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Atopic dermatitis (AD) is characterized by dominance of T helper type 2 (Th2) cytokines associated with infiltration of inflammatory cells. Th2 cytokines, especially IL-13, also contribute to fibrotic remodeling of the skin, as shown in murine AD models. Dupilumab is an interleukin (IL)-4 receptor-α antagonist, which blocks both IL-4 and IL-13 signaling, and is approved for the treatment of moderate to severe AD. Dupilumab is effective for treating AD symptoms such as eczema and pruritus; however, its efficacy for skin fibrosis remains unknown. We herein report that dupilumab successfully treated atypical AD-associated plaques with marked fibrosis on histology. This article is protected by copyright. All rights reserved.