Mutations in the NRG1 gene are associated with Hirschsprung disease
Mutations in the NRG1 gene are associated with Hirschsprung disease
Hirschsprung disease (HSCR, congenital colon aganglionosis) is a relatively common complex genetic condition caused by abnormal development of the enteric nervous system (ENS). Through a recent genome-wide association study conducted on Chinese HSCR patients, we identified a new HSCR contributing locus, neuregulin 1 (NRG1; 8p12), a gene known to be involved in the development of the ENS. As genes in which disease-associated common variants are found are to be considered as candidates for the search of deleterious rare variants (RVs) in the coding sequences, we sequenced the NRG1 exons of 358 sporadic HSCR patients and 333 controls. We identified a total of 13 different heterozygous RVs including 8 non-synonymous (A28G, E134K, V266L, H347Y, P356L, V486M, A511T, P608A) and 3 synonymous amino acid substitutions (P24P, T169T, L483L), a frameshift (E239fsX10), and a c.503-4insT insertion. Functional analysis of the most conserved non-synonymous substitutions, H347Y and P356L, showed uneven intracellular distribution and aberrant expression of the mutant proteins. Except for T169T and V486M, all variants were exclusive to HSCR patients. Overall, there was a statistically significant over-representation of NRG1 RVs in HSCR patients (p = 0.008). We show here that not only common, but also rare variants of the NRG1 gene contribute to HSCR. This strengthens the role of NRG1.
- University of Hong Kong (香港大學) China (People's Republic of)
- University of Hong Kong China (People's Republic of)
- Huazhong University of Science and Technology China (People's Republic of)
Male, 572, Genotype, Neuregulin-1, DNA Mutational Analysis, Immunoblotting, Hirschsprung Disease - genetics, Immunoenzyme Techniques, Neuregulin-1 - genetics, Mutation - genetics, Case-Control Studies, COS Cells, Chlorocebus aethiops, Mutation, Animals, Humans, Female, Hirschsprung Disease, Genome-Wide Association Study
Male, 572, Genotype, Neuregulin-1, DNA Mutational Analysis, Immunoblotting, Hirschsprung Disease - genetics, Immunoenzyme Techniques, Neuregulin-1 - genetics, Mutation - genetics, Case-Control Studies, COS Cells, Chlorocebus aethiops, Mutation, Animals, Humans, Female, Hirschsprung Disease, Genome-Wide Association Study
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