2024 Update of the TSOC Expert Consensus of Fabry Disease.

As an X-linked inherited lysosomal storage disease that is caused by α-galactosidase A gene variants resulting in progressive accumulation of pathogenic glycosphingolipid (Gb3) accumulation in multiple tissues and organs, Fabry disease (FD) can be classified into classic or late-onset phenotypes. In classic phenotype patients, α-galactosidase A activity is absent or severely reduced, resulting in a more progressive disease course with multi-systemic involvement. Conversely, late-onset phenotype, often with missense variants (e.g., IVS4+919G>A) in Taiwan, may present with a more chronic clinical course with predominant cardiac involvement (cardiac subtype), as they tend to have residual enzyme activity, remaining asymptomatic or clinically silent during childhood and adolescence. In either form, cardiac hypertrophy remains the most common feature of cardiac involvement, potentially leading to myocardial fibrosis, arrhythmias, and heart failure. Diagnosis is established through α-galactosidase enzyme activity assessment or biomarker analyisis (globotriaosylsphingosine, Lyso-Gb3), advanced imaging modalities (echocardiography and cardiac magnetic resonance imaging), and genotyping to differentiate FD from other cardiomyopathy. Successful therapeutic response relies on early recognition and by disease awareness from typical features in classic phenotype and cardiac red flags in cardiac variants for timely therapeutic interventions. Recent advances in pharmacological approach including enzyme replacement therapy (agalsidase alfa or beta), oral chaperone therapy (migalastat), and substrate reduction therapy (venglustat) aim to prevent from irreversible organ damage. Genotype- and gender-based monitoring of treatment effects through biomarker (Lyso-Gb3), renal assessment, and cardiac responses using advanced imaging modalities are key steps to optimizing patient care in FD.

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Publication Details

Journal
PubMed
Published
2024-09-01
DOI
https://doi.org/10.6515/acs.202409_40(5).20240731a
Citations
3
Primary Topic
Lysosomal Storage Disorders Research
Type
article
Field-Weighted Citation Impact
0.98

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article

2024 Update of the TSOC Expert Consensus of Fabry Disease.

Hao‐Chih Chang, Wen‐Jone Chen, Wei‐Ting Chang, Chun‐Yao Huang et al.
3 citations
PubMed
Lysosomal Storage Disorders Research
0.98
article

2024 Update of the TSOC Expert Consensus of Fabry Disease.

Hao‐Chih Chang, Wen‐Jone Chen, Wei‐Ting Chang, Chun‐Yao Huang, Wen-Hwa Wang, Jyh‐Ming Jimmy Juang, Chun‐Yuan Chu, Kuo‐Tzu Sung, Chung-Lieh Hung, Chengshu Wang, Chih‐Hung Lai, Chia‐Hsiu Chang, Ling Kuo, Tzung‐Dau Wang, Yung‐Ta Kao, Yen‐Wen Wu, Hsinyu Tseng, Heng-Hsu Lin, Po-Sheng Chen, Chun-Li Wang, Chih‐Chan Lin, Wen‐Chung Yu
article en
3 citations

Abstract

As an X-linked inherited lysosomal storage disease that is caused by α-galactosidase A gene variants resulting in progressive accumulation of pathogenic glycosphingolipid (Gb3) accumulation in multiple tissues and organs, Fabry disease (FD) can be classified into classic or late-onset phenotypes. In classic phenotype patients, α-galactosidase A activity is absent or severely reduced, resulting in a more progressive disease course with multi-systemic involvement. Conversely, late-onset phenotype, often with missense variants (e.g., IVS4+919G>A) in Taiwan, may present with a more chronic clinical course with predominant cardiac involvement (cardiac subtype), as they tend to have residual enzyme activity, remaining asymptomatic or clinically silent during childhood and adolescence. In either form, cardiac hypertrophy remains the most common feature of cardiac involvement, potentially leading to myocardial fibrosis, arrhythmias, and heart failure. Diagnosis is established through α-galactosidase enzyme activity assessment or biomarker analyisis (globotriaosylsphingosine, Lyso-Gb3), advanced imaging modalities (echocardiography and cardiac magnetic resonance imaging), and genotyping to differentiate FD from other cardiomyopathy. Successful therapeutic response relies on early recognition and by disease awareness from typical features in classic phenotype and cardiac red flags in cardiac variants for timely therapeutic interventions. Recent advances in pharmacological approach including enzyme replacement therapy (agalsidase alfa or beta), oral chaperone therapy (migalastat), and substrate reduction therapy (venglustat) aim to prevent from irreversible organ damage. Genotype- and gender-based monitoring of treatment effects through biomarker (Lyso-Gb3), renal assessment, and cardiac responses using advanced imaging modalities are key steps to optimizing patient care in FD.

PubMedVol. 40(5)
National Yang Ming Chiao Tung University (TW), Kaohsiung Medical University (TW), National Health Research Institutes (TW), Chang Gung University (TW), Kaohsiung Medical University Chung-Ho Memorial Hospital (TW), Mackay Memorial Hospital (TW), Taipei Veterans General Hospital (TW), Chang Gung Memorial Hospital (TW), Mackay Medical University (TW), Taichung Veterans General Hospital (TW), Chi Mei Medical Center (TW), Kaohsiung Veterans General Hospital (TW), National Cheng Kung University Hospital (TW), Institute of Biomedical Science (GB), Taipei Medical University (TW), Yuan Ze University (TW)
National Science and Technology Council
Openalex Percentile: Top 23%
Lysosomal Storage Disorders Research
0.98
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