A case of latent heterozygous Fabry disease in a female living kidney donor candidate.
1-Deoxynojirimycin
/ administration & dosage
Biopsy
/ methods
Fabry Disease
/ diagnosis
Female
Hematuria
/ diagnosis
Heterozygote
Humans
Kidney
/ pathology
Kidney Transplantation
/ standards
Living Donors
Medical Chaperones
Microscopy, Electron
/ methods
Middle Aged
Mutation
Podocytes
/ pathology
Treatment Outcome
alpha-Galactosidase
/ genetics
Chaperone therapy
GLA gene mutation
Heterozygous Fabry disease
Kidney transplantation
Migalastat
α-Galactosidase A
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
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
30-34Références
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