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一名女性活体肾捐献者候选人中存在潜伏性杂合 Fabry 病。

A case of latent heterozygous Fabry disease in a female living kidney donor candidate.

机构信息

Division of Nephrology, Kagoshima University Hospital, Kagoshima, Japan.

Division of Urology, Kagoshima University Hospital, Kagoshima, Japan.

出版信息

CEN Case Rep. 2021 Feb;10(1):30-34. doi: 10.1007/s13730-020-00510-9. Epub 2020 Jul 25.

Abstract

A 52-year-old woman had been found to have hematuria at her annual checkup 5 years in a row. She hoped to donate her kidney to her husband, so we performed a percutaneous kidney biopsy at our department. It was difficult for us to detect apparent abnormalities under a light microscopic examination, and she was determined to meet the eligibility criteria for living kidney transplantation. However, the sample for electron microscopy was not evaluated before kidney donation. She subsequently underwent living kidney transplantation as a donor. A 1-h biopsy revealed swelling and obvious vacuolation of the glomerular podocytes, which were characteristic of Fabry disease. Her medical history and examinations were reviewed. No findings or episodes were observed. Pre-donation electronmicroscopy revealed numerous zebra bodies in the podocytes. A definite diagnosis of heterozygous Fabry disease was made based on the GLA gene mutation despite the normal range of leukocyte α-Gal A activity. Based on the pathological deposition of GL-3, chaperone therapy was initiated to suppress the progression of organ damage. In this case, we could not confirm a diagnosis of Fabry disease despite performing a renal biopsy prior to kidney donation. Kidney donor candidates may sometimes have factors that cannot be assumed based on medical or family history. Thus, it is important to perform a renal biopsy before kidney donation when necessary, and to always conduct a detailed evaluation including electron microscopy.

摘要

一位 52 岁的女性在连续 5 年的年度体检中发现血尿。她希望将自己的肾脏捐献给丈夫,因此我们在本科室为她进行了经皮肾活检。在光镜下很难发现明显的异常,她也符合活体肾移植的条件。然而,在捐赠肾脏之前,电子显微镜的样本未进行评估。随后,她作为供体接受了活体肾移植。1 小时的活检显示肾小球足细胞肿胀和明显的空泡化,这是法布瑞病的特征。我们回顾了她的病史和检查,没有发现任何症状或病史。供体电子显微镜检查显示足细胞中有大量斑马小体。尽管白细胞 α-Gal A 活性在正常范围内,但基于 GLA 基因突变,诊断为杂合子 Fabry 病。根据 GL-3 的病理性沉积,启动了伴侣蛋白治疗以抑制器官损伤的进展。在这种情况下,尽管在捐赠肾脏前进行了肾活检,但我们仍无法确诊 Fabry 病。肾脏供体候选者有时可能存在不能仅依据病史或家族史来假设的因素。因此,在必要时进行肾活检,并始终进行包括电子显微镜在内的详细评估非常重要。

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本文引用的文献

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European expert consensus statement on therapeutic goals in Fabry disease.欧洲专家共识声明:法布瑞氏病治疗目标。
Mol Genet Metab. 2018 Jul;124(3):189-203. doi: 10.1016/j.ymgme.2018.06.004. Epub 2018 Jun 12.
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Renal transplantation between siblings with unrecognized Fabry disease.
Transplant Proc. 2013 Jan-Feb;45(1):115-8. doi: 10.1016/j.transproceed.2012.10.024.

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