Allelic dropout in the ENG gene, affecting the results of genetic testing in hereditary hemorrhagic telangiectasia

Genet Test Mol Biomarkers. 2012 Dec;16(12):1419-23. doi: 10.1089/gtmb.2012.0230. Epub 2012 Oct 9.

Abstract

Background: Hereditary hemorrhagic telangiectasia (HHT) is an autosomal-dominant vascular disorder with three disease-causing genes identified to date: ENG, ACVRL1, and SMAD4. We report an HHT patient with allelic dropout that on routine sequence analysis for a known mutation in the family (c.817-3T>G in ENG) initially seemed to be homozygous for the mutation.

Aim: To explore the possibility of allelic dropout causing a false result in this patient.

Methods: Mutation analysis of additional family members was performed and haplotype analysis carried out. New primers were designed to reveal the presence of a possible sequence variant, which could explain the presumed allelic dropout.

Results: Allelic dropout caused by a six-nucleotide duplication close to the standard reverse primer was the assumed cause of a false homozygous diagnosis.

Conclusion: Sequence variants outside of the primer regions can be the cause of allelic dropout, creating unforeseen errors in genotyping. Our finding emphasizes the need for careful quality control in all molecular genetic studies.

MeSH terms

  • Activin Receptors, Type II / genetics
  • Adolescent
  • Antigens, CD / genetics*
  • Base Sequence
  • Endoglin
  • Female
  • Genetic Testing*
  • Genotype
  • Humans
  • Loss of Heterozygosity
  • Male
  • Molecular Sequence Data
  • Receptors, Cell Surface / genetics*
  • Smad4 Protein / genetics
  • Telangiectasia, Hereditary Hemorrhagic / genetics*

Substances

  • Antigens, CD
  • ENG protein, human
  • Endoglin
  • Receptors, Cell Surface
  • SMAD4 protein, human
  • Smad4 Protein
  • ACVRL1 protein, human
  • Activin Receptors, Type II