Focal dermal hypoplasia (Goltz-Gorlin syndrome): a new case with a novel variant in the PORCN gene (c.1250T>C:p.F417S) and unusual spinal anomaly

Am J Med Genet A. 2013 Jul;161A(7):1750-4. doi: 10.1002/ajmg.a.35964. Epub 2013 May 21.

Abstract

Focal dermal hypoplasia (FDH; Goltz-Gorlin syndrome; OMIM 305600) is a disorder that features involvement of the skin, skeletal system, and eyes. It is caused by loss-of-function mutations in the PORCN gene. We report a young girl with FDH, microphthalmos associated with colobomatous orbital cyst, dural ectasia and cystic malformation of the spinal cord, and a de novo variant in PORCN. This association has not been previously reported, and based on these observations the phenotypic spectrum of FDH might be broader than previously appreciated. It would be prudent to alter the suggested surveillance for this rare disorder.

Publication types

  • Case Reports

MeSH terms

  • Acyltransferases
  • Child, Preschool
  • Cysts / etiology
  • Cysts / genetics
  • Female
  • Focal Dermal Hypoplasia / complications
  • Focal Dermal Hypoplasia / genetics*
  • Humans
  • Infant
  • Infant, Newborn
  • Membrane Proteins / genetics*
  • Microphthalmos / etiology
  • Microphthalmos / genetics
  • Mutation, Missense*
  • Pregnancy
  • Spinal Cord Diseases / etiology*
  • Spinal Cord Diseases / genetics

Substances

  • Membrane Proteins
  • Acyltransferases
  • PORCN protein, human