Evidence map›Paper›PMID 34354036›Full record

ArticleThe American journal of case reports2021

First 2 Fabry Cases with Novel Mutation and Their Associated Clusters in Malaysia.

Andy Sing Ong Tang, Qi Ying Wong, Ingrid Pao Lin Ting, Panting Selvesten, Siaw Tze Yeo, Lee Ping Chew, Tem Lom Fam, Clare Hui Hong Tan

Open access · hybridAbstract readCase Reports
In one paragraph

Article in The American journal of case reports, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact, top 91% 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

2 citing papers in PubMed, 1 citations in OpenAlex.

  1. Article
  2. 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

8 authors at 2 institutions in 1 country.

Andy Sing Ong TangDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.
Qi Ying WongDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.
Ingrid Pao Lin TingDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.ORCID 0000-0002-6476-8073
Panting SelvestenDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.
Siaw Tze YeoDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.
Lee Ping ChewHaematology Unit, Department of Internal Medicine, Sarawak General Hospital, Ministry of Health, Kuching, Sarawak, Malaysia.
Tem Lom FamDepartment of Internal Medicine, Miri Hospital, Ministry of Health, Miri, Sarawak, Malaysia.
Clare Hui Hong TanNephrology Unit, Department of Internal Medicine, Sarawak General Hospital, Ministry of Health, Kuching, Sarawak, Malaysia.
Ministry of Health · MYSarawak General Hospital · MY

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND No cases of Fabry disease (FD) have been reported thus far in Malaysia. We aimed to report the demographic characteristics, clinical manifestations, molecular results, and treatment outcomes of 2 FD cases. This study was a retrospective review of 2 family clusters of FD on follow-up in Sarawak, Malaysia. CASE REPORT Two index patients were confirmed to have FD. Index patient 1, who had nephrotic-range proteinuria and cornea verticillata, carried a variant within exon 4 of the GLA gene: c.610 T>C (p.Trp204Arg). Agalsidase beta (Fabrazyme®) enzyme replacement therapy was initiated, with the absence of neutralizing antibody after 24 months. No hypersensitivity or adverse reactions were reported. The patient's proteinuria and renal function remained stable. Other family members who carried the same mutation were asymptomatic. Index patient 2, who had residual activity of alpha-galactosidase A and a normal globotriaosylsphingosine level, carried a novel GLA mutation of c.548-5T>A. He was diagnosed with end-stage renal disease on regular dialysis and had nonspecific headache with 1 episode of seizure a few years prior to FD genetic screening. One brother had chronic neuropathic pain but refused further investigations. Other family members who had the same mutation were asymptomatic. This mutation has never been reported in literature, and its pathogenicity warrants further studies. CONCLUSIONS It is of utmost importance to increase awareness of FD among clinicians, so that appropriate screening may be done to determine its true prevalence and prompt treatment can be initiated early.

Indexed as

Fabry DiseaseKidney Failure, Chronicalpha-GalactosidaseHumansMalaysiaMaleMutationRetrospective Studiesalpha-Galactosidase

Identifiers

PMID34354036
PMCPMC8351246
OpenAlexW3174390502

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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Registered trials

None linked

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.