Variable response to low-dose naltrexone in patients with Darier disease: a case series.
Acitretin
/ administration & dosage
Adolescent
Adult
Darier Disease
/ drug therapy
Dermatologic Agents
/ administration & dosage
Dose-Response Relationship, Drug
Drug Therapy, Combination
Female
Humans
Isotretinoin
/ administration & dosage
Male
Naltrexone
/ administration & dosage
Sarcoplasmic Reticulum Calcium-Transporting ATPases
/ genetics
Treatment Outcome
Young Adult
Journal
Journal of the European Academy of Dermatology and Venereology : JEADV
ISSN: 1468-3083
Titre abrégé: J Eur Acad Dermatol Venereol
Pays: England
ID NLM: 9216037
Informations de publication
Date de publication:
May 2019
May 2019
Historique:
received:
16
10
2018
accepted:
19
12
2018
pubmed:
4
2
2019
medline:
18
12
2019
entrez:
4
2
2019
Statut:
ppublish
Résumé
Darier disease is a rare autosomal-dominant genodermatosis with a loss of function of a Ca To assess the efficacy of low-dose naltrexone as a treatment option in Darier disease. Six patients with biopsy-proven Darier disease (four had severe, one had moderate and one 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. The clinical response to naltrexone varied after 12 weeks. The four patients with severe Darier disease showed worsening after initial improvement during the first 4 weeks, whereas the two patients with a mild to moderate clinical manifestation clearly improved, showing almost full remission after 12 weeks with complete flattening of the keratotic papules. 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.
Sections du résumé
BACKGROUND
BACKGROUND
Darier disease is a rare autosomal-dominant genodermatosis with a loss of function of a Ca
OBJECTIVE
OBJECTIVE
To assess the efficacy of low-dose naltrexone as a treatment option in Darier disease.
METHODS
METHODS
Six patients with biopsy-proven Darier disease (four had severe, one had moderate and one 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
RESULTS
The clinical response to naltrexone varied after 12 weeks. The four patients with severe Darier disease showed worsening after initial improvement during the first 4 weeks, whereas the two 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
CONCLUSIONS
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.
Substances chimiques
Dermatologic Agents
0
Naltrexone
5S6W795CQM
Sarcoplasmic Reticulum Calcium-Transporting ATPases
EC 3.6.3.8
ATP2A2 protein, human
EC 7.2.2.10
Isotretinoin
EH28UP18IF
Acitretin
LCH760E9T7
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
950-953Informations de copyright
© 2019 European Academy of Dermatology and Venereology.