Giant café-au-lait macule in neurofibromatosis 1: a type 2 segmental manifestation of neurofibromatosis 1?

J Am Acad Dermatol. 2008 Mar;58(3):493-7. doi: 10.1016/j.jaad.2007.03.013.

Abstract

Type 2 segmental manifestation of autosomal dominant dermatoses refers to pronounced segmental lesions superimposed on the ordinary nonsegmental phenotype, indicating loss of heterozygosity occurring at an early stage of embryogenesis. We describe a 20-year-old Taiwanese woman with typical lesions of neurofibromatosis type 1 (NF1) in the form of characteristic café-au-lait spots, neurofibromas, axillary freckling and Lisch nodules. In addition, a giant garment-like or "bathing-trunk" café-au-lait macule involved the lower half of the trunk, the buttocks, and parts of the thighs, being superimposed on the ordinary smaller spots of NF1. This large café-au-lait macule may be best explained as an example of type 2 segmental NF1. A novel mutation (3009delG) in exon 23 was also identified in this patient, which has not yet been described in sporadic and familial NF1.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Buttocks
  • Cafe-au-Lait Spots / etiology*
  • Cafe-au-Lait Spots / pathology*
  • Codon, Nonsense
  • Exons
  • Female
  • Frameshift Mutation
  • Gene Deletion
  • Genes, Neurofibromatosis 1*
  • Humans
  • Mutation*
  • Neurofibromatosis 1 / complications*
  • Neurofibromatosis 1 / genetics*
  • Thigh

Substances

  • Codon, Nonsense