• J Formos Med Assoc · Aug 2021

    Case Reports

    Unusual presentation of linear wrist blisters associated with hereditary angioedema: The first case report in Taiwan.

    • Cheng-Tung Lin, Shyh-Dar Shyur, Li-Ching Fang, Hao-Hsuan Huang, and Yi-Yang Shih.
    • Department of Allergy, Immunology and Rheumatology, Mackay Children's Hospital, Taipei, Taiwan; Department of Pediatrics, Yonghe Cardinal Tien Hospital, New Taipei City, Taiwan. Electronic address: lintw2005@gmail.com.
    • J Formos Med Assoc. 2021 Aug 1; 120 (8): 1642-1646.

    AbstractHereditary angioedema (HAE) is an autosomal dominant disease characterized clinically by recurrent episodes of swelling in the tissues of the extremities, face, abdomen, and respiratory tract. It is most often caused by C1 esterase inhibitor (C1 INH) gene mutation. This swelling may lead to bradykinin release, resulting in recurrent, paroxysmal, painful angioedema. Blister formation is an uncommon cutaneous manifestation of HAE. Herein, we report a case of a patient with HAE who developed linear wrist blisters on her skin, with swelling, as a rare complication of HAE. She was treated with attenuated androgens (Danazol) for two weeks at our clinic, after which the blisters showed dramatic improvement. To date, only a few HAE cases have been reported across the world. Therefore, it is important to focus on and recognize the development of edema blisters as a flare of HAE, which could consequently avoid unnecessary dermatological diagnostic workup and treatment.Copyright © 2021 Formosan Medical Association. Published by Elsevier B.V. All rights reserved.

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