Angioedema of the periorbital region that developed during treatment with etanercept in a case of refractory adult-onset Still's disease.

A 78-year-old Japanese man with adult-onset Still's disease that was refractory to conventional treatment, such as prednisolone (PSL) concomitant with methotrexate (MTX). Etanercept (50 mg/week) was added to PSL (12.5 mg/day) and MTX (12 mg/week). His manifestation improved dramatically, nonetheless massive edema of the periorbital region developed by the fourth injection, which kept his palpebral fissure completely closed. There was also a marked injection site reaction to etanercept. A diagnosis of angioedema due to etanercept was thus made, and the drug was discontinued. His angioedema began to ameliorate soon after antihistamines were introduced without any critical involvement, such as laryngeal obstruction.