The aim of this study was to define the spectrum of aversive feeding behaviours seen in children with neurodisability and assess the usefulness of Child Eating Behaviour Questionnaire (CEBQ) to systemically delineate eating behaviours amongst these children.
This study was conducted in multidisciplinary tertiary gastroenterology clinic. The team consisted of gastroenterologist, neurologist, speech and language therapist and dietician. In phase one of the study case notes for a period of six months were retrospectively reviewed. Feeding behaviours of all children seen in clinic were reviewed in the context of diagnosis and clinical examination. Patients were divided into two groups: with and without aversive feeding behaviours. Feeding behaviors of the former group were described. Use of anti reflux treatment and gastrostomy was compared between the two groups.
Phase two of this study was done prospectively. Children attending the clinic who were showing aversive feeding behaviors were assessed by using CEBQ completed by the parents. Patients with oromotor or structural abnormality were excluded from this assessment. CEBQ is a validated tool for assessing eight eating styles in children . It is a parent rated questionnaire about rewarding (like food responsiveness, enjoyment of food, emotional overeating and desire to drink) and aversive (like slowness in eating, satiety response, fussiness and emotional under eating) feeding styles. It has 35 items with a score of 1-5. The results of all domains of the questionnaire were analyzed using Microsoft excel and scores compared to the normal population. This study was approved by the audit department for baseline service evaluation. Formal ethical approval was not obtained.
|Group 1: Children with feeding aversion|
|Patient||Diagnosis||Comorbidity||Feeding difficulties unexplained by oromotor difficulties|
|1||Russel silver syndrome||Poor growth||Not interested in voluntary feeding|
|2||Russell silver variant||-||Not interested in feeding|
|3||Russell Silver Syndrome||-||Aversive feeding behaviour|
|4||Congenital CMV infection||Bilateral hearing loss||Aversion to feeding|
|5||Expreterm||Chronic lung disease, atrial septal defect||Slow to feed|
|6||Expreterm||-||Not interested in feeding|
|7||Expreterm||-||Aversion to feeding|
|9||Expreterm, maternal parvovirus infection||-||Drinks excess of fluids|
|10||Expreterm||Epilepsy, visual impairment||Selective eating|
|11||Developmental delay||Epilepsy, swallowing difficulty||Aversion for fluids|
|12||Developmental delay||-||Reluctant to try solids|
|13||Developmental delay, post extradural haematoma evacuation||Constipation||Aversion to feeding|
|14||Repaired encepahlocele||Developmental delay, VP shunt||Not like solids|
|15||Cerebral palsy||Epilepsy||Reduced oral intake|
|16||Cerebral palsy||Epilepsy||Aversion to feeding|
|17||Ataxic cerebral palsy||-||Aversion to feeding|
|18||Goldenhar syndrome||Oromotor dysfunction||Takes very little by mouth|
|19||Costello syndrome||Developmental delay||Aversion to feeding|
|Group 2: Children with oromotor difficulties|
|1||Cerebral palsy||Epilepsy||Struggle to swallow solids|
|3||Chromosome 18q deletion||Eczema||Yes|
|4||Developmental delay||Epilepsy||Aspiration with thin fluids|
|6||Pierre Robin sequence with repaired cleft palate||None||Yes|
|7||Klippel Feil syndrome||Hydrocephalus||Aspiration with liquids|
|9||Smith Lemli Optiz syndrome||None||Yes|
|14||Ex-preterm||Hydrocephalus and epilepsy||Yes|
|16||Hypoxic ischemic encephalopathy||None||Yes|
|18||Cerebral palsy||Movement disorder||Yes|
|Patient||Age in years||Sex (M: Male, F: Female)||Diagnosis||Description of feeding behaviours|
|A||2||M||Not recorded||Food refusal of solids, intermittently, once a month. Will still be drinking milk during these time.|
|B||4||M||Down's syndrome with feeding gastrostomy. Cleft lip and palate repaired.||Feeding aversion, gradually improving|
|C||4||M||Lissencephaly, epilepsy, visual impairment||Aversive behavior for food and feeding.|
|D||Not recorded||M||Cerebral palsy||Aversive behavior to food, gradually improving.|
|E||8||M||Expreterm, chronic lung disease||No signs of hunger, no oral intake|
|F||10||M||Leigh's syndrome||Takes long time to feed and reduced fluid intake|
|G||8||M||Wolf-Hirschhorn syndrome||Feeding aversion may put a banana and enjoys chocolate mousse.|
|H||3||F||Expreterm||Aversion to solid foods, can manage some pureed foods.|
|I||2||F||Cardiac rhabdomyoma and global developmental delay||Fussy eater, likes to chew on textured objects like cardboard.|
|J||3||F||Hypoxic ischaemic encephalopathy||Only tolerates smooth textures like pureed foods.|
|K||4||M||Russel Silver syndrome||Aversion to feeds only has juices and soft textured foods.|
|L||2||M||Aicardi Goutieres syndrome||Refusal of oral feeds|
|M||8||M||Unconfirmed genetic syndrome||Feeding aversion, now resolving.|
|N||19||M||Cerebral palsy, epilepsy, ventriculoperitoneal shunt and global developmental delay.||Refuses to eat, keeps food in mouth for long periods before swallowing.|
|O||6||M||Global developmental delay, tracheostomy for trachea-bronchomalacia.||Aversion to food and feeding.|
|P||3||F||Global developmental delay and visual impairment.||Refusal of all solid meals.|
|Mean of study group||Standard deviation||Population mean||Standard deviation||P value|
Table 3: Results of CEBQ, mean of study in each subdomain compared with the population mean.
