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一名患有与弥漫性系膜硬化相关的快速进展性婴儿肾病综合征男孩的TRPC6基因杂合错义变异。

Heterozygous missense variant in TRPC6 in a boy with rapidly progressive infantile nephrotic syndrome associated with diffuse mesangial sclerosis.

作者信息

Hanafusa Hiroaki, Hidaka Yoshihiko, Yamaguchi Tomomi, Shimojo Hisashi, Tsukahara Takanori, Murase Tsubasa, Matsuoka Daisuke, Chiba Nao, Shimada Shun, Morokawa Hirokazu, Omori Norio, Minoura Hironori, Nagano China, Takano Kyoko, Nakamura Katsuya, Wakui Keiko, Fukushima Yoshimitsu, Uehara Takeshi, Nakazawa Yozo, Iijima Kazumoto, Nozu Kandai, Kosho Tomoki

机构信息

Department of Medical Genetics, Shinshu University School of Medicine, Matsumoto, Japan.

Center for Medical Genetics, Shinshu University Hospital, Matsumoto, Japan.

出版信息

Am J Med Genet A. 2021 Jul;185(7):2175-2179. doi: 10.1002/ajmg.a.62216. Epub 2021 Apr 21.

Abstract

Transient receptor potential channel C6 encoded by TRPC6 is involved in slit diaphragm formation in podocytes, and abnormalities of the TRPC6 protein cause various glomerular diseases. The first identified pathogenic variant of TRPC6 was found to cause steroid-resistant nephrotic syndrome that typically developed in adulthood and then slowly led to end-stage renal disease, along with a renal pathology of focal segmental glomerulosclerosis. Here, we report a patient with rapidly progressing infantile nephrotic syndrome and a heterozygous missense TRPC6 variant. The patient, a 2-year-old Japanese boy, developed steroid-resistant nephrotic syndrome at age 11 months. His renal function deteriorated rapidly, and peritoneal dialysis was introduced at age 1 year and 6 months. His renal pathology, obtained at age 1 year and 1 month, was consistent with diffuse mesangial sclerosis (DMS). Clinical exome analysis and custom panel analysis for hereditary renal diseases revealed a reported heterozygous missense variant in TRPC6 (NM_004621.5:c.523C > T:p.Arg175Trp). This is the first report of a patient with a TRPC6-related renal disorder associated with DMS.

摘要

由TRPC6编码的瞬时受体电位通道C6参与足细胞裂孔隔膜的形成,TRPC6蛋白异常会导致各种肾小球疾病。首个被鉴定出的TRPC6致病变体被发现会引发类固醇抵抗性肾病综合征,该病通常在成年期发病,随后缓慢发展为终末期肾病,并伴有局灶节段性肾小球硬化的肾脏病理改变。在此,我们报告一名患有快速进展性婴儿肾病综合征且携带杂合错义TRPC6变体的患者。该患者为一名2岁日本男孩,11个月大时患上类固醇抵抗性肾病综合征。其肾功能迅速恶化,1岁6个月时开始进行腹膜透析。他在1岁1个月时的肾脏病理检查结果符合弥漫性系膜硬化(DMS)特征。针对遗传性肾病的临床外显子组分析及定制基因检测板分析显示,TRPC6基因存在一个已报道过的杂合错义变体(NM_004621.5:c.523C>T:p.Arg175Trp)。这是首例与弥漫性系膜硬化相关的TRPC6相关性肾脏疾病患者报告。

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