Evidence map›Paper›PMID 38383316›Full record

ArticleBMC nephrology2024

Case report: enzyme replacement therapy for Fabry disease presenting with proteinuria and ventricular septal thickening.

Zewei Chen, Bo Yin, Juan Jiao, Tianyang Ye

Open access · goldAbstract readCase Reports
In one paragraph

Article in BMC nephrology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.3field-weighted citation impact, top 44% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 1 citations in OpenAlex.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors at 1 institution in 1 country.

Zewei ChenDepartment of Nephrology, The First Navy Hospital of Southern Theater Command, Zhanjiang, Guangdong, China.
Bo YinDepartment of Internal Medicine, The First Navy Hospital of Southern Theater Command, Zhanjiang, Guangdong, China.
Juan JiaoDepartment of Internal Medicine, The First Navy Hospital of Southern Theater Command, Zhanjiang, Guangdong, China.
Tianyang YeDepartment of Internal Medicine, The First Navy Hospital of Southern Theater Command, Zhanjiang, Guangdong, China. yetianyang1201@163.com.
PLA Navy General Hospital · CN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Fabry disease (FD) is an uncommon, X-linked, lysosomal storage disease that causes defects in the glycosphingolipid metabolic pathway due to deficient or absent lysosomal α-galactosidase (α-Gal A) activity. This leads to the accumulation of globotriaosylceramide (GL-3) within lysosomes in a wide range of cells, including endothelial, cardiac, renal, and corneal cells, and consequently, the progressive appearance of clinical symptoms in target organs. Enzyme replacement therapy (ERT), which involves the exogenous supplementation of α-Gal A enzyme and has been successfully administered for treating FD.Here, we report a case of a 37-year-old male with complaints of recurrent proteinuria and ventricular septal thickening. A renal biopsy revealed vacuolization and foamy changes in podocytes, and the presence of myelin-like bodies and zebra bodies. The white blood cell α-Gal A activity was very low, while the Lyso-GL-3 level was high. Additionally, genetic analysis revealed a gene variant c.902G > A p. Arg301Gln. The patient was diagnosed with FD, and subsequently received intravenous ERT with a dose of Agalsidase α (0.2 mg/kg, 17.5 mg every 2 weeks). Currently, the values of proteinuria and ventricular septum thickness remain stable during the 6-month follow-up. Initiating ERT at an early age can effectively decrease the deposition of GL-3, attenuate the progressive clinical manifestations of FD, and provide greater long-term benefits.

Indexed as

Fabry DiseaseAdultalpha-GalactosidaseEnzyme Replacement TherapyHeart VentriclesHumansKidneyMaleProteinuriaalpha-GalactosidaseAgalsidase αEnzyme replacement therapyFabry diseaseFabry nephropathyGenetic testing

Identifiers

PMID38383316
PMCPMC10882756
OpenAlexW4391994097

What OpenQuestion holds

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Registered trials

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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.