Successful pregnancies in an adult with Meier-Gorlin syndrome harboring biallelic CDT1 variants

Am J Med Genet A. 2021 Mar;185(3):871-876. doi: 10.1002/ajmg.a.62016. Epub 2020 Dec 18.

Abstract

Meier-Gorlin syndrome is an autosomal recessively inherited disorder of growth retardation, accompanied by microtia and patellae a/hypoplasia and characteristic facies. Pathogenic variants in genes associated with the initiation of DNA replication underlie the condition, with biallelic variants in CDT1 the most common cause. Using 10× Chromium genome sequencing, we report CDT1 variants in an adult female, with an inframe amino acid deletion inherited in trans with a deep intronic variant which likely serves as the branchpoint site in Intron 8. Splicing defects arising from this variant were confirmed through in vitro analysis. At 49 years, she represents the oldest patient with a molecular diagnosis described in the literature and is the first reported patient with Meier-Gorlin syndrome to have carried a successful pregnancy to term. Both of her pregnancies were complicated by postpartum hemorrhage and upon subsequent necessary hysterectomy, revealed uterine abnormalities. There is scant knowledge on reproductive ability and success in patients with Meier-Gorlin syndrome. Successful pregnancies among other clinically recognizable forms of primordial dwarfism have also not been described previously. This case is therefore of clinical interest for many forms of inherited growth retardation, and will assist in providing more information and clinical guidance for females of reproductive age.

Keywords: DNA replication; Meier-Gorlin syndrome; growth disorders; pregnancy.

Publication types

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

MeSH terms

  • Alleles
  • Alternative Splicing
  • Base Sequence
  • Cell Cycle Proteins / deficiency
  • Cell Cycle Proteins / genetics*
  • Codon, Nonsense / genetics
  • Congenital Microtia / genetics*
  • Female
  • Frameshift Mutation*
  • Growth Disorders / genetics*
  • Haplotypes / genetics
  • Humans
  • Introns / genetics
  • Micrognathism / genetics*
  • Middle Aged
  • Parity
  • Patella / abnormalities*
  • Phenotype
  • Point Mutation*
  • Postpartum Hemorrhage / genetics
  • Pregnancy
  • Pregnancy Complications / genetics*
  • Sequence Deletion
  • Uterus / abnormalities
  • Uterus / pathology
  • Whole Genome Sequencing

Substances

  • CDT1 protein, human
  • Cell Cycle Proteins
  • Codon, Nonsense

Supplementary concepts

  • Meier-Gorlin syndrome