Generalized Hailey-Hailey disease associated with c.2395C>T ATP2C1 gene mutation and fatal outcome.


Journal

Dermatology online journal
ISSN: 1087-2108
Titre abrégé: Dermatol Online J
Pays: United States
ID NLM: 9610776

Informations de publication

Date de publication:
15 Apr 2024
Historique:
received: 10 05 2024
accepted: 10 05 2024
medline: 4 7 2024
pubmed: 4 7 2024
entrez: 3 7 2024
Statut: epublish

Résumé

Hailey-Hailey disease (HHD) is a rare, autosomal dominant genodermatosis caused by a mutation of the ATP2C1 gene and presenting as an erosive dermatosis, particularly in the intertriginous areas. Generalized HHD is a rare variant. We present a case of widespread, recalcitrant HHD in a middle-aged woman with a fatal outcome. No other underlying dermatosis was identified, with the possible exception of drug sensitivity to carbamazepine. Diagnosis of HHD was confirmed by histology and genetic studies which showed a c.2395C>T mutation in the ATP2C1 gene. Concurrent pemphigus was excluded. Cases of generalized HHD are extremely rare and present a challenge in diagnosis and management. Increased awareness of this severe clinical variant is needed to improve quality of care for patients with this form of HHD.

Identifiants

pubmed: 38959923
doi: 10.5070/D330263583
doi:

Substances chimiques

ATP2C1 protein, human EC 7.2.2.10
Calcium-Transporting ATPases EC 7.2.2.10

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Auteurs

Robert J Pariser (RJ)

Department of Dermatology, Eastern Virginia Medical School, Norfolk, Virginia, USA. rpariser@gmail.com.

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Classifications MeSH