INTRODUCTION:Acquired benign esophagorespiratory fistula (ERF) is an uncommon condition. If not identified early, it can lead to considerable morbidity and mortality. We report a case of a woman with an ovarian cystadenoma who developed spontaneous esophageal perforation and subsequently an esophagobronchial fistula with disastrous complications.
CASE PRESENTATION:A 39-year-old woman with history of depression and substance abuse presented to the emergency room with a month of painless abdominal distension, vomiting and sudden onset of respiratory distress requiring emergent intubation in the ED. Her clinical picture and initial laboratory investigations were consistent with septic shock. She was also noted to have a large abdominal mass. The endotracheal tube (ETT) yielded gastric secretions despite confirmation of its tracheal placement. Chest CT showed necrotizing pneumonia of both the lower lobes and a 6x4 cm air filled cavity at the right lung base communicating with the esophagus. Abdominal CT demonstrated a complex cystic left adnexal mass measuring 21×26×27 cms. Four hours after admission, the patient had a cardiorespiratory arrest and was successfully resuscitated. Independent lung ventilation with a left double-lumen ETT was instituted to improve oxygenation. After hemodynamic stabilization, the patient underwent a right thoracotomy on day 2. An esophago-bronchial fistula resulting from perforation of the lower esophagus into a right basal bronchus was noted. The necrotic lung was debrided, the bronchus repaired, the esophagus diverted, and mediastinal chest drains inserted. Over the next two weeks she was treated with antibiotics, parenteral nutrition but continued to be ventilator dependent and encephalopathic which was presumed to be from hypoxic-ischemic injury sustained during cardiac arrest. On day 17 the patient had an exploratory laparatomy with resection of a 14.3 lbs mucinous cystadenoma and left salpingooophorectomy. Cervical esophagostomy, adhesiolysis and placement of a gastrostomy tube were also done at the same time. Following this surgery, her ICU course was complicated by ventilator-associated pneumonia, ARDS, prolonged respiratory failure requiring tracheostomy, polymicrobial sepsis, anemia, critical illness polyneuropathy and encephalopathy. After gradual improvement in the clinical status, on day 54, the patient underwent reconstructive surgery with gastro-esophageal re-anastomosis and a jejunostomy tube placement. With steady clinical progress over the next few weeks, she was weaned off ventilator support and the tracheostomy decannulated on day 96. Remarkable neurological and physical recovery ensued and the patient was discharged without any lines or tubes on day 113 of this hospitalization.
DISCUSSIONS:Most fistulas between the esophagus and tracheo-bronchial tree are secondary to malignancy. Benign ERF’s are less common and may be traumatic or inflammatory in origin. In a case series, all inflammatory ERFs were localized to the right side beneath the bifurcation of trachea whereas traumatic ones were situated at a higher level. Inflammatory ERFs are often due to granulomatous disease –commonly tuberculosis, rarely histoplasmosis, actinomycosis and syphilis. There have been case reports of benign ERF in association with peptic esophageal strictures, Barrett’s ulcer, Crohn’s disease, but an association with a benign ovarian tumor has not been reported to date. Diagnosis in these cases is often delayed and most patients present when pulmonary complications have already developed. Spontaneous closure of the fistula cannot be expected to occur as passage of food and saliva tends to keep the tract infected and patent. The treatment of choice is esophageal diversion with resection of mediastinal nodes and necrotic lung tissue. Early surgical intervention is imperative and diligent pre-operative preparation of these patients is necessary along with use of appropriate antibiotics.
CONCLUSION:Acquired esophagorespiratory fistula (ERF) is rare but potentially life threatening. Early diagnosis and direct surgical repair provides the best chance for survival.
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