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

Hereditary 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 bradykin...

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Veröffentlicht in:Journal of the Formosan Medical Association 2021-08, Vol.120 (8), p.1642-1646
Hauptverfasser: Lin, Cheng-Tung, Shyur, Shyh-Dar, Fang, Li-Ching, Huang, Hao-Hsuan, Shih, Yi-Yang
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Sprache:eng
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Zusammenfassung:Hereditary 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.
ISSN:0929-6646
1876-0821
DOI:10.1016/j.jfma.2021.02.006