Elsevier

Kidney International

Volume 98, Issue 5, November 2020, Pages 1308-1322
Kidney International

Clinical Investigation
Common risk variants in NPHS1 and TNFSF15 are associated with childhood steroid-sensitive nephrotic syndrome

https://doi.org/10.1016/j.kint.2020.05.029Get rights and content
Under a Creative Commons license
open access

To understand the genetics of steroid-sensitive nephrotic syndrome (SSNS), we conducted a genome-wide association study in 987 childhood SSNS patients and 3,206 healthy controls with Japanese ancestry. Beyond known associations in the HLA-DR/DQ region, common variants in NPHS1-KIRREL2 (rs56117924, P=4.94E-20, odds ratio (OR) =1.90) and TNFSF15 (rs6478109, P=2.54E-8, OR=0.72) regions achieved genome-wide significance and were replicated in Korean, South Asian and African populations. Trans-ethnic meta-analyses including Japanese, Korean, South Asian, African, European, Hispanic and Maghrebian populations confirmed the significant associations of variants in NPHS1-KIRREL2 (Pmeta=6.71E-28, OR=1.88) and TNFSF15 (Pmeta=5.40E-11, OR=1.33) loci. Analysis of the NPHS1 risk alleles with glomerular NPHS1 mRNA expression from the same person revealed allele specific expression with significantly lower expression of the transcript derived from the risk haplotype (Wilcox test p=9.3E-4). Because rare pathogenic variants in NPHS1 cause congenital nephrotic syndrome of the Finnish type (CNSF), the present study provides further evidence that variation along the allele frequency spectrum in the same gene can cause or contribute to both a rare monogenic disease (CNSF) and a more complex, polygenic disease (SSNS).

Keywords

glomerulus
nephrotic syndrome
pediatric nephrology
podocyte

Cited by (0)

29

Current address: Genome Medical Science Project (Toyama), National Center for Global Health and Medicine (NCGM), Tokyo, Japan.

30

These authors contributed equally.

31

Current address: Department of Microbiology, Hoshi University School of Pharmacy and Pharmaceutical Sciences, Tokyo, Japan.

32

Current address: Human Biosciences Unit for the Top Global Course, Center for the Promotion of Interdisciplinary Education and Research (CPIER), Kyoto University, Kyoto, Japan.

33

A list of members of the Research Consortium on Genetics of Childhood Idiopathic Nephrotic Syndrome in Japan, the Korean Consortium of Hereditary Renal Diseases in Children, the Midwest Pediatric Nephrology Consortium (Genetics of Nephrotic Syndrome Study Group), and NEPHROVIR are listed in the Appendix. A list of members and their affiliations are in the Supplementary Notes.

34

Current address: Department of Pediatrics, Kitasato University, Kanagawa, Japan.