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Epiphrenic diverticulum composed of airway components attributed to a bronchopulmonary-foregut malformation: report
J Yoshida1, S Ikeda, S Mizumachi
1Department of Surgery, Shimonoseki City Hospital, Japan.
Abstract:
Bronchopulmonary-foregut malformation (BPFM), defined originally as pulmonary sequestration with or without communication to the esophagus, has been acknowledged to include congenital foregut diverticula. We present herein the case of a 43-year-old woman with a 9-year history of dysphagia, in whom a barium meal examination demonstrated a 2.5-cm epiphrenic diverticulum and several fistulae. A laparotomy was performed and the lower esophagus without communication to the lung was pulled down and resected, followed by an esophagogastrostomy carried out with fundopexy. Since her operation, the patient has been free of symptoms. Histologically, the diverticulum was observed to be lined by stratified squamous cells, but its shape was formed by mural cartilage, smooth muscle cells, and three ciliated-cell cysts. The dysphagia was considered to have been derived from the kinked esophagus created by the rigid diverticulum, being the possible developmental arrest of a supernumerary lung bud. These findings indicate that this case may involve BPFM in the broad sense. Although several cases of bronchogenic cysts located beneath or across the diaphragm have been reported as a subgroup of BPFM, congenital epiphrenic diverticula has rarely been described.
Insights
This study reports a rare case of congenital epiphrenic diverticulum, a form of bronchopulmonary-foregut malformation (BPFM). Surgical resection successfully treated the patient's long-standing dysphagia.
Area of Science:
- Thoracic surgery
- Gastroenterology
- Pediatric surgery
Background:
- Bronchopulmonary-foregut malformation (BPFM) encompasses congenital anomalies involving the foregut and respiratory tract.
- While bronchogenic cysts are recognized BPFM subtypes, congenital epiphrenic diverticula are infrequently described.
Observation:
- A 43-year-old woman presented with a 9-year history of dysphagia.
- Barium meal revealed a 2.5-cm epiphrenic diverticulum and fistulae.
- Surgical findings showed esophageal diverticulum without pulmonary communication.
Findings:
- Histological analysis revealed a diverticulum with stratified squamous cells, mural cartilage, smooth muscle, and ciliated-cell cysts.
- The diverticulum's rigidity likely caused esophageal kinking, leading to dysphagia.
- This case suggests BPFM in a broader sense, possibly from a supernumerary lung bud developmental anomaly.
Implications:
- Congenital epiphrenic diverticula, though rare, should be considered within the spectrum of BPFM.
- Surgical intervention can effectively resolve dysphagia caused by these malformations.
- Further research may clarify the embryological origins and classification of these complex congenital anomalies.