Case Report: Diagnostic challenges in Fabry disease: misinterpreted obstructive hypertrophic cardiomyopathy and the role of enzyme replacement therapy

Fabry disease (FD) is a rare X-linked inherited lysosomal storage disorder. The abnormal accumulation of metabolic substrates induces inflammation and fibrosis in cells, resulting in organ dysfunction. The clinical manifestations of FD are diverse and non-specific. In the present study, we report a case initially treated as obstructive hypertrophic cardiomyopathy for several years, which was finally identified as FD through whole-exome sequencing (WES). The patient, diagnosed with obstructive hypertrophic cardiomyopathy, underwent left ventricular outflow tract surgery before visiting our hospital. WES was proposed by our cardiomyopathy center and, unexpectedly, a mutation [c.595T>C (p.Val199Met)] in exon 4 of the GLA gene was identified. A subsequent analysis of plasma α-galactosidase and globotriaosylsphingosine levels confirmed the diagnosis of FD. Although enzyme replacement therapy (ERT) was initiated immediately after diagnosis, the patient experienced aortic valve damage and left heart enlargement 2 years later. Subsequently, the patient underwent transcatheter aortic valve replacement. This case implies that FD should be considered a potential cause in patients with unexplained left ventricular hypertrophy. Delayed initiation of ERT may compromise its efficacy.

Authors

Institutions

Publication Details

Journal
Frontiers in Cardiovascular Medicine
Published
2024-11-22
DOI
https://doi.org/10.3389/fcvm.2024.1479374
Citations
1
Primary Topic
Lysosomal Storage Disorders Research
Type
article
Field-Weighted Citation Impact
0.33
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Case Report: Diagnostic challenges in Fabry disease: misinterpreted obstructive hypertrophic cardiomyopathy and the role of enzyme replacement therapy

Dinghu Xu, Qin Tao, Mingqiang Ao, Weili Cheng et al.
1 citations
Frontiers in Cardiovascular Medicine
Lysosomal Storage Disorders Research
0.33
article

Case Report: Diagnostic challenges in Fabry disease: misinterpreted obstructive hypertrophic cardiomyopathy and the role of enzyme replacement therapy

Dinghu Xu, Qin Tao, Mingqiang Ao, Weili Cheng, Yuqing Zhang
article en
1 citations

Abstract

Fabry disease (FD) is a rare X-linked inherited lysosomal storage disorder. The abnormal accumulation of metabolic substrates induces inflammation and fibrosis in cells, resulting in organ dysfunction. The clinical manifestations of FD are diverse and non-specific. In the present study, we report a case initially treated as obstructive hypertrophic cardiomyopathy for several years, which was finally identified as FD through whole-exome sequencing (WES). The patient, diagnosed with obstructive hypertrophic cardiomyopathy, underwent left ventricular outflow tract surgery before visiting our hospital. WES was proposed by our cardiomyopathy center and, unexpectedly, a mutation [c.595T>C (p.Val199Met)] in exon 4 of the GLA gene was identified. A subsequent analysis of plasma α-galactosidase and globotriaosylsphingosine levels confirmed the diagnosis of FD. Although enzyme replacement therapy (ERT) was initiated immediately after diagnosis, the patient experienced aortic valve damage and left heart enlargement 2 years later. Subsequently, the patient underwent transcatheter aortic valve replacement. This case implies that FD should be considered a potential cause in patients with unexplained left ventricular hypertrophy. Delayed initiation of ERT may compromise its efficacy.

Frontiers in Cardiovascular MedicineVol. 11
Nanjing Medical University (CN)
Good health and well-being
Openalex Percentile: Top 36%
Lysosomal Storage Disorders Research
0.33
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.

Case Report: Diagnostic challenges in Fabry disease: misinterpreted obstructive hypertrophic cardiomyopathy and the role of enzyme replacement therapy — Dinghu Xu, Qin Tao, et al. · Frontiers in Cardiovascular Medicine (2024) | TGRS Research Map | TGRS