Intramural oesophageal bronchogenic cyst: a rare pathological finding

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A 37‐year‐old woman presented with a 2‐year history of epigastric discomfort, reflux and intermittent dysphagia. The constant dull pain was relieved by esomeprazole. Physical examination was unremarkable. Initial abdominal ultrasound and chest computed tomography (CT) failed to reveal any obvious cause for her symptoms. Gastroscopy revealed short segment Barrett's oesophagus without metaplasia or dysplasia. No submucosal pathology was appreciated.
Barium swallow suggested a small sliding hiatal hernia with an associated anterior ovoid submucosal lesion, suggestive of a duplication cyst (Fig. 1). Endoscopic ultrasound (EUS) revealed a 4 cm dumb‐bell shaped cystic structure without any solid component at 38 cm from the incisors. There was no uptake on the contrast‐enhanced images consistent with a cyst.
Due to significant ongoing symptoms, the patient underwent a laparotomy to excise the cyst. Dissection of the hiatus provided sufficient access to the lesion and thoracotomy was not required (Fig. 2). The cyst was able to be excised from the muscularis propria without oesophageal resection. A small breach in the mucosa was repaired with interrupted absorbable sutures and the excision site patched with a 360° fundoplication. The patient made an uneventful recovery and was discharged home 8 days later on a soft diet.
Histological examination revealed a benign intramural cyst with a lining of ciliated respiratory‐type epithelium (Fig. 3). These appearances are consistent with a bronchogenic cyst.
Bronchogenic cysts are most commonly located within the middle or superior mediastinum, close to the trachea or mainstem bronchi.1 The pathogenesis of these developmental cysts is still largely unknown. It has been postulated that an accessory bud detaches from the primitive ventral foregut during division in the fourth week of gestation.2 Oesophageal bronchogenic cysts are likely to result from abnormal separation from the tracheobronchial tree. Intramural bronchogenic cyst is a rare subtype of these.4
Oesophageal bronchogenic cysts mostly affect the mid to distal oesophagus of young women and the clinical presentation is variable.2 Patients are usually asymptomatic; however, some may exhibit chest pain, dysphagia, cough or dyspnoea.3 Rare cases have presented with acute back pain due to rupture into the oesophageal lumen.5 Complications include infection, bleeding and ulceration.6
Radiological diagnosis alone of bronchogenic cysts can be difficult. CT scan may show a smoothly contoured high density mass; however, these can be missed easily if not being specifically searched for, as was the case with our patient. Endoscopy may detect a rare communication of the cyst into the oesophageal lumen and exclude fixed oesophageal narrowing. EUS is the investigation of choice in the diagnosis of cysts of the oesophagus and is particularly valuable in excluding malignancy.7 Definitive diagnosis requires resection and histologic examination.
Symptomatic oesophageal cysts should be resected if the perioperative risk is deemed acceptable. This depends on patient factors and the anticipated surgical approach: laparoscopy, thoracoscopy and open surgery have all been described depending on the cyst size, location and the experience of the surgeon. Asymptomatic cysts may also be considered for resection if the diagnosis is uncertain. Close long‐term follow‐up is advocated for those managed non‐operatively, as symptoms ultimately requiring surgery developed in 45% of patients in one series.9 The overall occult malignancy rate is very low at 0.7%.
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