Evidence map›Paper›PMID 36178639›Full record

ArticleMetabolic brain disease2022

Late-onset Fabry disease due to a new (p.Pro380Leu) pathogenic variant of GLA Gene.

Vittoria Cianci, Angelo Pascarella, Sara Gasparini, Vincenzo Donadio, Rocco Liguori, Alex Incensi, Carmelo Massimiliano Rao, Claudio Franzutti, Giuseppe Scappatura, Umberto Aguglia and 1 more

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In one paragraph

Article in Metabolic brain disease, 2022. 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
0.2field-weighted citation impact, top 46% 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, 2 citations in OpenAlex.

  1. Article
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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

11 authors at 3 institutions in 2 countries.

Vittoria CianciRegional Epilepsy Centre, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy.ORCID 0000-0002-2352-5710
Angelo PascarellaRegional Epilepsy Centre, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy.ORCID 0000-0002-0085-9494
Sara GaspariniRegional Epilepsy Centre, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy.ORCID 0000-0001-6779-435X
Vincenzo DonadioComplex Operational Unit Clinica Neurologica, IRCCS Institute of Neurological Sciences of Bologna, Bologna, Italy.ORCID 0000-0002-1290-7318
Rocco LiguoriComplex Operational Unit Clinica Neurologica, IRCCS Institute of Neurological Sciences of Bologna, Bologna, Italy.ORCID 0000-0002-1815-1013
Alex IncensiComplex Operational Unit Clinica Neurologica, IRCCS Institute of Neurological Sciences of Bologna, Bologna, Italy.ORCID 0000-0003-2163-5403
Carmelo Massimiliano RaoCardiology Department, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy.
Claudio FranzuttiRadiology Department, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio, Calabria, Italy.
Giuseppe ScappaturaRadiology Department, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio, Calabria, Italy.
Umberto AgugliaRegional Epilepsy Centre, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy.ORCID 0000-0002-4574-2951
Edoardo FerlazzoRegional Epilepsy Centre, Great Metropolitan Hospital "Bianchi-Melacrino-Morelli", Reggio Calabria, Italy. ferlazzo@unicz.it.ORCID 0000-0003-3531-8337
Magna Graecia University · ITAzienda ospedaliera "Bianchi-Melacrino-Morelli" · ITInstitute of Neurological Sciences · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Fabry disease is a rare X-linked lysosomal storage disorder due to pathogenic variants of the galactosidase alpha (GLA) gene, leading to a deficiency of alpha-galactosidase A. The inadequate enzymatic activity leads to progressive glycosphingolipids accumulation within tissues and subsequent multi-systemic dysfunction, with predominant involvement of heart, kidney, and nervous system. Two subtypes are recognized: the classic type and the late-onset type. We here describe the clinical characteristics of a patient with late-onset Fabry disease carrying a not previously identified GLA gene variant. This 50-year-old man came to hospital because of an acute ischemic stroke. He also complained of acroparesthesia and had angiokeratomas in the nape and the back. Blood alpha-galactosidase A activity was low, plasmatic lyso-Gb3 level was borderline, cardiac MRI showed cardiac fibrosis, brain MRI documented cerebrovascular disease, and skin biopsy revealed small fiber neuropathy without globotriaosylceramide-3 skin deposits. Genetic study by means of targeted next-generation sequencing analysis disclosed a missense substitution c.1139C>T (p.Pro380Leu) in the GLA gene. We suggest that this novel variant should be considered as pathogenic and associated with a late-onset variant of Fabry disease with a predominant neurological phenotype.

Indexed as

Fabry DiseaseIschemic Strokealpha-GalactosidaseGalactosidasesHumansMaleMutationPhenotypealpha-GalactosidaseGalactosidasesGLA pathogenic variantLate-onset Fabry DiseaseSmall fiber neuropathyStroke

Identifiers

PMID36178639
OpenAlexW4298086620

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.