AbstractsMedical & Health Science

Background: A subset (~8-15%) of the patients born with esophageal atresia (EA) suffer from a lack of esophageal tissue, which makes a primary anastomosis difficult to achieve. This is most common in patients born without a distal fistula between the trachea and the distal esophageal segment. Purpose: To investigate the clinical course and outcome in patients born with long-gap esophageal atresia (LGEA) in the western region of Sweden and to develop an experimental model of guided tissue regeneration in the intrathoracic esophagus. Methods: A retrospective study of 16 consecutive patients born with LGEA between 1995 and 2010 was performed. The patients had been followed according to a structured program at one and seven years of age. The experimental studies had been performed in growing piglets, where 3 cm of the intrathoracic esophagus had been replaced with a silicone stented Biodesign® mesh. The piglets were provided with a gastrostomy through a small midline laparotomy. Factors influencing the clinical and histological outcome had been recorded. In Paper II, six piglets underwent surgery, in Paper III, ten and, in Paper IV, six. Results: No mortality was seen in the patients with LGEA. The mean age at definitive surgery was 147 days. The patients were small for gestational age. Eleven of sixteen (70%) had a delayed primary anastomosis as a definitive procedure, three had a gastroplasty and two underwent a colonic interposition. After surgery, anastomotic leakage was seen in seven of 16 (45%) patients and stricture developed in 11 of 16 (70%). At follow-up, some catch-up in weight was seen at seven years of age, but no catch-up in stature was seen. Spirometry performed at one and seven years of age showed obstruction or restriction in 9 of 14 (55%) measurements. The spirometry findings did not indicate any further need for surgery. Multiple breath washout was within the normal range in 11 of 15 (75%) measurements at one and seven years of age. Three of four (75%) of the patients with a pathological lung clearance index (LCI) at multiple breath washout required further surgery to prevent pulmonary damage due to aspiration. All patients either underwent surgery or were receiving continuous medical treatment for gastroesophageal reflux, and 7 of 16 (45%) had gastrostomy at the end of the study period. All patients were able to drink orally, but two of 16 (13%) were unable to eat solid foods. In the first experimental study (Paper II), six animals lived for one to 17 weeks after surgery. Four animals were alive for at least four weeks and in two of them (50%) the stent was lost prior to four weeks. Piglets that lived longer than four weeks had recurrent stricture and required dilation. Histology showed connective tissue and intense angiogenesis in three piglets. In two of them, living four and 17 weeks respectively after surgery, the bridging area contained islets of immature-looking cells in the submucosa. The remaining three piglets only had inflammatory cells and fibrosis in the bridging area. In Paper III, the…