SR: Satiety Response; SE: Slowness of Eating; EU: Emotional Undereating;
FU: Fussiness; FR: Food Responsiveness; EF: Enjoyment of Food;
DD: Desire to Drink; EO: Emotional Overeating
Our clinical experience at a tertiary hospital feeding clinic for children with neurodevelopmental conditions suggests that children who are labelled as having "feeding aversion" can show a spectrum of behaviours like some had sensory aversion to food and feeding whereas others showed lack of food seeking. In our study we have described the spectrum of these behaviours. Irrespective of the behavior phenotype management was with multidisciplinary approach with support of dietician, speech and language therapist and gastroenterologist. The burden of care is high amongst these patients as 47% patients needed gastrostomy for supporting calorie and fluid intake.
The intrinsic component to the feeding behaviour is not always recognized in these children characterized by undereating. There may be fruitless attempts at identifying a structural abnormality, ascription of the problem to normal structural variants (tongue-tie), or to poor parenting. There is little research to describe in detail the patterns of deficient eating seen in these children with neurodevelopmental disorders. This is in marked contrast to the well-studied overeating behavioural phenotype characteristic of Prader-Willi syndrome, which is often taken as a model of intrinsic appetite control and obesity in normal population . In addition there is little controlled research to evaluate interventions, which can partly be due to the lack of consistency amongst different workers, from different professional disciplines, in use of terminology and outcome measures. Speech and language therapist observation is essential in all these children but it is not readily quantified or can be used in large scale studies and remains subjective.
Different assessment tools are developed like behaviour assessment procedure , semi structured interviews of parents and video tape of lunch times , Child Eating Behaviour Inventory (CEBI) , Dutch Eating Behaviour Questionnaire  and CEBQ . There are more research based tests for classifying children further although not used for clinical purposes such as 6-n-propylthiouracil (PROP) testing of supertasters, fungiform papillae counting [19,20]. As none of the tools capture the entire spectrum of feeding problem "multidimensional approach" to assessment taken by Archer and Szatman 1990  in a child with "food aversion" need to be considered. This includes parental report, excluding gastro-oesophageal reflux, video fluoroscopy, and speech and language therapy assessment. We used CEBQ which captures feeding styles seen in our practice essentially a tool, which with other measures, would contribute for multidimensional assessment of this cohort of children with feeding aversion.
CEBQ has 35 items and was easy to complete in the clinic setting and only mean of 6 items remained unanswered. Amongst the rewarding eating styles of emotional overeating, desire to drink, food responsiveness and enjoyment of food the scores of study cohort were significantly low. This reinforces our observation that for our study population feeding is not identified as a rewarding experience. CEBQ scores add objectivity to this observation but also identify the subdomains where the interventions can be targeted and the response can be monitored. It was not possible to cluster patients into separate groups based on scores across subdomains. Three patients B, D and M who had already shown clinical improvement did not reflect in their subdomain scores and similarly patient E with no sign of hunger had comparable scores to others. This could be attributed to the complexity of underlying medical diagnosis, previous interventions and the heterogeneity of motor, sensory, cognitive and behaviour co-morbidities which all impact on feeding. A cross sectional view of this study is the limiting factor that did not demonstrate correlation with the clinical phenotype.
Our study has been helpful in showing the spectrum of aversive feeding behaviours and CEBQ can have a potential role in multidimensional assessment of feeding aversion amongst children with neurodisability. Its role in longitudinal follow up and response to strategies is yet to be proven.
This study identifies that feeding difficulties not related to oropharyngeal structural or motor impairment can be complex to assess and manage needing feeding gastrostomy. CEBQ has affirmed non-rewarding experience of feeding in this group and with its limitations can be helpful assessment of feeding aversion.
The authors declare no conflict of interest.
Citation: Kulshrestha R, Dalzell M, Kumar R (2018) Feeding Aversion in Children with Neurodisability and its Assessment Using CEBQ: A Tertiary Feeding Clinic Experience. J Gastroenterol Hepatology Res 3: 017.
Copyright: © 2018 Kulshrestha R, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.