Evidence map›Paper›PMID 36383556›Full record

ArticlePloS one2022

Systematic cascade screening in the Danish Fabry Disease Centre: 20 years of a national single-centre experience.

Grigoris Effraimidis, Åse Krogh Rasmussen, Morten Dunoe, Lis F Hasholt, Flemming Wibrand, Soren S Sorensen, Allan M Lund, Lars Kober, Henning Bundgaard, Puriya D W Yazdanfard and 13 more

Erratum issued Registry-linked trialOpen access · goldAbstract read
In one paragraph

Article in PloS one, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. It is linked to trial NCT06776419 (the Role of cArdiac Inflammation, endoThelial Dysfunction, and FIbrosis in fabrY Disease), which is not on this map. Cited by 6 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed, 1 pooled it
1.3field-weighted citation impact, top 20% 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.

NCT06776419 recruitingnot on this mapstarted 2025, after this paper: background citation

the Role of cArdiac Inflammation, endoThelial Dysfunction, and FIbrosis in fabrY Disease

TypeobservationalSponsorCaroline Michaela KistorpRan2025 to 2031Enrolled54ConditionsFabry Disease, Cardiovascular DiseasesArmsCardiac Magnetic Ressonance Imaging, 82Rubidium-positron emission tomography and computer-tomography
3 · Its place in the literature

Who cites it

6 citing papers in PubMed, 1 synthesis or guideline pooled it, 11 citations in OpenAlex.

  1. Pooled it
  2. Review
  3. Review
  4. Review
  5. Article
  6. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

23 authors at 5 institutions in 2 countries.

Grigoris EffraimidisDepartment of Endocrinology and Metabolism, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0001-7313-8391
Åse Krogh RasmussenDepartment of Endocrinology and Metabolism, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Morten DunoeDepartment of Clinical Genetics, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Lis F HasholtInstitute of Cellular and Molecular Medicine, University of Copenhagen, Copenhagen, Denmark.
Flemming WibrandDepartment of Clinical Genetics, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Soren S SorensenDepartment of Nephrology, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0001-5898-8048
Allan M LundDepartment of Clinical Medicine, Faculty of Health and Clinical Sciences, Copenhagen University, Copenhagen, Denmark.ORCID 0000-0002-6091-7879
Lars KoberDepartment of Clinical Medicine, Faculty of Health and Clinical Sciences, Copenhagen University, Copenhagen, Denmark.ORCID 0000-0002-6635-1466
Henning BundgaardDepartment of Clinical Medicine, Faculty of Health and Clinical Sciences, Copenhagen University, Copenhagen, Denmark.
Puriya D W YazdanfardDepartment of Endocrinology and Metabolism, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Peter OturaiDepartment of Clinical Medicine, Faculty of Health and Clinical Sciences, Copenhagen University, Copenhagen, Denmark.ORCID 0000-0002-9828-9185
Vibeke A LarsenDepartment of Radiology, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Vitor Hugo Fraga de AbreuDepartment of Radiology, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0001-8416-013X
Lotte Hahn EnevoldsenDepartment of Clinical Physiology and Nuclear Medicine, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0001-7325-937X
Tatiana KristensenDepartment of Clinical Physiology and Nuclear Medicine, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Kirsten SvenstrupDepartment of Neurology, Copenhagen Neuromuscular Center, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Margrethe Bastholm BilleDepartment of Clinical Neurophysiology, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Farah ArifDepartment of Ophthalmology, Rigshospitalet-Glostrup (Copenhagen University Hospital), Copenhagen, Denmark.
Mette MogensenDepartment of Dermatology, Bispebjerg Hospital, Copenhagen University Hospitals, Copenhagen, Denmark.ORCID 0000-0001-6479-6034
Mads KlokkerDepartment of Otorhinolaryngology and Head and Neck Surgery and Audiology, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0002-4230-4251
Vibeke BackerDepartment of Clinical Medicine, Faculty of Health and Clinical Sciences, Copenhagen University, Copenhagen, Denmark.
Caroline KistorpDepartment of Endocrinology and Metabolism, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.
Ulla Feldt-RasmussenDepartment of Endocrinology and Metabolism, Rigshospitalet (Copenhagen University Hospital), Copenhagen, Denmark.ORCID 0000-0002-5903-3355
Copenhagen University Hospital · DKUniversity of Copenhagen · DKBispebjerg Hospital · DKGlostrup Hospital · DKUniversity of Thessaly · GR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

The lysosomal storage disorder Fabry disease is caused by deficient or absent activity of the GLA gene enzyme α-galactosidase A. In the present study we present the molecular and biochemical data of the Danish Fabry cohort and report 20 years' (2001-2020) experience in cascade genetic screening at the Danish National Fabry Disease Center. The Danish Fabry cohort consisted of 26 families, 18 index patients (9 males and 9 females, no available data for 8 index-patients) and 97 family members with a pathogenic GLA variant identified by cascade genetic testing (30 males and 67 females). Fourteen patients (5 males and 9 females; mean age of death 47.0 and 64.8 years respectively) died during follow-up. The completeness of the Fabry patient identification in the country has resulted in a cohort of balanced genotypes according to gender (twice number of females compared to males), indicating that the cohort was not biased by referral, and further resulted in earlier diagnosis of the disease by a lower age at diagnosis in family members compared to index-patients (mean age at diagnosis: index-patients 42.2 vs. family members 26.0 years). Six previously unreported disease-causing variants in the GLA gene were discovered. The nationwide screening and registration of Fabry disease families provide a unique possibility to establish a complete cohort of Fabry patients and to advance current knowledge of this inherited rare lysosomal storage disorder.

Indexed as

Fabry Diseasealpha-GalactosidaseDenmarkFemaleGenetic TestingGenotypeHumansMaleMiddle AgedMutationalpha-Galactosidase

Identifiers

PMID36383556
PMCPMC9668118
OpenAlexW4309138927

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Registered trials

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.