Evaluation of genetic risk loci for intracranial aneurysms in sporadic arteriovenous malformations of the brain

J Neurol Neurosurg Psychiatry. 2015 May;86(5):524-9. doi: 10.1136/jnnp-2013-307276. Epub 2014 Jul 21.

Abstract

Background: In genome-wide association studies (GWAS) five putative risk loci are associated with intracranial aneurysm. As brain arteriovenous malformations (AVM) and intracranial aneurysms are both intracranial vascular diseases and AVMs often have associated aneurysms, we investigated whether these loci are also associated with sporadic brain AVM.

Methods: We included 506 patients (168 Dutch, 338 American) and 1548 controls, all Caucasians. Controls had been recruited as part of previous GWAS. Dutch patients were genotyped by KASPar assay and US patients by Affymetrix SNP 6.0 array. Associations in each cohort were tested by univariable logistic regression modelling, with subgroup analysis in 205 American cases with aneurysm data. Meta-analysis was performed by a Mantel-Haenszel fixed-effect method.

Results: In the Dutch cohort none of the single nucleotide polymorphisms (SNPs) were associated with AVMs. In the American cohort, genotyped SNPs near SOX-17 (OR 0.74; 95% CI 0.56-0.98), RBBP8 (OR 0.76; 95% CI 0.62-0.94) and an imputed SNP near CDKN2B-AS1 (OR 0.79; 95% CI 0.64-0.98) were significantly associated with AVM. The association with SNPs near SOX-17 and CDKN2B-AS1 but not RBBP8 were strongest in patients with AVM with associated aneurysms. In the meta-analysis we found no significant associations between allele frequencies and AVM occurrence, but rs9298506, near SOX-17 approached statistical significance (OR 0.77; 95% CI 0.57-1.03, p=0.08).

Conclusions: Our meta-analysis of two Caucasian cohorts did not show an association between five aneurysm-associated loci and sporadic brain AVM. Possible involvement of SOX-17 and RBBP8, genes involved in cell cycle progression, deserves further investigation.

Keywords: CEREBROVASCULAR DISEASE; GENETICS; STROKE.

Publication types

  • Meta-Analysis
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Carrier Proteins / genetics
  • Case-Control Studies
  • Cation Transport Proteins
  • Cyclins / genetics
  • Endodeoxyribonucleases
  • GTPase-Activating Proteins
  • Gene Frequency / genetics
  • Genetic Predisposition to Disease / genetics*
  • Genome-Wide Association Study
  • Humans
  • Intracranial Aneurysm / complications*
  • Intracranial Aneurysm / genetics*
  • Intracranial Arteriovenous Malformations / complications*
  • Intracranial Arteriovenous Malformations / genetics*
  • Nuclear Proteins / genetics
  • Polymorphism, Single Nucleotide
  • RNA, Long Noncoding / genetics
  • SOXF Transcription Factors / genetics
  • Tumor Suppressor Proteins / genetics
  • White People / genetics

Substances

  • CDKN2B antisense RNA, human
  • CNNM2 protein, human
  • Carrier Proteins
  • Cation Transport Proteins
  • Cyclins
  • GTPase-Activating Proteins
  • Nuclear Proteins
  • RNA, Long Noncoding
  • SOXF Transcription Factors
  • STARD13 protein, human
  • Tumor Suppressor Proteins
  • Endodeoxyribonucleases
  • RBBP8 protein, human