Frame shift mutation of LHX1 is associated with Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome

Hum Reprod. 2012 Sep;27(9):2872-5. doi: 10.1093/humrep/des206. Epub 2012 Jun 26.

Abstract

Background: The Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome is characterized by congenital aplasia of the uterus and the upper part of the vagina in women who usually have normal ovaries and a 46, XX karyotype. MRKH can occur as an isolated form (type I) or in combination with various malformations as a syndromic or a type II MRKH. To date, in most of the cases the underlying etiology remains unclear. Recently, in approximately 6% of MRKH patients, deletions of chromosomal region 17q12 have been identified. The LHX1 gene, which is located in the deletion interval, has been suggested to be a strong candidate, because targeting inactivation of Lhx1 causes a complex phenotype including aplasia of the Müllerian ducts.

Methods and results: By sequence analysis of LHX1 in a large cohort of MRKH patients, we detected a heterozygous frame shift mutation resulting in a premature stop codon. Previously, we have reported a heterozygous missense mutation of LHX1 in another MRKH patient.

Conclusions: We conclude that heterozygous mutations of LHX1 might be one cause of the MRKH syndrome in a subgroup of patients.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • 46, XX Disorders of Sex Development
  • Abnormalities, Multiple / genetics*
  • Codon, Terminator
  • Cohort Studies
  • Congenital Abnormalities
  • Female
  • Frameshift Mutation*
  • Gene Deletion
  • Heterozygote
  • Humans
  • Karyotyping
  • Kidney / abnormalities
  • LIM-Homeodomain Proteins / genetics*
  • Mullerian Ducts / abnormalities
  • Mutation, Missense
  • Phenotype
  • Somites / abnormalities
  • Spine / abnormalities
  • Transcription Factors / genetics*
  • Uterus / abnormalities
  • Vagina / abnormalities

Substances

  • Codon, Terminator
  • LHX1 protein, human
  • LIM-Homeodomain Proteins
  • Transcription Factors

Supplementary concepts

  • Mullerian aplasia