Fanconi's anaemia associated with multicentric Bowen's disease and decreased NK cytotoxicity

Br J Dermatol. 1993 Nov;129(5):615-8. doi: 10.1111/j.1365-2133.1993.tb00497.x.

Abstract

We report a patient with Fanconi's anaemia and multiple lesions of vulvo-anal Bowen's disease. She was thrombocytopenic and lymphopenic, and NK-mediated cytotoxicity was undetectable. The vulvar lesions did not contain papillomavirus DNA. In vitro studies showed a possible benefit from acitretin treatment on bone marrow stem cells. However, low-dose acitretin given for 14 months did not prevent the development of an anal squamous carcinoma. Acitretin therapy was, however, associated with a sustained rise in the platelet count.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Anus Neoplasms / complications
  • Anus Neoplasms / immunology
  • Bowen's Disease / complications*
  • Bowen's Disease / immunology
  • Cytotoxicity, Immunologic / physiology*
  • Fanconi Anemia / complications*
  • Fanconi Anemia / immunology
  • Female
  • Humans
  • Killer Cells, Natural / physiology*
  • Skin Neoplasms / complications*
  • Skin Neoplasms / immunology
  • Vulvar Neoplasms / complications
  • Vulvar Neoplasms / immunology