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[Olmsted syndrome].

P Koch1, U Reinhold, W Tilgen

  • 1Hautklinik und Poliklinik der Universität des Saarlandes, Homburg/Saar, Allemagne. hapkoc@krzsun.med-rz.uni-sb.de

Annales De Dermatologie Et De Venereologie
|June 8, 2001
PubMed
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Olmsted syndrome, a rare disorder, involves skin thickening and plaques. Acitretin treatment showed significant improvement in palmoplantar and genital lesions in a young adult patient.

Area of Science:

  • Dermatology
  • Genetics

Background:

  • Olmsted syndrome is a rare congenital disorder characterized by palmoplantar keratoderma and periorificial plaques.
  • Fewer than 20 cases have been documented in medical literature.
  • Associated cutaneous and systemic findings vary in frequency.

Observation:

  • A 17-year-old female presented with extensive hyperkeratotic plaques on the genitalia, perianal region, and thighs.
  • The patient also exhibited palmoplantar keratoderma, nail dystrophy, and a small facial plaque.
  • Initial treatment with daily acitretin (25 mg) led to improvement in palmoplantar and genital lesions within one month.

Findings:

  • Sustained improvement in palmoplantar and nail lesions was observed over seven months of acitretin therapy.
  • Despite treatment, residual perianal and genital lesions persisted two years post-therapy.

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  • Acitretin demonstrated notable efficacy in managing key manifestations of Olmsted syndrome.

    • Implications:

    • The findings support acitretin as a potential treatment of choice for Olmsted syndrome in adults.
    • Further research is warranted to fully elucidate the long-term efficacy and optimal management strategies for this rare condition.
    • This case contributes to the understanding of Olmsted syndrome's clinical presentation and therapeutic responses.