Evidence map›Paper›PMID 42608886›Full record

ArticleThe American journal of case reports2026

Long-Term Cardiac Remodeling During Migalastat Therapy in a Heterozygote Woman With Fabry Disease (N224S): A 9 Year Case Report.

Clement Tan, Vaikunthan Thanabalasingam, Chaminda Sella Kapu, Matthew Hiskens, Zhihua Zhang

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Article in The American journal of case reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

5 authors.

Clement TanDepartment of Cardiology, Coronary Care Unit & Cardiac Catheterisation Laboratory, Mackay Base Hospital, Mackay, Queensland, Australia.
Vaikunthan ThanabalasingamDepartment of Cardiology, Coronary Care Unit & Cardiac Catheterisation Laboratory, Mackay Base Hospital, Mackay, Queensland, Australia.
Chaminda Sella KapuDepartment of Cardiology, Coronary Care Unit & Cardiac Catheterisation Laboratory, Mackay Base Hospital, Mackay, Queensland, Australia.
Matthew HiskensMackay Institute of Research and Innovation, Mackay Base Hospital, Mackay, Queensland, Australia.
Zhihua ZhangDepartment of Cardiology, Coronary Care Unit & Cardiac Catheterisation Laboratory, Mackay Base Hospital, Mackay, Queensland, Australia.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND Fabry disease (FD) is a rare X‑linked lysosomal disorder in which long‑term cardiac outcomes, particularly in heterozygous females, remain incompletely defined. The N224S alpha-galactosidase A gene (GLA) variant is recognized as pathogenic, yet its cardiac phenotype and response to prolonged migalastat therapy are not well characterized. Although clinical trials and observational studies have reported variable cardiac responses to migalastat, real‑world longitudinal data beyond 5 years remain limited. Understanding long‑term structural and functional changes is especially important in patients with pre‑existing cardiac involvement, where distinguishing favorable remodeling from fibrosis‑related progression can be challenging. CASE REPORT We describe a 9‑year follow‑up of a 71‑year‑old heterozygous woman with FD due to the N224S mutation treated with migalastat. Serial transthoracic echocardiography demonstrated a sustained reduction in left ventricular mass index (LVMI) over long‑term therapy. Left ventricular systolic function remained preserved throughout follow‑up, and there was no evidence of progressive structural deterioration. Available Doppler‑derived diastolic indices, including a gradual decline in early diastolic mitral inflow velocity to early diastolic mitral annular velocity (E/e'), suggested improving left ventricular filling pressures over time. Although LVMI showed minor year‑to‑year variability, these changes were consistent with expected technical or physiological variation and did not alter the clear long‑term downward trend. CONCLUSIONS This case provides real‑world evidence of favorable long‑term cardiac remodeling during migalastat therapy in a heterozygous woman with the N224S variant. The sustained reduction in LVMI, preserved systolic function, and improving diastolic indices together indicate a durable cardiac response and extend the limited longitudinal data available for variant‑specific outcomes in FD.

Indexed as

1-DeoxynojirimycinEnzyme InhibitorsFabry DiseaseVentricular RemodelingAgedalpha-GalactosidaseEchocardiographyFemaleHeterozygoteHumans1-Deoxynojirimycinalpha-GalactosidaseEnzyme Inhibitorsmigalastat

Identifiers

PMID42608886
PMCPMC13499433

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.