Novel compound heterozygous mutations in BCS1L gene causing Bjornstad syndrome in two siblings

Am J Med Genet A. 2017 May;173(5):1348-1352. doi: 10.1002/ajmg.a.38146. Epub 2017 Mar 21.

Abstract

Bjornstad syndrome is a rare condition characterized by pili torti and sensorineural hearing loss associated with pathological variations in BCS1L. Mutations in this gene are also associated with the more severe complex III deficiency and GRACILE syndrome. We report the first Italian patients with Bjornstad syndrome, two siblings with pili torti and sensorineural hearing loss, in whom we detected two novel compound heterozygous mutations in BCS1L. A thorough clinical evaluation did not reveal any features consistent with complex III deficiency or GRACILE syndrome.

Keywords: BCS1L; Bjornstad syndrome; hearing loss; novel mutations; pili torti; siblings.

MeSH terms

  • ATPases Associated with Diverse Cellular Activities
  • Electron Transport Complex III / genetics*
  • Female
  • Hair Diseases / genetics*
  • Hair Diseases / pathology
  • Hair Diseases / physiopathology
  • Hearing Loss, Sensorineural / genetics*
  • Hearing Loss, Sensorineural / physiopathology
  • Humans
  • Male
  • Mitochondrial Diseases / congenital*
  • Mitochondrial Diseases / genetics
  • Mitochondrial Diseases / physiopathology
  • Mutation, Missense
  • Pedigree
  • Siblings

Substances

  • BCS1L protein, human
  • ATPases Associated with Diverse Cellular Activities
  • Electron Transport Complex III

Supplementary concepts

  • Bjornstad syndrome
  • Pili Torti