Article Text

Original research
Children with neurodisability and feeding difficulties: a UK survey of parent-delivered interventions
  1. Helen Taylor1,
  2. Lindsay Pennington1,2,
  3. Dawn Craig1,
  4. Christopher Morris3,
  5. Helen McConachie1,
  6. Jill Cadwgan4,
  7. Diane Sellers5,
  8. Morag Andrew2,
  9. Johanna Smith6,
  10. Deborah Garland7,
  11. Elaine McColl1,
  12. Charlotte Buswell2,
  13. Julian Thomas1,2,
  14. Allan Colver1,
  15. Jeremy Parr1,2
  1. 1 Population Health Sciences Institute, Newcastle University, Newcastle upon Tyne, UK
  2. 2 Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UK
  3. 3 Peninsula Childhood Disability Research Unit (PenCRU), Institute of Health Research, University of Exeter Medical School, Exeter, UK
  4. 4 Paediatric Neurosciences, Evelina London Children's Hospital, London, UK
  5. 5 Chailey Clinical Services, Sussex Community NHS Foundation Trust, Lewes, UK
  6. 6 Patient and Public Involvement, Newcastle University, Newcastle upon Tyne, UK
  7. 7 National Autistic Society, London, UK
  1. Correspondence to Professor Jeremy Parr; jeremy.parr{at}ncl.ac.uk

Abstract

Background Eating, drinking and swallowing difficulties (EDSD) are common in children with neurodisability, and have physical and non-physical causes. EDSD have substantial impacts on the child and family. Little is currently documented about what advice is usually given by professionals, including the interventions commonly used, and what informally constitutes ‘best clinical practice’. We aimed to identify current UK practice of parent-delivered interventions for EDSD for children with neurodisability, and the outcomes valued by professionals and parents.

Methods Two populations were sampled: health professionals working with children and young people (aged 0–18 years) with neurodisability who experience EDSD (n=421); parents of children with neurodisability aged up to 12 years who experience EDSD (n=359). Questionnaires were developed based on the findings from updates of three systematic reviews, a mapping review of interventions used with this population, and in consultation with health professionals and parents. The questionnaires were distributed through UK health professional and parent networks and mainstream and specialist schools.

Results Diverse professional groups, including speech and language therapists, occupational therapists, paediatricians and dietitians, support children with EDSD and neurodisability. A range of parent-delivered interventions, such as food and drink modification, positioning and modification of mealtime environment, were recommended by health professionals and are used by and acceptable to parents. Health professionals thought the interventions were effective but parents’ views were less consistent. Both health professionals and parents rated better general health and improved nutrition as the most important outcomes.

Conclusions These survey findings outline current UK practice of parent-delivered interventions for EDSD in young children with neurodisability. The survey suggests key outcomes to measure in assessing the effectiveness of interventions. Further research is now needed to fully evaluate the effectiveness of interventions and move towards an evidence-based approach to best practice.

  • neurodisability
  • neurology
  • therapeutics
  • growth
  • health services research

Data availability statement

Data are available upon reasonable request. Data that support the findings of this study are available on request from the corresponding author, Professor Jeremy R Parr. Email: Jeremy.Parr@ncl.ac.uk. Address: Level III Sir James Spence Institute, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne, NE1 4LP, UK.

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What is known about the subject?

  • Eating, drinking and swallowing difficulties (EDSD) are common in children with neurodisability and have physical and non-physical causes.

  • EDSD have a substantial impact on a child and family.

  • Little is documented about the advice usually given by health professionals, the interventions commonly used and what constitutes ‘best clinical practice’.

What this study adds?

  • A wide range of parent-delivered interventions are recommended by health professionals and used by parents to support young children with neurodisability and EDSD.

  • Many interventions are viewed by parents as acceptable to deliver at home although views on their effectiveness are mixed.

  • Better general health and improved nutrition are viewed as the most important outcomes for young children with neurodisability and EDSD by health professionals and parents

Introduction

Long-term conditions affecting the brain, nerves and muscles are often grouped under the term ‘neurodisability’.1 Eating, drinking and swallowing difficulties (EDSD) are common in children with neurodisability, and have physical and non-physical causes. Physical causes relate to reduced muscle control and coordination, impairing the efficiency and safety of sucking, chewing and swallowing. Non-physical causes include sensory sensitivities, such as extreme sensitivity to certain textures or flavours, and rigidity or rituals associated with food or mealtimes. Physical and non-physical EDSD frequently coexist (referred to hereon as mixed EDSD).

