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Brentuximab vedotin is an antibody-drug conjugate that is approved by the US Food and Drug Administration to treat refractory Hodgkin lymphoma and systemic anaplastic large cell lymphoma. Numerous clinical trials are currently evaluating the efficacy of brentuximab for other conditions, including T-cell lymphoma and steroid refractory graft-versus-host disease. Although brentuximab has been associated with various adverse effects, such as neutropenia, peripheral sensory neuropathy, fatigue, nausea, anemia, upper respiratory tract infection, diarrhea, pyrexia, rash, thrombocytopenia, cough, and vomiting, no reports exist to date of changes in hair pigmentation related to brentuximab. We report herein the first case, to our knowledge, of hair repigmentation associated with the use of brentuximab.