A case of refractory gastric ulcer associated with cytomegalovirus in adult-onset Still’s disease

Abstract

A 60-year-old female with adult-onset Still’s disease developed hematemesis after an increase in steroid dosage. Emergency upper gastrointestinal endoscopy showed a large irregularly shaped shallow A₁ stage ulcer, and hemostatic clipping of the exposed vessel was performed. Intracellular inclusion bodies characteristic of cytomegalovirus (CMV) were not detected, although serum CMV antigen was positive. The patient was treated with proton pump inhibitors (PPI) and discharged. Six weeks later the patient was re-admitted to the hospital for treatment of hemophagocytic syndrome, and steroid dosage was increased. On day 49 after admission the patient had recurrent hematemesis, and endoscopy revealed active bleeding from the same large ulcer. Histological findings of a gastric biopsy showed CMV intracellular inclusion bodies in HE staining and CMV-immunohistochemistry, and serum CMV antigenemia was present. Anti-CMV agents were administered. When serum CMV C7-HRP and CMV-DNA were no longer detected and intracellular inclusion bodies in a gastric biopsy became negative, these medications were ceased. Four weeks later, serum CMV C7-HRP and CMV-DNA became positive and on endoscopy the gastric ulcer also appeared exacerbated. The patient was re-treated with anti-CMV agents, continuing until the gastric ulcer improved to H₁ stage. During treatment of CMV-associated gastric ulceration, treatment should not be ceased when serum CMV antigen and intracellular inclusion bodies in biopsies alone become negative, rather anti-CMV agents should be continued until ulcers improve.