EDSD have substantial impacts on children and families. They lead to inadequate calorie intake or a restricted diet, affecting a child’s nutrition, growth and general physical health.2 EDSD make mealtimes stressful for children and their families and impact negatively on quality of life and social participation. For example, a child who requires smooth textured food may be fed separately from family mealtimes, missing social interaction; a child who will eat only a small range of foods or refuses to eat at the table may likewise miss out; both situations cause parental stress. In the UK, parents and carers of children with neurodisability and EDSD are supported by multidisciplinary teams (MDTs) of health professionals.3 Following assessment, individualised advice is given to parents and carers on how and what to feed their child to improve the safety and efficiency of eating and drinking, 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.4 Advice often comprises multiple interventions to address a child’s EDSD holistically.

The need for robust evidence about the effectiveness of therapy interventions was ranked as the top priority by young people, parents and professionals in the British Academy of Childhood Disability James Lind Alliance Research Priority Setting Partnership.5 Little is currently documented about how EDSD are managed, which interventions are commonly used, what constitutes ‘best clinical practice’ and whether there is robust evidence for the effectiveness of practice.6–9 Guidance to clinicians is limited; NICE guidance on the treatment of both children with cerebral palsy and children with autism spectrum disorder (ASD)9 10 recommends health professionals consider assessment and intervention in relation to EDSD, but provides no guidance on assessment or which interventions should be provided. As interventions are time-consuming, involve changes to family feeding routines and are sometimes contrary to parents’ beliefs about how their child should be fed,8 11 12 evidence is needed about whether interventions work.

As part of a larger research programme, Focus on Early Eating, Drinking and Swallowing (FEEDS),13 we aimed to:

  1. Identify which MDT health professionals work with children with EDSD and their parents, and which interventions they recommend

  2. Determine if the recommended EDSD interventions are acceptable for families to deliver at home

  3. Determine if the recommended EDSD interventions are considered effective by parents and health professionals

  4. Identify how the outcomes of EDSD interventions are valued by parents and health professionals

Methods

Participants

Health professionals working with children and young people (aged 0–18 years) with neurodisability who experience EDSD, and parents of children with neurodisability (aged up to 12 years) who experience EDSD, were recruited. Health professionals were recruited through relevant national bodies, such as the Royal College of Speech and Language Therapists, the College of Occupational Therapy and the British Academy of Childhood Disability. Professionals were also recruited through national and regional neurodisability networks, 24 English National Health Service (NHS) Trusts, special interest dietetic groups and local and national nursing networks. Parents were recruited through national and regional parent networks and charities such as the National Network of Parent Carer Forums, the National Autistic Society and Cerebral Palsy UK, 24 English NHS Trusts, and mainstream and specialist schools. Parents of children with ASD were also recruited through two research databases,14 15 Full recruitment strategies are outlined elsewhere.13

Measure

The questionnaire design was informed by updates of three systematic reviews,7–9 a mapping review16 17 of interventions used with this population, and in consultation with health professionals and parents. The research team developed the questionnaire drawing on clinical expertise and best practice in survey design.18 Parallel versions were developed for health professionals and parents, ensuring wording and terminology were appropriate yet comparable (see online supplemental appendices 1 and 2).

Supplemental material

Supplemental material

The questionnaire comprised three sections: (1) respondent’s demographic characteristics; (2) items about interventions, including usage (Do you use it?), effectiveness (In your opinion, is it effective?), acceptability (Was it acceptable to you to deliver at home?), timescales for change (Over what timescale does change usually occur?) and training (Is training given to parents?); (3) important outcomes (What do you think are the potential benefits of interventions for EDSD? From the list of outcomes, which are the most important for the child, parents and family?). Health professionals were also asked how they assessed outcomes.

The questionnaire asked about 25 interventions (see table 1) and 32 outcomes (see table 2), with the option to add other interventions and outcomes. Most questions offered fixed-choice responses; there were opportunities for free-text responses (eg, to provide details of the explanation/diagnosis around the child’s EDSD). Questions on respondent demographics and experience of using each intervention were compulsory for completion. Respondents only answered questions regarding further detail about each intervention they used (eg, whether it was acceptable to deliver).

Table 1

Interventions presented in the survey

Table 2

Outcomes presented in the survey

Patient and public involvement

Parent coinvestigators codesigned the research. Parents and health professionals were consulted on the list of interventions and outcomes to be included and supplementary questions, including the acceptability and effectiveness of interventions and time taken to achieve change. Parent coinvestigators contributed to questionnaire design, advising on use of language and clear descriptions of interventions. Online and paper versions of the questionnaire were piloted by parents and health professionals. A Parent Advisory Group (PAG) considered a summary of the survey findings and whether the findings were consistent with their experiences of supporting their child with EDSD, and service provision. The PAG was recruited through social media and community networks. There was equal representation from parents of children with physical and mixed EDSD and children with non-physical EDSD. Their children’s age ranged from 6 to 16 years (for further details see Parr et al 2021).13

Procedure

Recruitment was from March to September 2018. The questionnaire was distributed by email, and hosted on Qualtrics,19 with paper versions available. Letters were sent to potential participants with a link to the appropriate version of the questionnaire. Respondents were offered the opportunity to enter a prize draw to win 1 of 10 £100 vouchers. The FEEDS study protocol was followed throughout and there were no deviations (see online supplemental appendix 3).

