法布里肾病的年轻女性患者在只有尿桑体与伴侣治疗。

IF 0.7 Q4 UROLOGY & NEPHROLOGY Case Reports in Nephrology and Dialysis Pub Date : 2021-11-29 eCollection Date: 2021-09-01 DOI:10.1159/000520157
Tsugumi Fukunaga, Shingo Nakayama, Takuo Hirose, Kishin Muramatsu, Akari Endo, Yuka Miyake, Go Anan, Ikuko Oba-Yabana, Atsuhiro Kanno, Hannah Nakamura, Junichi Tani, Kimitoshi Nakamura, Kazuhiro Sumitomo, Wako Yumura, Katsutoshi Furukawa, Takefumi Mori
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引用次数: 0

摘要

法布里病(FD)是一种鞘脂代谢的x连锁疾病,由α-半乳糖苷酶a缺乏或活性降低引起。我们报告一例罕见的法布里肾病(FN),患者为21岁的日本女性,仅表现为尿桑体;肾活检后给予药物伴侣治疗(PCT)。因其母亲在我院社区医学部诊断为FD,患者接受了详细的检查。患者无肾功能不全及蛋白尿,尿中仅检出桑体。α-半乳糖苷酶A活性低,遗传分析显示为R301Q突变。经皮肾活检,光镜下发现肾小球足细胞增大和空泡,电镜下发现足细胞中有髓鞘和斑马体。经肾活检及基因分析诊断为FN。选择PCT作为预防心脏事件和肾功能障碍的治疗方法。本病例提示肾活检可能是必要的,即使是年轻妇女只有桑葚体诊断FN。用尿中桑椹体的计数来评价治疗效果是有用的。此外,由于其口服给药,PCT可能适合不能经常去医院的患者。
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Fabry Nephropathy in a Young Female Patient Presenting with Only Urinary Mulberry Bodies Treated with Chaperone Therapy.

Fabry disease (FD) is an X-linked disorder of the sphingolipid metabolism, caused by deficiency or decreased activity of α-galactosidase A. We report a rare case of Fabry nephropathy (FN) in a 21-year-old Japanese female patient presenting with only urinary mulberry bodies; she was treated with pharmacological chaperone therapy (PCT) after renal biopsy. The patient underwent a detailed examination because her mother was diagnosed with FD in the Division of Community Medicine of our hospital. She did not have renal dysfunction or proteinuria, and only mulberry bodies were detected in the urine. The activity of α-galactosidase A was low, and genetic analysis revealed the R301Q mutation. A percutaneous renal biopsy was performed, and the findings revealed enlargement and vacuolation of glomerular podocytes by light microscopy, and myelin and zebra bodies were detected in podocytes by electron microscopy. She was diagnosed with FN by renal biopsy and gene analysis. PCT was selected as the treatment to prevent cardiac events and renal dysfunction. The present case suggests that renal biopsy may be necessary even for young women with only mulberry bodies for the diagnosis of FN. It could be useful to evaluate the effect of treatment using the counts of mulberry bodies in the urine. In addition, due to its oral administration, PCT may be suitable for patients who are unable to visit the hospital frequently.

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来源期刊
CiteScore
1.20
自引率
0.00%
发文量
36
审稿时长
10 weeks
期刊介绍: This peer-reviewed online-only journal publishes original case reports covering the entire spectrum of nephrology and dialysis, including genetic susceptibility, clinical presentation, diagnosis, treatment or prevention, toxicities of therapy, critical care, supportive care, quality-of-life and survival issues. The journal will also accept case reports dealing with the use of novel technologies, both in the arena of diagnosis and treatment. Supplementary material is welcomed.
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