Dupilumab is an interleukin (IL)-4 receptor alpha antagonist that was approved by the US Food and Drug Administration in March 2017 for the treatment of adult patients with moderate to severe atopic dermatitis not adequately controlled with topical therapies. Dupilumab is a fully human monoclonal antibody that binds to the alpha subunit of the IL-4 receptor and inhibits downstream signaling of IL-4 and IL-13, thus inhibiting the helper T cell, type 2 (TH2) pathway activation and cytokines that play a key role in atopic diseases.1 While dupilumab has been associated with adverse reactions including injection-site reactions, conjunctivitis, blepharitis, oral herpes, keratitis, eye pruritus, other herpes simplex virus infection, and dry eye,1 there have been no reports to our knowledge of hair growth in alopecia areata (AA) related to dupilumab. We report herein the first known case of hair growth in a patient with alopecia totalis associated with the use of dupilumab.