co-facilitated the first round of professional focus groups, co-facilitated the stakeholder consultation workshops and co-wrote Chapter 7 of the report.

Julian Thomas (https://orcid.org/0000-0002-6694-0762) (Consultant, Gastroenterology and Co-Investigator) contributed to the design of the survey and Delphi survey, co-facilitated the first round of professional focus groups and co-wrote Chapter 7 of the report.

Allan Colver (https://orcid.org/0000-0002-3623-8729) (Professor, Community Child Health and Co-Investigator) contributed to the design and delivery of the project and reviewed sections of the report.

Publications

Taylor H, Pennington L, Craig D, Morris C, McConachie H, Cadwgan J, et al. (in preparation). How do UK clinical teams work with families of children with neurodisability to alleviate, through parent-delivered interventions, eating, drinking and swallowing difficulties?

Taylor H, Pennington L, Craig D, Morris C, McConachie H, Cadwgan J, et al. (in preparation). Identifying key interventions and outcomes for children with neurodisability and eating, drinking and swallowing difficulties: a Delphi survey.

Data-sharing statement

Requests to share anonymised data will be considered after the research team have published research papers arising from the project.

Patient data

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

Disclaimers

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

Role of the group

The role of the PAG is to advise on the research activities conducted as part of the research project ‘FEEDS: Focus on Early Eating, Drinking and Swallowing’. This project aims to answer the question ‘What interventions, which could be delivered at home by parents, are available to improve eating in young children with neurodisability and are suitable for investigation in pragmatic trials?’.

Responsibilities

• Provide advice on issues that affect parents who have a child with developmental difficulties who experience EDSD.

• Provide a forum for discussion of study results.

• Help to guide the development of future stages of the project (e.g. focus groups, Delphi survey and consensus workshops).

• Provide feedback on the acceptability and feasibility of proposed trial designs, including:

  • reviewing appropriate documents

  • identifying, discussing and addressing issues of common concern

  • exchanging ideas, strengthening skills and sharing examples.

Membership

This group is for parents of children with a developmental difficulty who experience EDSD, or have in the past. There will be between six and eight members. Membership of this group is by invitation only and will last until July 2019.

Accountability

The group facilitators (Johanna Smith and Deborah Garland) will report on the activities of the group to members of the FEEDS research team.

Review

On an as-needed basis, the group will review the relevance and value of its work.

Working methods/ways of working

There will be a shared learning approach. Participation in this group will be primarily through face-to-face meetings and e-mails, in which information and resources will be shared. Therefore, group members must use their own e-mail account to access the content and participate alongside the face-to-face meetings. We ask that you respect other group members’ privacy and do not discuss or share the content of the group with non-members. Johanna Smith, Deborah Garland and Helen Taylor from Newcastle University will be the responsible for organising the face-to-face meetings and the administration of e-mails.

Marshall et al.’s5 2012 search update

PROSPERO registration number: CRD42017074408.

The following strategies were used to update the Marshall et al. 5 2012 review.

The search was conducted by Fiona Beyer on the 4–8 August 2017.

National Institute for Health and Care Excellence’s Clinical Practice guideline6 search update

PROSPERO registration number: CRD42017074665.

The other searches are copied and pasted directly from the guideline appendix. However, they carried out the CINAHL search on Ovid and we have it through EBSCOhost; here is the EBSCOhost version. Note that a couple of the thesaurus headings listed in the appendix for CINAHL did not appear, so have been left out or substituted.

Marshall et al.5 review update

National Institute for Health and Care Excellence guidelines review (2017)6 update