Recurrent 8q13.2-13.3 microdeletions associated with branchio-oto-renal syndrome are mediated by human endogenous retroviral (HERV) sequence blocks

BMC Med Genet. 2014 Aug 19:15:90. doi: 10.1186/s12881-014-0090-9.

Abstract

Background: Human endogenous retroviral (HERV) sequences are the remnants of ancient retroviral infection and comprise approximately 8% of the human genome. The high abundance and interspersed nature of homologous HERV sequences make them ideal substrates for genomic rearrangements. A role for HERV sequences in mediating human disease-associated rearrangement has been reported but is likely currently underappreciated.

Methods and results: In the present study, two independent de novo 8q13.2-13.3 microdeletion events were identified in patients with clinical features of Branchio-Oto-Renal (BOR) syndrome. Nucleotide-level mapping demonstrated the identical breakpoints, suggesting a recurrent microdeletion including multiple genes such as EYA1, SULF1, and SLCO5A1, which is mediated by HERV1 homologous sequences.

Conclusions: These findings raise the potential that HERV sequences may more commonly underlie recombination of dosage sensitive regions associated with recurrent syndromes.

Publication types

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

MeSH terms

  • Base Sequence
  • Branchio-Oto-Renal Syndrome / genetics*
  • Branchio-Oto-Renal Syndrome / pathology
  • Chromosome Mapping
  • Chromosomes, Human, Pair 8*
  • Comparative Genomic Hybridization
  • Endogenous Retroviruses / chemistry
  • Endogenous Retroviruses / genetics*
  • Female
  • Gene Deletion
  • Hearing Loss / genetics
  • Hearing Loss / pathology
  • Humans
  • Infant
  • Infant, Newborn
  • Intracellular Signaling Peptides and Proteins / genetics
  • Male
  • Nuclear Proteins / genetics
  • Organic Anion Transporters / genetics
  • Polymorphism, Single Nucleotide
  • Protein Tyrosine Phosphatases / genetics
  • Sequence Alignment
  • Sulfotransferases / genetics
  • Tomography, X-Ray Computed

Substances

  • Intracellular Signaling Peptides and Proteins
  • Nuclear Proteins
  • Organic Anion Transporters
  • SLCO5A1 protein, human
  • SULF1 protein, human
  • Sulfotransferases
  • EYA1 protein, human
  • Protein Tyrosine Phosphatases