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Variable response to low-dose naltrexone in patients with Darier disease: a case series.

Title
Variable response to low-dose naltrexone in patients with Darier disease: a case series.
Publication Type
Journal Article
Research Type
Human
Reported as
Case Report/Series/Restrospective Study
Date
February 03, 2019
Authors
D. Boehmer, K. Eyerich, U. Darsow, T. Biedermann, A. Zink
Institution
Technical University of Munich
Link
Abstract

BACKGROUND: Darier disease is a rare autosomal-dominant genodermatosis with a loss of function of a Ca2+ -ATPase pump (SERCA2-pump). Clinically, the disease is characterized by red-brown keratotic papules mainly in seborrheic areas and has only limited and unsatisfactory treatment options. Previously, low-dose naltrexone was described as a successful treatment option in Hailey-Hailey-disease, a genodermatosis with a genetic mutation coding for a similar loss of function of a Ca2+ -ATPase pump (hSPCA1-pump).

OBJECTIVE: To assess the efficacy of low-dose naltrexone as a treatment option in Darier disease.

METHODS: Six patients with biopsy-proven Darier disease (4 had severe, 1 had moderate and 1 mild clinical manifestations). The patients received off-label therapy with naltrexone (5 mg per os [p.o.]) and magnesium (200 mg p.o.). Patients were followed up every 4 weeks for minimally 12 weeks. Upon clinical presentation, the disease severity and subjective pain and itch scores were assessed, and standardized photographs were obtained.

RESULTS: The clinical response to naltrexone varied after 12 weeks. The 4 patients with severe Darier disease showed worsening after initial improvement during the first 4 weeks, whereas the 2 patients with a mild to moderate clinical manifestation clearly improved, showing almost full remission after 12 weeks with complete flattening of the keratotic papules.

CONCLUSION: Low-dose naltrexone did not have an effect on severe Darier disease compared to Hailey-Hailey disease, but it was beneficial in mild to moderate forms of the disease. Further studies are needed to confirm these observations of variable responses.

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