1787 Esophageal Stricture in Pregnancy Secondary to Stevens-Johnson Syndrome

Abstract

INTRODUCTION: Stevens-Johnson syndrome (SJS) is a rare, potentially life-threatening, mucocutaneous reaction leading to extensive necrosis and detachment of the epidermis. SJS commonly follows a viral prodrome or drug hypersensitivity reaction with an incidence of 1 to 2 cases per million per year. The long-term sequelae include mucosal, pulmonary, cutaneous, vulvovaginal and rarely esophageal complications. We present a rare case involving a pregnant patient with multiple esophageal strictures due to SJS requiring repeat balloon dilations. CASE DESCRIPTION/METHODS: A 32 year old pregnant female with a history of drug induced SJS, complicated by multiple esophageal strictures and vaginal stenosis, presented with symptoms of recurrent, persistent solid food dysphagia and weight loss. Barium esophagram showed a mid-esophageal stricture measuring 5 mm in diameter and a distal stricture measuring 2 mm in diameter just proximal to the gastroesophageal junction. Due to the severity of symptoms, pregnancy, and the etiology of the stricture, the patient was started on a graded stepwise dilating approach by esophagogastroduodenoscopy (EGD). Repeat dilations were done by a “rule of 3” approach using a Savary dilator every two to three weeks. A maximal diameter of 10 to 11 mm was achieved and symptom relief improved throughout the patient’s pregnancy. The patient had no procedural complications and delivered a healthy baby at 39 weeks by cesarean section. DISCUSSION: Over 12,000 EGDs are performed annually on pregnant women and limited follow up data showed it as relatively safe for both the mother and fetus. SJS with esophageal involvement is exceptionally rare in adults with only a few reported cases and even fewer cases during pregnancy. Esophageal strictures secondary to SJS share many similarities to strictures caused by corrosive ingestion in both presentation and management. Corrosive ingestion strictures are treated by repeated dilation and one study showed a long term satisfaction of greater than 90%. In our case this was weighed against the higher incidence of iatrogenic injury to both the mother and fetus. Further research will need to be done to determine the long term complications in pregnant patients after repeat dilations.