Title: A mixed methods exploration of early feeding in children with repaired trache-oeosophageal fistula and oesophageal atresia.
Funder: Health Education England

Abstract: Background Trache-oesophageal fistula (TOF) and Oesophageal atresia (OA) are congenital abnormalities affecting 1 in 3500 live births in the UK. Improved surgical techniques have increased survival to over 90%. However, life-long respiratory, gastrointestinal and feeding morbidities exist. Little data exists that informs healthcare practitioners about the exact nature, extent or impact of the feeding difficulties. Aim To characterise feeding and swallowing skills and difficulties in babies and young children with repaired TOF/OA and their impact on parents. Objectives To investigate the nature of pharyngeal and oesophageal stage dysphagia in children under 1 year of age. To identify factors associated with feeding outcome at 1 year of age. To describe parental experiences of establishing feeding and mealtimes in children with TOF/OA. To determine how feeding difficulties associated with TOF/OA impact on parental quality of life and well-being. To inform design of future large-scale studies of feeding/swallowing.Methods A multiphase, convergent parallel mixed methods study has been designed to provide a holistic understanding of swallow function, feeding outcome and the psychosocial impact of feeding difficuties. Retrospective cohort study (months 4-22): factors associated with feeding outcome.A 10-year retrospective case note review (n=80-100) of infants who underwent surgical repair for TOF/OA at Great Ormond Street Hospital. The primary outcome will be feeding outcome as defined by the Functional Oral Intake Scale. The secondary outcome will be height/head circumference for weight z-scores. Analysis of multiple variables will identify associations with feeding outcome. Prospective cohort study (months 10-54): swallow physiology and outcome.15-20 infants undergoing surgical repair of TOF/OA at Great Ormond Street Hospital will be recruited. Data collection will occur at Sequential, exploratory mixed methods study (months 23-40): impact of feeding difficulties on mealtimes and parental wellbeingIn collaboration with a parent support group an online forum (approximately 30 participants) will identify the impact of feeding difficulties on mealtimes and parents. This information will inform selection of questionnaires, to be completed by approximately 100 parents of children aged six months-4 years with TOF/OA. These data will determine the occurrence of identified phenomena. Impact The study will have 4 main areas of impact: Improved information for parents regarding feeding/swallowing difficulties associated with TOF/OA. Improved knowledge can help reduce anxiety. Improved information for professionals. Greater understanding of swallow dysfunction is required to develop targeted interventions. Development of an assessment algorithm for feeding/swallowing to guide clinicians as to which patients need assessment and at what time points, supporting appropriate use of resources. Informing future study design. Data will identify risk variables, define outcome measures and inform recruitment/retention for future multi-centre intervention or observational studies.

Lay abstract: Aims To describe the swallowing abilities of babies born with trache-oesophageal fistula and/or oesophageal atresia. To understand what factors contribute to a baby having feeding difficulties as a result of a trache-oesophageal fistula and/or oesophageal atresia. To understand how parents are impacted by the feeding difficulties of their baby with trache-oesophageal fistula and/or oesophageal atresia. Background A trache-oesophageal fistula (TOF) is an abnormal connection (a fistula) between the windpipe (trachea) and the food pipe (oesophagus). Oesophageal atresia (OA) occurs when the food pipe has not joined properly during foetal development, resulting in a gap between the upper and lower ends.In the UK approximately 300 babies a year are born with TOF and/or OA, 90% are born with both. These conditions are often not identified until the baby is born and starts to feed. As a result of the fistula, swallowed milk can enter from the food pipe into the windpipe and then enter the lungs. As a result of an atresia, milk cannot enter the stomach so either comes out of the mouth or enters the windpipe when swallowed.Surgery, usually carried out in the first few days of life, repairs the fistula and connects up the food pipe, allowing the baby to start swallowing milk. The surgery is extremely successful, with 90-95% of babies now surviving. However, approximately 50-80% of babies will have ongoing feeding or swallowing difficulties. These difficulties can cause chest infections, lead to food refusal, distress at mealtimes and cause considerable worry for parents. Currently we have limited understanding of: the exact nature of the feeding and swallowing difficulties the risk factors for developing difficulties the impact that feeding difficulties have on parents. Research design To improve our understanding a study has been designed consisting of 3 complementary projects.Project 1: Understanding medical factors associated with feeding difficulty This study involves a review of hospital notes for children who had repair of TOF/OA at Great Ormond Street Hospital between 2008-2018. We will collect information on the surgery, the age at which bottle or breastfeeding was started and medical conditions that can impact on feeding. Statistical analysis identify factors that are associated with how the child is feeding at 1 year of age.Project 2: Understanding how swallow function is affected by TOF/OAThis study will provide detailed information about the swallow function of 15-20 babies born with TOF/OA. The families of babies who have TOF/OA surgery at Great Ormond Street Hospital will be invited to take part. Their baby will have a detailed swallow assessment before they are 4 months old and at 8-9 months old. We will collect information about their feeding, growth, chest infections and any hospital readmission at 4 months, 8-9 months and 1 year of age. The results will improve our understanding of the type of swallowing difficulty so that treatment can be more effective. The possibility of carrying out a large-scale study of feeding and swallowing in children with TOF/OA will be explored.Project 3: Exploring the impact of TOF/OA related feeding difficultiesIn conjunction with a TOF/OA parent support group, an online forum will identify the ways in which feeding difficulties impact on mealtimes and parental well-being. This information will then be used to design a questionnaire, which families with children aged 6 months-4 years who were born with TOF/OA, will be invited to complete.This will help to identify how wide-ranging these issues are. Dissemination The results of the study will be shared through presentation at meetings and conferences, through UK and international parent support groups and through submission to medical/nursing journals.

Research activity codes: 7.1 INDIVIDUAL CARE NEEDS;8.4 RESEARCH DESIGN AND METHODOLOGIES (HEALTH SERVICES)
Health categories: ORAL AND GASTROINTESTINAL