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
Population
Three UK populations were sampled:
Parents of children with neurodisability aged up to 12 years who experience EDSD. Although our focus was on young children aged up to 8 years, broadening the age limit to 12 years aimed to include parents with adequate recall in relation to the interventions that they had used historically.
Health professionals (e.g. speech and language therapists, occupational therapists, dietitians and paediatricians) working with children and young people (aged 0–18 years) with neurodisability who experience EDSD. Although the focus of the study was on young children (up to the age of 8 years), HPs often work with a wider range of ages which makes it difficult to separate those working exclusively with the younger age range.
Education professionals (e.g. teaching assistants, class teachers, head teachers and special education needs co-ordinators) working with children and young people (aged 0–18 years) with neurodisability who experience EDSD. As with HPs, education professionals often work with children from this wider age range, which makes it difficult to separate out those working exclusively with the younger age range.
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
shows participant recruitment and flow through the study.
Participant recruitment and flow through study.
Parents
shows the characteristics of the 359 respondents who completed the parent version of the survey. Most of the respondents were mothers (95%) and most were from England (92%). The majority of respondents were aged between 31 and 50 years (85%). Most of the respondents were white British (89%). The children reported on ranged in age from 2 months to 12 years 11 months [mean 7 years 5 months, standard deviation (SD) 3 years 3 months]. Half of the respondents reported that their child had non-physical EDSD (51%) and half reported their child’s main diagnosis as ASD (51%). The majority of children attended specialist (40%) or mainstream (39%) schools. Around one-third of parents reported that they had heard about the study through the research databases of children with ASD (32%).98,99 The remaining respondents heard about the survey through voluntary organisations or charities (22%), PICs (19%), schools (13%), social media (10%) and other sources (36%).
Demographic characteristics of parents who completed the survey
shows the characteristics of respondents who completed the HP and education professional versions of the survey.
Demographic characteristics of HPs and education professionals who completed the survey
Health professionals
The majority of HPs were from England (89%) and the largest group of HPs was speech and language therapists (31%). The majority of HPs worked with children with mixed EDSD (i.e. physical and non-physical causes to their EDSD) (75%). Respondents reported working across a range of age groups. Respondents’ years of experience working with children with neurodisability ranged from 1 year to 38 years (mean 12 years 2 months, SD 8 years 8 months). The majority of respondents were employed by the NHS (87%) and most worked in community-based services (72%). The majority of HPs worked with parents (88%) and education professionals (69%) to deliver interventions. We did not ask HPs where they heard about the survey.
Education professionals
Education professionals were predominantly from England (98%) and the largest professional group was teaching assistants/learning support assistants (31%). The majority of education professionals worked with children with mixed EDSD (65%). Respondents reported working across a range of age groups, with the majority working with children aged ≥ 4 years. Respondents’ years of experience working with children with neurodisability ranged from 1 year to 33 years (mean 10 years 2 months, SD 8 years 6 months). The majority of respondents worked within specialist schools (74%) and heard about the study through schools (55%).
Use of interventions
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%).
Use of interventions by parents, HPs and education professionals overall and split by the causes of EDSD
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
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%).
Number and percentage of parents and education professionals reporting interventions as ‘acceptable’
Effectiveness of interventions
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%).
Number and percentage of parents, HPs and education professionals reporting interventions as effective overall and split by the causes of EDSD
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
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%).
Percentage of parents’, HPs’ and education professionals’ viewing each outcome as ‘important’ overall and split by the causes of EDSD, ordered by overall parent responses
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.
