Buschke-Ollendorff syndrome

Joint Bone Spine. 2008 Mar;75(2):212-4. doi: 10.1016/j.jbspin.2007.04.027. Epub 2008 Mar 3.

Abstract

Buschke-Ollendorff syndrome (BOS) is an autosomal dominant disorder characterized by elastin-rich hamartomas and osteopoikilosis.

Case report: In a 21-year-old woman, osteopoikilosis led to the diagnosis of BOS. She had multiple, grouped, buff-colored papules over the thighs and trunk. There was no pain or pruritus associated with the skin lesions. Examination of a biopsy specimen from a papule showed thick uniform collagen fibers and normal numbers of broad interlacing elastic fibers.

Discussion: BOS is a rare disease that affects 1/20,000 population. The diagnosis rests on a thorough physical examination and careful examination of radiographs. BOS must be distinguished from other bone abnormalities such as sclerotic bone metastases, particularly when osteopoikilosis is the inaugural manifestation.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Biopsy
  • Chromosome Disorders / diagnosis*
  • Chromosome Disorders / genetics
  • Collagen / metabolism
  • DNA-Binding Proteins
  • Elastic Tissue / metabolism
  • Female
  • Hamartoma / diagnosis*
  • Hamartoma / metabolism
  • Hamartoma / pathology
  • Humans
  • Membrane Proteins / genetics
  • Mutation / genetics
  • Nuclear Proteins / genetics
  • Osteopoikilosis / diagnostic imaging*
  • Radiography
  • Skin Diseases / diagnosis*
  • Skin Diseases / metabolism
  • Skin Diseases / pathology
  • Syndrome

Substances

  • DNA-Binding Proteins
  • LEMD3 protein, human
  • Membrane Proteins
  • Nuclear Proteins
  • Collagen