Supplemental material

Analysis

Data were analysed using SPSS V.24.0.20 Analysis was descriptive, mostly reporting percentages of respondents in each question category. Information on the use of interventions with children with physical EDSD and mixed EDSD was combined as consultation with parents and health professionals found most children with primarily physical EDSD also have nonphysical EDSD.

Results

A total of 421 health professionals and 359 parents responded and were included in analysis. Table 3 outlines the respondent characteristics. Online supplemental appendix 4 shows participant study flow.

Supplemental material

Table 3

Characteristics of the health professionals and parents who responded

Characteristics of health professionals

Respondents reported working with infants, toddlers, preschool and school-aged children. Experience of working with children with neurodisability ranged from 0 to 38 years (mean: 12 years 2 months, SD: 8 years 8 months). Most respondents were NHS employed (86.9%) and worked in community services (71.5%) with many working across multiple settings. Most health professionals worked with children with mixed EDSD (74.6%). All respondents worked with all types of EDSD, except clinical psychologists, who reported working only with children with non-physical or mixed EDSD, and dietitians and physiotherapists, who reported only working with children with physical or mixed EDSD (see figure 1). Respondents worked alongside other professional groups including dietitians, speech and language therapists, paediatricians and occupational therapists. Most health professionals (87.9%) worked with parents to deliver interventions; 68.6% worked with education professionals.

Figure 1

Type of EDSD worked with by professional group. EDSD, Eeating,drinking and swallowing difficulties.

Characteristics of parents

Most parent respondents were aged 31–50 years (85.0%), and 92.5% were mothers. Half of respondents were parents of children with non-physical difficulties (51.0%), who reported their child’s main diagnosis as ASD. Respondents’ children’s ages ranged from 2 months to 12 years 11 months (mean: 7 years 5 months, SD 3 years 3 months); most had ongoing EDSD difficulties (88.0%).

Health professionals’ recommendation and delivery of EDSD interventions

Health professionals reported using a wide range of interventions (most used multiple interventions, median: 11, range: 0–25) (table 4) across a range of settings, including family homes, education and NHS settings. They trained parents to deliver interventions (mean across interventions: 91.1%, range: 73.7%–97.0%) and provided ongoing family support (mean 57.9%, range: 41.7%–85.5%).

Table 4

Use of interventions by health professionals overall and split by causes of EDSD

Parents’ use of and views on acceptability of EDSD interventions

Table 5 shows the frequency of parent use of each intervention. Parents thought that all interventions recommended by health professionals were acceptable to deliver at home (mean: 94.0%, range: 80.0%–100%).

Table 5

Use of interventions by parents overall and split by causes of EDSD

Effectiveness of EDSD interventions

Most health professionals reported that the interventions used were effective (mean: 98.5%, range: 90.9%–100%). Parents’ views were less consistent (mean: 48.8%, median: 51.4%, range: 29.6%–64.7%). For parents of children with physical and mixed EDSD, the interventions most frequently rated as effective were energy supplements (67.5%), hand-over-hand prompting (64.2%), modification of utensils (63.3%), modification of environment (63.0%) and food or drink modification (62.8%). For parents of children with non-physical EDSD, the interventions most frequently rated as effective were sensorimotor therapies (100%), sensory aids, (64.3%), energy supplements (60.7%) and hand-over-hand prompting (58.5%).

Evaluation of outcomes

Health professionals and parents agreed the two most important outcomes to measure in assessing the success of interventions were improved nutrition (parents 39.8%; health professionals 30.9%) and better general health (parents 30.9%; health professionals 31.6%) (table 6). All other outcomes important to parents related to children’s physical health; by contrast, health professionals prioritised outcomes related to child and family well-being, including better child quality of life and less parent/caregiver stress.

Table 6

Top five most important outcomes identified by parents and healthcare professionals

Discussion

This large UK survey found a wide range of interventions were used for children with EDSD and most children received multiple interventions. Parents found EDSD interventions acceptable to use at home. Health professionals perceived EDSD interventions used as effective, while parents’ views were less consistent. Both parents and professionals said the most important outcomes to measure were nutrition and general health. The findings are an important step in developing an evidence base about the effectiveness of EDSD interventions.

