Refractory Esophageal Stricture in Zollinger-Ellison Syndrome Treated with a Removable Stent

Abstract

A 63 year old man with a past medical history significant for gastrinoma resection 15 years prior and Barrett's esophagus presented with abdominal pain. Upper endoscopy (EGD) revealed grade IV esophagitis and multiple duodenal ulcers. A recurrent gastrinoma with Zollinger-Ellison Syndrome (ZES) was suspected and the patient was started on high dose proton pump inhibition therapy four times daily. An octreotide scan and liver biopsy confirmed the diagnosis of recurrent gastrinoma in the liver. Gastrin level at this time was 906 pg/mL. On the ninth day of hospitalization the patient developed significant dysphagia and underwent a second EGD. Findings were consistent with a pinpoint stricture at 28 cm. An 8.6 mm diameter 160 Olympus endoscope could not traverse the stricture and esophageal dilation was performed with through-the-scope (TTS) balloons at 6, 7, and 8 mm. A barium esophagram confirmed a 9 cm long stricture. Eight weekly endoscopies were performed for serial dilations of the stricture (some on an outpatient basis). During each session the stricture was severely narrowed and dilation was restarted using an 8 mm balloon. Triamcinolone was injected at 2 of the sessions. The patient's esophageal stricture remained refractory to all endoscopic and medical treatment. The decision was made to place a removable 12 cm long silicone coated Polyflex® self-expanding esophageal stent (Boston Scientific, Watertown, MA, USA). During the ninth endoscopy the stent was placed after balloon dilation. The stent remained in place for 5 weeks; during that time the patient noted significant improvement in his dysphagia and quality of life. The stent was then removed utilizing standard endoscope and rat tooth forcep techniques. One week after removal the patient remained asymptomatic. Comments: Esophageal complications of ZES are rare and include Barrett's esophagus, esophagitis, stricture, and perforation. This case is unique in that it is the first case to document rapid time progression from Barrett's to severe esophagitis and stricture formation in less then 5 months. Also, severe esophagitis to stricture formation occurred within 9 days. This is also the first case of successful placement of a temporary removable esophageal stent for a refractory stricture due to ZES. We suggest that an esophageal stent is beneficial in patients with ZES and challenging esophageal strictures resistant to conventional treatment.