Allele-specific chromatin remodeling in the ZPBP2/GSDMB/ORMDL3 locus associated with the risk of asthma and autoimmune disease

Am J Hum Genet. 2009 Sep;85(3):377-93. doi: 10.1016/j.ajhg.2009.08.007.

Abstract

Common SNPs in the chromosome 17q12-q21 region alter the risk for asthma, type 1 diabetes, primary biliary cirrhosis, and Crohn disease. Previous reports by us and others have linked the disease-associated genetic variants with changes in expression of GSDMB and ORMDL3 transcripts in human lymphoblastoid cell lines (LCLs). The variants also alter regulation of other transcripts, and this domain-wide cis-regulatory effect suggests a mechanism involving long-range chromatin interactions. Here, we further dissect the disease-linked haplotype and identify putative causal DNA variants via a combination of genetic and functional analyses. First, high-throughput resequencing of the region and genotyping of potential candidate variants were performed. Next, additional mapping of allelic expression differences in Yoruba HapMap LCLs allowed us to fine-map the basis of the cis-regulatory differences to a handful of candidate functional variants. Functional assays identified allele-specific differences in nucleosome distribution, an allele-specific association with the insulator protein CTCF, as well as a weak promoter activity for rs12936231. Overall, this study shows a common disease allele linked to changes in CTCF binding and nucleosome occupancy leading to altered domain-wide cis-regulation. Finally, a strong association between asthma and cis-regulatory haplotypes was observed in three independent family-based cohorts (p = 1.78 x 10(-8)). This study demonstrates the requirement of multiple parallel allele-specific tools for the investigation of noncoding disease variants and functional fine-mapping of human disease-associated haplotypes.

Publication types

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

MeSH terms

  • Adolescent
  • Alleles*
  • Asthma / complications
  • Asthma / genetics*
  • Autoimmune Diseases / complications
  • Autoimmune Diseases / genetics*
  • Base Sequence
  • Cell Line
  • Child
  • Chromatin Assembly and Disassembly / genetics*
  • Chromosomes, Human, Pair 17 / genetics
  • DNA Mutational Analysis
  • Egg Proteins / genetics*
  • Egg Proteins / metabolism
  • Female
  • Genes, Reporter
  • Genetic Predisposition to Disease
  • Haplotypes
  • Humans
  • Male
  • Membrane Proteins / genetics*
  • Membrane Proteins / metabolism
  • Molecular Sequence Data
  • Neoplasm Proteins / genetics*
  • Neoplasm Proteins / metabolism
  • Pedigree
  • Polymorphism, Single Nucleotide / genetics
  • Regulatory Sequences, Nucleic Acid / genetics
  • White People / genetics

Substances

  • Egg Proteins
  • GSDMB protein, human
  • Membrane Proteins
  • Neoplasm Proteins
  • ORMDL3 protein, human
  • ZPBP2 protein, human