This survey identified the range of health professionals working with children who have EDSD, highlighting the diversity of needs. Most professionals reported working with children with mixed EDSD. A wide range of interventions addressed physiological and behavioural needs. Most interventions were used with all types of EDSD, indicating overlap in approach, regardless of the cause of the child’s difficulties. This highlights the importance of MDTs and is inline with the concept that an individual’s functional characteristics should form the basis for planning interventions rather than their neurodevelopmental diagnosis.21

Parents thought all interventions recommended by health professionals were acceptable to deliver at home, despite some being time-consuming and involving considerable changes to usual family feeding routines. Parents were less consistent about whether specific interventions were effective. One reason for this discrepancy might be that the training parents received was not sufficient to enable them to optimally deliver the intervention. Other reasons may be that competing demands on parents’ time resulted in the intervention being implemented inconsistently or that interventions are only effective for some children. These explanations are inline with the views of health professionals gathered within a recent qualitative study by Beresford et al.22 Our findings therefore emphasise the importance of person-centred practice and support the assessment of families’ readiness and willingness to use different interventions when deciding how best to support children with EDSD and neurodisability, and directly monitoring intervention effects.

Key outcomes for intervention were general health and nutrition, demonstrating parents and professionals prioritised child physical health over other outcomes. Clarity on the key outcomes is an important step towards understanding how to evaluate healthcare interventions and in future effectiveness studies. Our wider programme included a measurement properties review of tools to measure change in EDSD, to inform the selection of outcome measures in future effectiveness studies.13

Limitations

This survey was completed by a large number of parents and health professionals; however, dissemination methods did not allow response rate calculation from participant groups or appraisal of the extent and nature of potential response bias. High numbers of responses were received from speech and language therapists but fewer responses from other professional groups (eg, health visitors and clinical psychologists). Respondents may have had stronger views or more experience of using interventions than non-responders, introducing bias. Although the number of respondents was sufficiently large to address the aims of this study, the survey was not sufficiently powered for subgroup analysis, such as how different types of professionals deliver interventions.

Limited information was available about the frame of reference used by health professionals responding (eg, child age), with most respondents reporting that they worked across the age range 0–18 years. Health professionals were only asked to report the EDSD type of the children with whom they worked (physical, non-physical or mixed EDSD); this limited opportunities for exploration of differences between how professionals work with children from different EDSD types.

Finally, parents and professionals may have found it difficult to rank outcomes due to the choices offered for example, choosing between a child health-related outcome and a parent quality of life outcome.

Conclusions

This is a large-scale survey of current UK practice of parent-delivered interventions for EDSD in young children with neurodisability. It demonstrates the diversity of interventions used with and needed by this group of children and the overlap in approaches across the different EDSD types. It also identifies the key outcomes important to both parents and health professionals. These findings provide a synthesis of the evidence on interventions and are an important first step in developing a toolkit of interventions (see Parr et al 2021 for further details).13

Data availability statement

Data are available upon reasonable request. Data that support the findings of this study are available on request from the corresponding author, Professor Jeremy R Parr. Email: Jeremy.Parr@ncl.ac.uk. Address: Level III Sir James Spence Institute, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne, NE1 4LP, UK.

Ethics statements

Ethics approval

This study was approved by The West Midlands and the Black Country Research Ethics Committee (17/WM/0439). Completion of the survey was taken as informed consent.

Acknowledgments

We are grateful to all the participants who gave their time to complete the survey. The Sponsor for the studies was provided by Newcastle upon Tyne Hospitals NHS Foundation Trust.

References

Supplementary materials

Footnotes

  • Twitter @Pen_CRU, @@jeremyrparr

  • Contributors JP was chief investigator, co-led the design and delivery of the study and supervised data analysis. LP co-led the design and delivery of the project. HT undertook data collection and analysis. CB, CM, JC, DS, JS, DG, JT and EM contributed to the design of the survey. All authors contributed to the study design, interpretation of results, writing of the manuscript and reviewed and approved the final version.

  • Funding This work was supported by the National Institute of Health Research (NIHR) Health Technology Assessment Programme (ref: 15/156/02). The NIHR HTA report from the research (including that reported in this manuscript) can be found at: https://www.journalslibrary.nihr.ac.uk/hta/hta25220/%23/full-report. The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care.

  • Competing interests DS received a research grant from Nutricia Advanced Medical Nutrition UK (Wiltshire, UK) from 2017 to 2018, honorarium payments from Nutricia Advanced Medical Nutrition UK from 2015 to 2019 and an honorarium payment from Nutricia Advanced Medical Nutrition UK in 2018. MA received fees from Nutricia Advanced Medical Nutrition UK to attend a conference in which she was presenting industry partner research work and lecture fees/symposium presentation fees from Nutricia Advanced Medical Nutrition UK and Nestlé SA (Vevey, Switzerland). JC reports personal fees from Novartis Pharmaceuticals (Basel, Switzerland) and Ispen Pharmaceuticals (Paris, France).

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

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