Chemotherapy‐induced eccrine squamous syringometaplasia in an infant

Abstract

There are few reports of chemotherapy-induced eccrine squamous syringometaplasia in children. We report the first case of an infant developing this condition after treatment with busulfan, fludarabine, and antithymocyte globulin in preparation for bone marrow transplantation. Twenty-eight days after transplantation, the infant developed faintly erythematous papules and plaques on the bilateral axillae, inguinal folds, and sites of adhesives. Punch biopsy revealed eccrine glands with dyskeratotic cells and focal squamous metaplasia consistent with chemotherapy-induced eccrine squamous syringometaplasia. | INTRODUCTIONEccrine squamous syringometaplasia (ESS) is defined as metaplasia of eccrine sweat ducts with transformation of the cuboidal epithelium of eccrine glands into two or more squamous layers.1 Two types of ESS have been described, chemotherapy-induced and incidental, that can be associated with several disorders. 2 When secondary to chemotherapy, the eruption is characterized by erythematous macules, papules, plaques, or vesicles in a generalized distribution or limited to the extremities, particularly the intertriginous and acral areas. 1-4 It typically appears 2-39 days after initiation of chemotherapy and has been associated with many agents. 2,3Despite the many identified causative agents, there are few reports of chemotherapy-induced ESS in children, with only two such cases in the literature. 3,4 We report a case of chemotherapyinduced ESS in a 7-month-old boy. To our knowledge, this is the first case of ESS reported in an infant. | CASE REPORTA 7-month-old boy with a history of Hurler's syndrome was admit-