Abstract
A 20-year-old South Asian male was admitted with a diffuse erythematous, indurated and non-blanching eruption associated with a fever of 39.7 degrees Celsius, lymphadenopathy, and edema of the face and hands. He had recently started trimethoprim-sulfamethoxazole for acne treatment. On admission, lab work-up revealed transaminitis (AST:59 u/L, ALT:95 u/L), leukocytosis (WBC 28.53 cells/L) with 12% eosinophils, and creatinine of 1.7 mg/dL. His RegiScar score was >5 and he was diagnosed with Drug-Related Eosinophilic and Systemic Syndrome (DRESS). He was hospitalized for 24 days, treated with prednisone, cyclosporine, and intravenous immunoglobulin, and discharged on oral steroids with clinical and laboratory improvement. Approximately 2.5 months after discharge while the patient was continuing an oral steroid taper, he developed patchy areas of alopecia with foci of poliosis near the vertex of his scalp. He also developed patches of vitiligo-like hypopigmentation on his extremities and trunk. A punch biopsy of the scalp demonstrated a dense peribulbar lymphocytic infiltrate with prominent interface folliculitis, numerous apoptotic bulbar keratinocyte apoptosis and abundant eosinophils. The patient did not elect for any treatments directed at the alopecia. Approximately 1 year after onset of DRESS, the patient noticed gradual improvement in hair density and re-pigmentation of hairs. DRESS is a severe cutaneous drug reaction that typically occurs 15-45 days after exposure to anticonvulsants or sulfonamides. Although the exact pathomechanism is not known, involvement by CD8+ T cells with cutaneous homing markers in those with most severe visceral complications has been proposed. We hypothesize that this mechanism may be involved in the pathophysiology of DRESS-associated alopecia, which appears to be a non-scarring process. This is the first reported case of peribulbar lymphocytic interface folliculitis with eosinophils in a non-scarring alopecia as a result of DRESS.Financial Disclosure: No current or relevant financial relationships exist.