Evidence map›Paper›PMID 34441839›Full record

ArticleJournal of clinical medicine2021

Detailed Phenotype of GLA Variants Identified by the Nationwide Neurological Screening of Stroke Patients in the Czech Republic.

Petra Reková, Gabriela Dostálová, David Kemlink, Jaroslava Paulasová Schwabová, Zora Dubská, Manuela Vaneckova, Martin Mašek, Ondřej Kodet, Helena Poupětová, Stella Mazurová and 7 more

Open access · goldAbstract read
In one paragraph

Article in Journal of clinical medicine, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
0.6field-weighted citation impact, top 34% 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

4 citing papers in PubMed, 7 citations in OpenAlex.

  1. Article
  2. Missed diagnosis of Fabry disease: should we screen patients with multiple sclerosis?Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024
    Observational
  3. Article
  4. 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

17 authors at 5 institutions in 1 country.

Petra RekováDepartment of Neurology and Centre of Clinical Neuroscience, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.ORCID 0000-0001-9881-4982
Gabriela Dostálová2nd Department of Medicine-Department of Cardiovascular Medicine, First Faculty of Medicine, Charles University, 128 08 Prague, Czech Republic.ORCID 0000-0003-4809-1274
David KemlinkDepartment of Neurology and Centre of Clinical Neuroscience, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.ORCID 0000-0003-1398-5796
Jaroslava Paulasová SchwabováDepartment of Neurology, Second Faculty of Medicine, Charles University and Motol University Hospital in Prague, 150 06 Prague, Czech Republic.
Zora DubskáDepartment of Ophthalmology, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.
Manuela VaneckovaDepartment of Radiology, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.
Martin MašekDepartment of Radiology, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.
Ondřej KodetDepartment of Dermatovenerology, First Faculty of Medicine, Charles University and General University Hospital in Prague, 128 08 Prague, Czech Republic.ORCID 0000-0002-1382-2535
Helena PoupětováDepartment of Paediatrics and Inherited Metabolic Disorders, First Faculty of Medicine, University and General University Hospital in Prague, 128 08 Prague, Czech Republic.
Stella MazurováDepartment of Paediatrics and Inherited Metabolic Disorders, First Faculty of Medicine, University and General University Hospital in Prague, 128 08 Prague, Czech Republic.
Aneta RajdovaDepartment of Neurology, Faculty of Medicine, Masaryk University and University Hospital Brno, 625 00 Brno, Czech Republic.
Eva VlckovaDepartment of Neurology, Faculty of Medicine, Masaryk University and University Hospital Brno, 625 00 Brno, Czech Republic.ORCID 0000-0003-2322-5539
Alena TáboříkováDepartment of Neurology and Stroke Centre, Country Hospital Chomutov, 430 12 Chomutov, Czech Republic.
Štěpánka FafejtováDepartment of Neurology and Stroke Centre, Hospital Karlovy Vary, 360 01 Karlovy Vary, Czech Republic.
Miroslava NevsimalovaDepartment of Neurology, Hospital Ceske Budejovice, 370 01 České Budějovice, Czech Republic.
Aleš Linhart2nd Department of Medicine-Department of Cardiovascular Medicine, First Faculty of Medicine, Charles University, 128 08 Prague, Czech Republic.
Aleš TomekDepartment of Neurology, Second Faculty of Medicine, Charles University and Motol University Hospital in Prague, 150 06 Prague, Czech Republic.
Charles University · CZGeneral University Hospital in Prague · CZMasaryk University · CZMuzeum Karlovy Vary · CZNemocnice České Budějovice · CZ

Funding

Ministerstvo Zdravotnictví Ceské Republiky MZCZ-DRO-VFN64165
6 · The paper itself

Abstract

Fabry disease (FD) is a rare X-linked disorder of glycosphingolipid metabolism caused by pathogenic variants within the alpha-galactosidase A (GLA) gene, often leading to neurological manifestations including stroke. Multiple screening programs seeking GLA variants among stroke survivors lacked detailed phenotype description, making the interpretation of the detected variant's pathogenicity difficult. Here, we describe detailed clinical characteristics of GLA variant carriers identified by a nationwide stroke screening program in the Czech Republic. A total of 23 individuals with 8 different GLA variants were included in the study. A comprehensive diagnostic workup was performed by a team of FD specialists. The investigation led to the suggestion of phenotype reclassification for the G325S mutation from late-onset to classical. A novel variant R30K was found and was classified as a variant of unknown significance (VUS). The typical manifestation in our FD patients was a stroke occurring in the posterior circulation with an accompanying pathological finding in the cerebrospinal fluid. Moreover, we confirmed that cornea verticillata is typically associated with classical variants. Our findings underline the importance of detailed phenotype description and data sharing in the correct identification of pathogenicity of gene variants detected by high-risk-population screening programs.

Indexed as

data sharingFabry diseaseGLA gene variantsphenotypescreening programsstroke

Identifiers

PMID34441839
PMCPMC8396867
OpenAlexW3191953550

What OpenQuestion holds

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LicenceCC BY
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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.