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


Journal

CEN case reports
ISSN: 2192-4449
Titre abrégé: CEN Case Rep
Pays: Japan
ID NLM: 101636244

Informations de publication

Date de publication:
02 2021
Historique:
received: 24 10 2019
accepted: 07 07 2020
pubmed: 28 7 2020
medline: 26 10 2021
entrez: 27 7 2020
Statut: ppublish

Résumé

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.

Identifiants

pubmed: 32712909
doi: 10.1007/s13730-020-00510-9
pii: 10.1007/s13730-020-00510-9
pmc: PMC7829293
doi:

Substances chimiques

1-Deoxynojirimycin 19130-96-2
migalastat C4XNY919FW
GLA protein, human EC 3.2.1.22
alpha-Galactosidase EC 3.2.1.22

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

30-34

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Auteurs

Masato Minami (M)

Division of Nephrology, Kagoshima University Hospital, Kagoshima, Japan. 8127masa@m3.kufm.kagoshima-u.ac.jp.

Emiko Mizuma (E)

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

Mai Nakahara (M)

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

Yumi Oda (Y)

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

Haruhito Yoshimine (H)

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

Koki Tokunaga (K)

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

Akihiko Mitsuke (A)

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

Yasutoshi Yamada (Y)

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

Hideki Enokida (H)

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

Kosuke Masutani (K)

Division of Nephrology and Rheumatology, Fukuoka University, Fukuoka, Japan.

Norihiko Goto (N)

Transplant Nephrology, Kidney Disease Center, Japanese Red Cross Nagoya Daini Hospital, Aichi, Japan.

Akio Ido (A)

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

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Classifications MeSH