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
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.
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