Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare, benign condition featuring nodular or papular lesions of the dermis, subcutaneous tissue, and adjacent lymph nodes, commonly affecting the head and neck. The etiology is unknown, but case reports have thus far demonstrated an idiopathic origin. We describe here a case of ALHE occurring as an adverse effect of anti-tumor necrosis factor-alpha (TNF-α) inhibitor use, which has not previously been reported. An 81-year-old man with a history of rheumatoid arthritis had poor symptomatic relief from salsalate, methotrexate, and prednisone, and with development of dizziness with hydroxychloroquine. Three years after his diagnosis, he was started on adalimumab, and anti-TNF inhibitor. While he had resolution of left hand joint pains, he also developed a new dry, itchy rash with erythematous papules involving his lower back, face, and scalp. Skin biopsies from the left lower back confirmed the presence of ALHE. Adalimumab was replaced with a prednisone taper, while fluocinonide cream and hydroxyzine were started, and the rash subsided. Four months later, after the prednisone taper had ended, adalimumab was resumed and the patient's rash recurred at the patient's back, buttocks, and left leg. Adalimumab was again discontinued, this time in favor of etanercept and then certolizumab, and the patient's rash resolved and did not recur again. The pathologic confirmation of ALHE and the temporal relationship between the development and resolution of the patient's rash and the use of adalimumab indicate that ALHE can occur as an adverse reaction of some anti-TNF-α inhibitors.

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