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Narrowband ultraviolet B phototherapy associated with improvement in Netherton syndrome.

Abstract
Netherton syndrome (NS) is a rare and severe autosomal recessive ichthyosis. We report the case of a patient with NS treated successfully with narrowband ultraviolet B (NB-UVB) phototherapy after failure of low-dose oral isotretinoin. A 16-year-old girl born to consanguineous parents presented with severe ichthyosis. On examination of her hair, she was found to have trichorrhexis invaginata. Genetic analysis revealed a SPINK5 mutation specific for NS. The patient's cousin had the same mutation. The patient developed Cushing syndrome after long-term application of topical steroids. She was treated with low-dose oral isotretinoin with no benefit. NB-UVB phototherapy was started, which resulted in marked improvement after 2 months. In conclusion, we report a case of NS with failure of low-dose oral isotretinoin, for which NB-UVB phototherapy, through its apoptosis-inducing and immunomodulatory effects, was effective. However, long-term use of UVB is limited by its potential side-effects.
AuthorsI Maatouk, R Moutran, R Tomb
JournalClinical and experimental dermatology (Clin Exp Dermatol) Vol. 37 Issue 4 Pg. 364-6 (Jun 2012) ISSN: 1365-2230 [Electronic] England
PMID22582910 (Publication Type: Case Reports, Journal Article)
Copyright© The Author(s). CED © 2012 British Association of Dermatologists.
Chemical References
  • Dermatologic Agents
  • Isotretinoin
Topics
  • Adolescent
  • Dermatologic Agents (therapeutic use)
  • Female
  • Humans
  • Isotretinoin (therapeutic use)
  • Netherton Syndrome (therapy)
  • Treatment Outcome
  • Ultraviolet Therapy (methods)

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