Literature DB >> 28668140

Clinical Features, Diagnosis, and Management of Patients With Anderson-Fabry Cardiomyopathy.

Haran Yogasundaram1, Daniel Kim1, Omar Oudit2, Richard B Thompson3, Frank Weidemann4, Gavin Y Oudit5.   

Abstract

Anderson-Fabry disease (AFD) is an X-linked recessive, multisystem disease of lysosomal storage. A mutation in the gene encoding the hydrolase enzyme α-galactosidase A results in its deficiency, or complete absence of activity. Subsequent progressive intracellular accumulation of glycosphingolipids, predominantly globotriaosylceramide, in various tissues, results in progressive organ dysfunction and failure, most commonly affecting the kidneys, nervous system, skin, eyes, vascular endothelium, and the heart. Cardiac involvement in AFD represents a leading cause of morbidity and mortality. Globotriaosylceramide accumulation affects cardiomyocytes, smooth muscle cells, vascular endothelial cells, and fibroblasts leading to various pathologies including valvular regurgitation, conduction disease and arrhythmias, coronary microvascular dysfunction, and right and left ventricular hypertrophy (LVH) leading to early diastolic dysfunction and late-stage systolic impairment. Diagnosis is on the basis of decreased plasma α-galactosidase activity in men and positive genetic testing in women. Contemporary large-scale screening studies have revealed a prevalence of 1%-5% in patients with unexplained LVH in multiple cohorts. Cardiac magnetic resonance imaging, with its unique tissue characterization capabilities, is the most important imaging modality to assess for cardiomyopathy in patients with AFD. Enzyme replacement therapy is indicated in AFD patients with significant organ involvement, and has been shown to clear sphingolipids from endothelial cells in other organs, as well as to reduce left ventricular mass as early as 6 months after starting treatment. There is increasing evidence that enzyme replacement therapy might be more effective if given at earlier stages of disease, before the development of LVH and myocardial fibrosis.
Copyright © 2017 Canadian Cardiovascular Society. Published by Elsevier Inc. All rights reserved.

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Year:  2017        PMID: 28668140     DOI: 10.1016/j.cjca.2017.04.015

Source DB:  PubMed          Journal:  Can J Cardiol        ISSN: 0828-282X            Impact factor:   5.223


  13 in total

1.  Secondary genomic findings in the 2020 China Neonatal Genomes Project participants.

Authors:  Hui Xiao; Jian-Tao Zhang; Xin-Ran Dong; Yu-Lan Lu; Bing-Bing Wu; Hui-Jun Wang; Zheng-Yan Zhao; Lin Yang; Wen-Hao Zhou
Journal:  World J Pediatr       Date:  2022-06-21       Impact factor: 9.186

Review 2.  Diagnosis and Screening of Patients with Fabry Disease.

Authors:  Irfan Vardarli; Christoph Rischpler; Ken Herrmann; Frank Weidemann
Journal:  Ther Clin Risk Manag       Date:  2020-06-22       Impact factor: 2.423

Review 3.  Histopathology in HCM.

Authors:  Beatriz San Millán Tejado; Cristina Jou
Journal:  Glob Cardiol Sci Pract       Date:  2018-08-12

4.  Corneal confocal microscopy detects corneal nerve damage and increased dendritic cells in Fabry disease.

Authors:  Gulfidan Bitirgen; Kultigin Turkmen; Rayaz A Malik; Ahmet Ozkagnici; Nazmi Zengin
Journal:  Sci Rep       Date:  2018-08-16       Impact factor: 4.379

5.  The utility of the Mayo Score for predicting the yield of genetic testing in patients with hypertrophic cardiomyopathy.

Authors:  Jiří Bonaventura; Patricia Norambuena; Pavol Tomašov; Denisa Jindrová; Hana Šedivá; Milan Macek; Josef Veselka
Journal:  Arch Med Sci       Date:  2018-10-08       Impact factor: 3.318

6.  Elevated Inflammatory Plasma Biomarkers in Patients With Fabry Disease: A Critical Link to Heart Failure With Preserved Ejection Fraction.

Authors:  Haran Yogasundaram; Anish Nikhanj; Brendan N Putko; Michel Boutin; Shailly Jain-Ghai; Aneal Khan; Christiane Auray-Blais; Michael L West; Gavin Y Oudit
Journal:  J Am Heart Assoc       Date:  2018-11-06       Impact factor: 5.501

Review 7.  Fabry Disease: Molecular Basis, Pathophysiology, Diagnostics and Potential Therapeutic Directions.

Authors:  Ken Kok; Kimberley C Zwiers; Rolf G Boot; Hermen S Overkleeft; Johannes M F G Aerts; Marta Artola
Journal:  Biomolecules       Date:  2021-02-12

Review 8.  Anderson-Fabry disease in heart failure.

Authors:  M M Akhtar; P M Elliott
Journal:  Biophys Rev       Date:  2018-06-16

9.  Screening for Fabry Disease in patients with unexplained left ventricular hypertrophy.

Authors:  Chandu Sadasivan; Josie T Y Chow; Bun Sheng; David K H Chan; Yiting Fan; Paul C L Choi; Jeffrey K T Wong; Mabel M B Tong; Tsz-Ngai Chan; Erik Fung; Kevin K H Kam; Joseph Y S Chan; Wai-Kin Chi; D Ian Paterson; Manohara Senaratne; Neil Brass; Gavin Y Oudit; Alex P W Lee
Journal:  PLoS One       Date:  2020-09-28       Impact factor: 3.240

Review 10.  The Human Explanted Heart Program: A translational bridge for cardiovascular medicine.

Authors:  Hao Zhang; Anissa Viveiros; Anish Nikhanj; Quynh Nguyen; Kaiming Wang; Wei Wang; Darren H Freed; John C Mullen; Roderick MacArthur; Daniel H Kim; Wayne Tymchak; Consolato M Sergi; Zamaneh Kassiri; Shaohua Wang; Gavin Y Oudit
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2020-10-22       Impact factor: 5.187

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