Successful use of topical minoxidil in the treatment of hypotrichosis associated with desmoplakin mutations.

Abstract

Syndromes associated with desmosomal protein mutations such as desmoplakin can result in hair abnormalities including congenital alopecia and hypotrichosis. Herein, we present an 8-year-old boy, with a combination of two heterozygous mutations in the DSP gene encoding desmoplakin associated with congenital alopecia and long-standing hypotrichosis, who was treated with off-label use of topical minoxidil 5% foam once daily with dramatic growth after 2 months of therapy and sustained results at 1 year. Topical minoxidil may be effective in management of congenital alopecia and hypotrichosis associated with desmoplakin mutations.

• Authors: Margaret Dowd, Nina Antonov, Maria Garzon, Christine Lauren
• Journal: Pediatric dermatology (Pediatr Dermatol) Vol. 36 Issue 6 Pg. 1010-1011 (Nov 2019) ISSN: 1525-1470 [Electronic] United States
• PMID: 31414503 (Publication Type: Case Reports, Journal Article)
• Copyright: © 2019 Wiley Periodicals, Inc.

Chemical References

• DSP protein, human
• Dermatologic Agents
• Desmoplakins
• Minoxidil

Topics

• Administration, Topical
• Alopecia (drug therapy, genetics)
• Child
• Dermatologic Agents (therapeutic use)
• Desmoplakins (genetics)
• Genetic Diseases, X-Linked (drug therapy, genetics)
• Humans
• Hypotrichosis (drug therapy, genetics)
• Male
• Minoxidil (therapeutic use)
• Mutation
• Off-Label Use