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Variable phenotypic presentations of renal involvement in Fabry disease: a case series

Fabry disease is an X-linked genetic deficiency in the alpha-galactosidase enzyme resulting in intracellular accumulation of glycosphingolipids and multisystem organ dysfunction. Typically 50% of males and 20% of affected females have renal involvement, ranging from proteinuria or reduced renal func...

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Autores principales: McCloskey, Sarah, Brennan, Paul, Sayer, John A
Formato: Online Artículo Texto
Lenguaje:English
Publicado: F1000 Research Limited 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5930549/
https://www.ncbi.nlm.nih.gov/pubmed/29770213
http://dx.doi.org/10.12688/f1000research.13708.1
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author McCloskey, Sarah
Brennan, Paul
Sayer, John A
author_facet McCloskey, Sarah
Brennan, Paul
Sayer, John A
author_sort McCloskey, Sarah
collection PubMed
description Fabry disease is an X-linked genetic deficiency in the alpha-galactosidase enzyme resulting in intracellular accumulation of glycosphingolipids and multisystem organ dysfunction. Typically 50% of males and 20% of affected females have renal involvement, ranging from proteinuria or reduced renal function, renal parapelvic cysts and progressive renal disease ultimately requiring transplantation or dialysis. The phenotypic presentation of Fabry disease is incredibly varied and will even vary between family members with the same confirmed genetic mutation. In a cohort of patients affected by Fabry disease in the North East of England we examine the different phenotypic presentations of eight index cases (6 male, 2 female) with predominantly renal disease and the renal manifestations within their family members. The mean age of presentation was 40 years of age (range 23-59 years). Various multisystem manifestations were observed including cardiac, neurological, cerebrovascular and skin involvement. Two of the male index patients reached end stage renal disease (ESRD) requiring renal replacement therapy. Two female index patients had phenotypes limited to hypertension and proteinuria at presentation and the remaining patients had either stable or progressive chronic kidney disease at the time of diagnosis. We demonstrate the need for a high index of suspicion in order to consider Fabry disease as a diagnosis and the importance of cascade genetic screening to identify affected family members so that treatment can be initiated in a timely fashion.
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spelling pubmed-59305492018-05-15 Variable phenotypic presentations of renal involvement in Fabry disease: a case series McCloskey, Sarah Brennan, Paul Sayer, John A F1000Res Clinical Practice Article Fabry disease is an X-linked genetic deficiency in the alpha-galactosidase enzyme resulting in intracellular accumulation of glycosphingolipids and multisystem organ dysfunction. Typically 50% of males and 20% of affected females have renal involvement, ranging from proteinuria or reduced renal function, renal parapelvic cysts and progressive renal disease ultimately requiring transplantation or dialysis. The phenotypic presentation of Fabry disease is incredibly varied and will even vary between family members with the same confirmed genetic mutation. In a cohort of patients affected by Fabry disease in the North East of England we examine the different phenotypic presentations of eight index cases (6 male, 2 female) with predominantly renal disease and the renal manifestations within their family members. The mean age of presentation was 40 years of age (range 23-59 years). Various multisystem manifestations were observed including cardiac, neurological, cerebrovascular and skin involvement. Two of the male index patients reached end stage renal disease (ESRD) requiring renal replacement therapy. Two female index patients had phenotypes limited to hypertension and proteinuria at presentation and the remaining patients had either stable or progressive chronic kidney disease at the time of diagnosis. We demonstrate the need for a high index of suspicion in order to consider Fabry disease as a diagnosis and the importance of cascade genetic screening to identify affected family members so that treatment can be initiated in a timely fashion. F1000 Research Limited 2018-03-22 /pmc/articles/PMC5930549/ /pubmed/29770213 http://dx.doi.org/10.12688/f1000research.13708.1 Text en Copyright: © 2018 McCloskey S et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Clinical Practice Article
McCloskey, Sarah
Brennan, Paul
Sayer, John A
Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title_full Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title_fullStr Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title_full_unstemmed Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title_short Variable phenotypic presentations of renal involvement in Fabry disease: a case series
title_sort variable phenotypic presentations of renal involvement in fabry disease: a case series
topic Clinical Practice Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5930549/
https://www.ncbi.nlm.nih.gov/pubmed/29770213
http://dx.doi.org/10.12688/f1000research.13708.1
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