Current characteristics and management of congenital esophageal stenosis: 40 consecutive cases from a multicenter study in the Kyushu area of Japan

Pediatr Surg Int. 2017 Oct;33(10):1035-1040. doi: 10.1007/s00383-017-4133-0. Epub 2017 Aug 11.

Abstract

Purpose: Congenital esophageal stenosis (CES) is rare, and the available clinical data are limited. We explored the current diagnosis, treatment and outcomes of CES.

Methods: A questionnaire survey was performed using medical records at pediatric surgical centers in the Kyushu area.

Results: Over 10 years, 40 patients (24 males) had CES. The incidence of associated anomalies was 52.5% (21/40), and that of esophageal atresia was 20.0% (8/40). The mean age at the diagnosis was 12.0 months (range, 1 day-8.8 years). Seven (17.5%) patients were diagnosed in the neonatal period. Ten (25.0%) developed CES due to tracheobronchial remnants, 27 (67.5%) due to fibromuscular stenosis (FMS) and 1 (2.5%) due to membranous stenosis + FMS. Thirty-six (90.0%) were treated by balloon dilatation (mean, 3 times; range, 1-20). Perforation at dilatation occurred in 7 (17.5%) patients, and all were diagnosed with FMS. Eighteen (45.0%) patients underwent radical operation (3 primary, 15 secondary to dilatation).

Conclusions: Our study clarified the characteristics and outcomes of CES, including neonatal diagnoses. CES occurred in 1 in every 33,000 births in the Kyushu area. Careful attention should be paid, even in cases of dilatation for FMS. CES requires long-term follow-up for symptom persistence after adequate and repeated treatment.

Keywords: Congenital esophageal stenosis; Dilatation; Neonate; Perforation.

Publication types

  • Multicenter Study

MeSH terms

  • Child
  • Child, Preschool
  • Dilatation / methods
  • Esophageal Stenosis / congenital*
  • Esophageal Stenosis / surgery
  • Esophageal Stenosis / therapy*
  • Esophagus / surgery
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Japan
  • Male
  • Retrospective Studies
  • Surveys and Questionnaires
  • Treatment Outcome