Mutation screening of SEMA3A and SEMA7A in patients with congenital hypogonadotropic hypogonadism

Pediatr Res. 2014 May;75(5):641-4. doi: 10.1038/pr.2014.23. Epub 2014 Feb 12.

Abstract

Background: Congenital hypogonadotropic hypogonadism (HH), a rare disorder characterized by absent, partial, or delayed puberty, can be caused by the lack or deficient number of hypothalamic gonadotropin-releasing hormone (GnRH) neurons. SEMA3A was recently implicated in the etiology of the disorder, and Sema7A-deficient mice have a reduced number of GnRH neurons in their brains.

Methods: SEMA3A and SEMA7A were screened by Sanger sequencing in altogether 50 Finnish HH patients (34 with Kallmann syndrome (KS; HH with hyposmia/anosmia) and 16 with normosmic HH (nHH)). In 20 patients, mutation(s) had already been found in genes known to be implicated in congenital HH.

Results: Three heterozygous variants (c.458A>G (p.Asn153Ser), c.1253A>G (p.Asn418Ser), and c.1303G>A (p.Val435Ile)) were found in SEMA3A in three KS patients, two of which also had a mutation in FGFR1. Two rare heterozygous variants (c.442C>T (p.Arg148Trp) and c.1421G>A (p.Arg474Gln)) in SEMA7A were found in one male nHH patient with a previously identified KISS1R nonsense variant and one male KS patient with a previously identified mutation in KAL1, respectively.

Conclusion: Our results suggest that heterozygous missense variants in SEMA3A and SEMA7A may modify the phenotype of KS but most likely are not alone sufficient to cause the disorder.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Antigens, CD / genetics*
  • DNA Mutational Analysis
  • Female
  • Finland
  • GPI-Linked Proteins / genetics
  • Genetic Variation
  • Gonadotropin-Releasing Hormone / metabolism
  • Heterozygote
  • Humans
  • Hypogonadism / congenital*
  • Hypogonadism / genetics*
  • Kallmann Syndrome / genetics*
  • Male
  • Mutation
  • Mutation, Missense
  • Neurons / metabolism
  • Phenotype
  • Semaphorin-3A / genetics*
  • Semaphorins / genetics*

Substances

  • Antigens, CD
  • GPI-Linked Proteins
  • SEMA3A protein, human
  • SEMA7A protein, human
  • Semaphorin-3A
  • Semaphorins
  • Gonadotropin-Releasing Hormone