Whole-exome analysis of foetal autopsy tissue reveals a frameshift mutation in OBSL1, consistent with a diagnosis of 3-M Syndrome

BMC Genomics. 2015;16 Suppl 1(Suppl 1):S12. doi: 10.1186/1471-2164-16-S1-S12. Epub 2015 Jan 15.

Abstract

Background: We report a consanguineous couple that has experienced three consecutive pregnancy losses following the foetal ultrasound finding of short limbs. Post-termination examination revealed no skeletal dysplasia, but some subtle proximal limb shortening in two foetuses, and a spectrum of mildly dysmorphic features. Karyotype was normal in all three foetuses (46, XX) and comparative genomic hybridization microarray analysis detected no pathogenic copy number variants.

Results: Whole-exome sequencing and genome-wide homozygosity mapping revealed a previously reported frameshift mutation in the OBSL1 gene (c.1273insA p.T425nfsX40), consistent with a diagnosis of 3-M Syndrome 2 (OMIM #612921), which had not been anticipated from the clinical findings.

Conclusions: Our study provides novel insight into the early clinical manifestations of this form of 3-M syndrome, and demonstrates the utility of whole exome sequencing as a tool for prenatal diagnosis in particular when there is a family history suggestive of a recurrent set of clinical symptoms.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autopsy*
  • Cytoskeletal Proteins / genetics*
  • DNA Mutational Analysis
  • Dwarfism / diagnosis*
  • Dwarfism / genetics*
  • Exome
  • Female
  • Fetus / metabolism*
  • Frameshift Mutation / genetics*
  • Humans
  • Male
  • Muscle Hypotonia / diagnosis*
  • Muscle Hypotonia / genetics*
  • Pedigree
  • Phenotype
  • Pregnancy
  • Spine / abnormalities*

Substances

  • Cytoskeletal Proteins
  • OBSL1 protein, human

Supplementary concepts

  • Miller-McKusick-Malvaux-Syndrome (3M Syndrome)