Congenital esophageal stenosis owing to ectopic tracheobronchial remnants: report of four cases and review of the literature / 中华儿科杂志

ABSTRACT

<p><b>OBJECTIVE</b>Congenital esophageal stenosis owing to tracheobronchial remnants (TBR) is a rare condition. This study was conducted to understand the clinical features of TBR.</p><p><b>METHOD</b>The data of the four cases with TBR admitted to our hospital and 76 patients identified from the literature were reviewed. The clinical manifestation, X-ray, endoscopy, biopsy and treatment were studied retrospectively.</p><p><b>RESULT</b>Of the total of 80 cases, 45 were male, 33 were female, and for 2 cases the gender was unknown. Symptoms of dysphagia and regurgitation developed at the age of 1-day to 12-month. Definitive treatment was carried out at the age of 1-month to 16-year. Twenty-seven patients had associated anomalies with esophageal atresia being the most prevalent. X-ray esophagography showed segmental stenosis at the distal third of the esophagus in all patients except three. An abrupt narrow segment at the lower esophagus with marked proximal dilatation was found in 32 cases. Esophagography of 12 cases showed distal esophageal stenosis with tapered narrowing. Esophagography of 20 cases showed flask-shaped shadow of distal esophageal stenosis and one patient showed linear projection of barium at the level of stenosis. Endoscopy found almost complete obstruction of the lower esophageal lumen without signs of the esophagitis or reflux. Esophagoscopic dilatation of the stenosis was attempted in 24 cases, but was ineffective, and 3 patients suffered esophageal perforation. Seventy-nine patients underwent resection of the stenotic segment. Histologic examination of the resected specimen showed cartilage, mucus glands, resembling bronchal tissue. Post-operative complication included anastomotic stenosis, anastomotic leakage, hiatal hernia, and gastroesophageal reflux.</p><p><b>CONCLUSION</b>TBR should be suspected in patients who present with a typical history of dysphagia after ingestion of solid food. Esophagography and esophagoscopy are the essential means for diagnosis. TBR should be different from achalasia and should be diagnosed by biopsy. Operation is the only choice of treatment.</p>