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与指甲-髌骨综合征相关的LMX1B基因的一种新变异。

A novel variation in the LMX1B gene with nail-patella syndrome.

作者信息

Zhang Lu, Xiong Jilong, Li Hiu-Ming, Li Xia, Yu Xuewen, Liang Yingying, Sun Huili, Yang ShuDong, Shao Mumin

机构信息

Department of Pathology, Shenzhen Traditional Chinese Medicine Hospital, The Fourth Clinical Medical College of Guangzhou University of Chinese Medicine, Shenzhen 518033, China.

Department of Nephrology, Shenzhen Traditional Chinese Medicine Hospital, The Fourth Clinical Medical College of Guangzhou University of Chinese Medicine, Shenzhen 518033, China.

出版信息

Pathol Res Pract. 2025 May;269:155936. doi: 10.1016/j.prp.2025.155936. Epub 2025 Mar 29.

DOI:10.1016/j.prp.2025.155936
PMID:40174274
Abstract

Nail-patella syndrome (NPS; OMIM #161200) is an autosomal dominant disorder characterized by developmental defects in dorsal limb structures, kidneys, and eyes. The incidence of NPS is attributed to variations in the LMX1B gene. In this report, we present a novel LMX1B variation identified in a Chinese family affected by NPS. The proband, a 15-year-old male, exhibited a history of proteinuria and microscopic hematuria accompanied by renal dysfunction, nail dysplasia, bilateral patellar dysplasia, bilateral shoulder and elbow joint dysplasia and iliac horns. Histological examination revealed mild glomerular lesions. Under electron microscopy, irregular thickening of the glomerular basement membrane was observed, characterized by an appearance resembling occasional electron lucent areas ("moth-eaten" appearance) and the presence of disorganized collagen fiber bundles. Pathological findings were consistent with NPS. Genetic analysis identified a novel heterozygous variant, c.791 A>C, p.(Gln264Pro), in the patient, his father and younger brother. This new variant has been annotated as potentially pathogenic according to the recommendation of the American Society for Medical Genetics and Genomics. This represents the first report of a novel variation in the LMX1B gene. These findings expand the spectrum of variations associated with LMX1B in NPS.

摘要

指甲-髌骨综合征(NPS;OMIM编号#161200)是一种常染色体显性疾病,其特征为上肢背侧结构、肾脏和眼睛出现发育缺陷。NPS的发病率归因于LMX1B基因的变异。在本报告中,我们展示了在一个受NPS影响的中国家庭中鉴定出的一种新型LMX1B变异。先证者为一名15岁男性,有蛋白尿和镜下血尿病史,伴有肾功能不全、指甲发育异常、双侧髌骨发育异常、双侧肩肘关节发育异常以及髂骨角。组织学检查显示轻度肾小球病变。在电子显微镜下,观察到肾小球基底膜不规则增厚,其特征为出现类似偶尔的电子透亮区的外观(“虫蚀样”外观)以及存在无序的胶原纤维束。病理结果与NPS一致。基因分析在患者、其父亲和弟弟中鉴定出一种新型杂合变异,c.791 A>C,p.(Gln264Pro)。根据美国医学遗传学与基因组学学会的建议,这种新变异已被标注为可能致病。这是关于LMX1B基因新型变异的首次报告。这些发现扩展了与NPS中LMX1B相关的变异谱。

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