Male-to-male transmission in Laurin-Sandrow syndrome and exclusion of RARB and RARG

Am J Med Genet A. 2005 Aug 30;137(2):148-52. doi: 10.1002/ajmg.a.30820.

Abstract

We report on a father and a son with nasal and limb defects characteristic of Laurin-Sandrow syndrome (LSS) excluding for the first time X-linked inheritance in this rare condition. Based on a search for genes expressed late during nose formation and early in limb formation we identified retinoic acid receptor B (RARB) and retinoic acid receptor G (RARG) as possible candidate genes and sequenced bidirectionally including all exons and intron-exon bounders. We identified a single nucleotide substitution in intron 2 of RARB, which is conserved in human, chimp, dog, mouse, rat, and chicken. However, it was located 83 bp from exon 2, suggesting it is a rare polymorphism which does not account for the phenotype. No other mutations were found. This suggests that another yet unknown gene is responsible for the condition.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Abnormalities, Multiple / genetics
  • Abnormalities, Multiple / pathology*
  • Adult
  • Child
  • Foot Deformities, Congenital / pathology*
  • Genetic Predisposition to Disease / genetics
  • Hand Deformities, Congenital / pathology*
  • Humans
  • Inheritance Patterns
  • Male
  • Nose / abnormalities*
  • Receptors, Retinoic Acid / genetics
  • Sex Factors
  • Syndrome

Substances

  • Receptors, Retinoic Acid