Long-term outcome of children with oesophageal atresia type III

Céline Legrand; Laurent Michaud; Julia Salleron; Dorothée Neut; Rony Sfeir; Caroline Thumerelle; Michel Bonnevalle; Dominique Turck; Frederic Gottrand

doi:10.1136/archdischild-2012-301730

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Arch Dis Child

doi:10.1136/archdischild-2012-301730

Original article

Long-term outcome of children with oesophageal atresia type III

¹Department of Pediatrics, Jeanne de Flandre Children's Hospital and Faculty of Medicine, University of Lille 2, Reference Center for Congenital Malformations of the Esophagus, Lille, France
²Biostatistics, University Hospital and Faculty of Medicine, University of Lille 2, Lille, France

Correspondence to Frederic Gottrand, Reference Center for Congenital Malformations of the Esophagus, Department of Pediatrics, Jeanne de Flandre Children's Hospital, 1 place de Verdun, 59037 Lille, France Lille, France; frederic.gottrand{at}chru-lille.fr

Contributors CL and DN designed the study, collected the data, wrote the first draft and participated in data analysis. LM and FG developed the idea, designed the study, supervised the data collection, and participated in data analysis and writing the manuscript. JS, RS, CT, MB and DT participated in data analysis and writing the manuscript.

Received 20 January 2012
Accepted 30 May 2012
Published Online First 29 June 2012

Abstract

Objective The aim of this study was to evaluate the outcome of patients with oesophageal atresia type III (EA), focusing on the presence of late sequelae and quality of life.

Methods This was a retrospective case ascertainment followed by clinical assessment of patients. The study parameters included the patients' demographic characteristics, associated abnormalities, presence of gastro-oesophageal reflux disease (GERD) and digestive or respiratory symptoms, results of a clinical examination to evaluate nutritional status, spirometry results and quality of life assessed using the PedsQL 4.0 questionnaire.

Results Of 81 patients with EA type III treated in our institution over a 10-year period, 57 (mean age 13.3 (SE 2.8) years) participated in the study. 39% of the patients underwent fundoplication and 46% presented with anastomotic stenosis requiring dilation. 75% of patients had normal nutritional status (16% were obese, 9% were undernourished). Only 19% of participants had no digestive symptoms; 61% had dysphagia and 35% had symptoms of GERD at the last follow-up. The main respiratory symptoms were chronic cough (19%) and dyspnoea on exertion (37%). Only 37% of patients had no respiratory symptoms. Spirometry showed that 50% of patients had proximal obstruction and/or pulmonary distension, and 11% had restriction syndrome. Their quality of life was good but was lower than in healthy controls (80 vs 84, p<0.05) and lower in patients born prematurely, with symptoms of GERD and with a barky cough.

Conclusion The high frequency of late sequelae in EA type III justifies regular and multidisciplinary follow-up through to adulthood.