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DOI 10.1016/j.kint.2020.05.029

Медицинская библиотека · Kidney international · 2020

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

Jia Xiaoyuan, Yamamura Tomohiko, Gbadegesin Rasheed, McNulty Michelle T, Song Kyuyong, Nagano China, Hitomi Yuki, Lee Dongwon, Aiba Yoshihiro, Khor Seik-Soon, Ueno Kazuko, Kawai Yosuke, Nagasaki Masao, Noiri Eisei, Horinouchi Tomoko, Kaito Hiroshi, Hamada Riku, Okamoto Takayuki, Kamei Koichi, Kaku Yoshitsugu, Fujimaru Rika, Tanaka Ryojiro, Shima Yuko, Research Consortium on Genetics of Childhood Idiopathic Nephrotic Syndrome in Japan, Baek Jiwon, Kang Hee Gyung, Ha Il-Soo, Han Kyoung Hee, Yang Eun Mi, Korean Consortium of Hereditary Renal Diseases in Children, Abeyagunawardena Asiri, Lane Brandon, Chryst-Stangl Megan, Esezobor Christopher, Solarin Adaobi, Midwest Pediatric Nephrology Consortium (Genetics of Nephrotic Syndrome Study Group), Dossier Claire, Deschênes Georges, NEPHROVIR, Vivarelli Marina, Debiec Hanna, Ishikura Kenji, Matsuo Masafumi, Nozu Kandai, Ronco Pierre, Cheong Hae Il, Sampson Matthew G, Tokunaga Katsushi, Iijima Kazumoto

СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

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).

Для цитирования: Jia Xiaoyuan, Yamamura Tomohiko, Gbadegesin Rasheed, McNulty Michelle T, Song Kyuyong, Nagano China, Hitomi Yuki, Lee Dongwon, Aiba Yoshihiro, Khor Seik-Soon, Ueno Kazuko, Kawai Yosuke, Nagasaki Masao, Noiri Eisei, Horinouchi Tomoko, Kaito Hiroshi, Hamada Riku, Okamoto Takayuki, Kamei Koichi, Kaku Yoshitsugu, Fujimaru Rika, Tanaka Ryojiro, Shima Yuko, Research Consortium on Genetics of Childhood Idiopathic Nephrotic Syndrome in Japan, Baek Jiwon, Kang Hee Gyung, Ha Il-Soo, Han Kyoung Hee, Yang Eun Mi, Korean Consortium of Hereditary Renal Diseases in Children, Abeyagunawardena Asiri, Lane Brandon, Chryst-Stangl Megan, Esezobor Christopher, Solarin Adaobi, Midwest Pediatric Nephrology Consortium (Genetics of Nephrotic Syndrome Study Group), Dossier Claire, Deschênes Georges, NEPHROVIR, Vivarelli Marina, Debiec Hanna, Ishikura Kenji, Matsuo Masafumi, Nozu Kandai, Ronco Pierre, Cheong Hae Il, Sampson Matthew G, Tokunaga Katsushi, Iijima Kazumoto. Common risk variants in NPHS1 and TNFSF15 are associated with childhood steroid-sensitive nephrotic syndrome.. Kidney international. 2020. DOI: 10.1016/j.kint.2020.05.029.

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