Journal Article

A Rare Case of Esophagitis Associated with Mycoplasma-Induced Rash and Mucositis in a 16-Year-Old Male

Puscasiu, Elena, Loh, Katrina, Khan, Seema

2018

Overview

Mycoplasma-induced rash and mucositis (MIRM) is a variant of Stevens-Johnson Syndrome (SJS) gered by pneumoniae. It is characterized by sparse/absent cutaneous involvement and moderate/severe involvement of two or more mucosal sites. Esophageal findings in MIRM have not been described and rarely identified in SJS as endoscopy is not routine. A 16-year-old male with Ehlers Danlos syndrome, PAI-1 mutation and Chiari malformation, presented with fever, URI symptoms and rapidly progressing oral blisters for three days. M. pneumoniae PCR was positive and HSV PCR was negative. He then developed eye and urogenital involvement, as well as severe odynophagia. Three years prior, a similar episode required multi-specialty evaluation and the lesions resolved without therapy or definitive diagnosis; however, viral etiology was suspected. Given the extent of oral lesions, he underwent endoscopy which showed mild ulcerations in the mid and distal esophagus. Biopsies showed intraepidermal acantholysis and a focal suprabasilar cleft. Repeat endoscopy during his second episode revealed erythema, friability and white mucosal protrusions, suggestive of pervasive exfoliative esophagitis. Esophageal biopsies described similar findings with increased severity. CMV, HSV and fungal staining were negative. The stomach and duodenum were normal both times. He was empirically treated for Mycoplasma and required nutrition via NG tube. Swallowed topical fluticasone was initiated and continued until follow-up endoscopy two months later. Due to concern for slow clinical response, he received two doses of high-dose IVIG administered three days after starting fluticasone and five days after completing a course of azithromycin. Rapid clinical improvement was noted. Two months later, he had complete endoscopic and histologic resolution of esophagitis. Fluticasone therapy was discontinued and he has remained asymptomatic. Digestive tract lesions are rare manifestations of MIRM or SJS. Limited cases have been reported and primarily in association with SJS. This is the first case with endoscopic confirmation of severe unique esophageal pathology associated with MIRM. It illustrates the extent to which esophageal mucosa can be affected in MIRM and provides reassurance that full recovery can be achieved once the underlying infection is treated. The efficacy of swallowed topical fluticasone in resolving esophagitis cannot be confirmed as additional concurrent therapies were given.

• Publisher: Wolters Kluwer Health Medical Research, Lippincott Williams & Wilkins
• Subject: Biopsy / Endoscopy / Esophagus / Gastroenterology / Mucositis