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Bullous-hemorrhagic Darier disease.
María Pilar Sánchez-Salas1, Francisco Javier García Latasa de Aranibar, Rosa Oncíns Torres
1Department of Dermatology, Hospital de Barbastro, Carretera N-240, s/n, 22300 Barbastro (Huesca), Spain. psanchezsalas@gmail.com
Skinmed
|March 18, 2011
Summary
This case study highlights an unusual presentation of Darier disease in a 48-year-old man. Despite atypical features, histopathology confirmed Darier disease, emphasizing the need for thorough dermatological evaluation.
Area of Science:
- Dermatology
- Genetics
- Pathology
Background:
- Darier disease is a rare autosomal dominant genodermatosis.
- Typically presents in childhood or adolescence with characteristic skin lesions.
Observation:
- A 48-year-old man presented with a 4-month history of pruritic papular hyperkeratotic lesions, nail abnormalities, and oral papules.
- Histopathology revealed acantholysis, suprabasal clefting, and dyskeratosis, suggesting Darier disease.
- The patient had no family history and an older age of onset, making the diagnosis atypical.
Findings:
- Biopsy confirmed Darier disease, despite atypical clinical presentation.
- Elevated uric acid and triglycerides were noted; retinoid treatment was halted due to hypertriglyceridemia.
- Treatment with antihistamines, 5-fluorouracil cream, and topical tazarotene yielded mild improvement.
Implications:
- This case underscores the importance of considering Darier disease even with atypical presentations.
- Management challenges include managing comorbidities like hypertriglyceridemia and finding effective, tolerable treatments.
- Further research into genetic variants and treatment strategies for late-onset Darier disease may be warranted.
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