Hannon, Edward;
(2022)
Long gap oesophageal atresia: Long Term Outcomes and Future Therapy.
Doctoral thesis (M.D(Res)), UCL (University College London).
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Abstract
Introduction Gastric transposition (GT) is currently the oesophageal replacement of choice for long gap oesophageal atresia (OA). Long-term outcomes of this are poorly understood. Different therapeutic options being explored include tissue engineering. Aims of the work in this thesis were to explore the long-term outcomes of GT and design animal models for in vivo testing of tissue engineered oesophagi. Methods Two long-term follow up studies of OA and GT for OA were completed in collaboration with patient support groups and from a cohort of patients from the author’s institution respectively. Outcomes included morbidity, symptoms and health related quality of life. Animal model development focussed on rat and rabbit models utilising the long cervical oesophagus to attempt circumferential implantation of tissue engineered oesophagi. Various modifications were made during the study including; gastrostomies, stenting, and variation of anaesthetic and operative technique. Results Long-term gastrointestinal and respiratory morbidity are frequently reported after OA repair. Following GT, adulthood morbidity includes strictures, redo surgery, supplementary feeding, anaemia and depression. Quality of life is negatively affected by OA and GT. A rat model for oesophageal tissue engineering was complicated by scaffold collapse and obstruction. Following seeding of cells and culture, scaffolds were mechanically weaker. Despite modifications in the rabbit model long term survival was limited by respiratory problems, scaffold obstruction and gastrostomy problems. Conclusions Long-term outcomes of OA and more specifically OA treated with GT are associated with significant morbidity including mental health problems and decreased quality of life. This mandates better follow up and transitional care when patients leave paediatric services. Neither rat nor rabbit models are robust enough to provide reliable outcomes suitable for pre-clinical studies of oesophageal tissue engineering. Nevertheless, lessons learned from these models will be useful in developing other preclinical models of oesophageal replacement and ultimately in clinical practice.
| Type: | Thesis (Doctoral) |
|---|---|
| Qualification: | M.D(Res) |
| Title: | Long gap oesophageal atresia: Long Term Outcomes and Future Therapy |
| Event: | UCL (University College London) |
| Open access status: | An open access version is available from UCL Discovery |
| Language: | English |
| Additional information: | Copyright © The Author 2022. Original content in this thesis is licensed under the terms of the Creative Commons Attribution 4.0 International (CC BY 4.0) Licence (https://creativecommons.org/licenses/by/4.0/). Any third-party copyright material present remains the property of its respective owner(s) and is licensed under its existing terms. Access may initially be restricted at the author’s request. |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health |
| URI: | https://discovery.ucl.ac.uk/id/eprint/10084778 |
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