Evidence map›Paper›PMID 33156427›Full record

ArticleMetabolic brain disease2021

Fabry disease: GLA deletion alters a canonical splice site in a family with neuropsychiatric manifestations.

Patrícia Varela, Gerson Carvalho, Renan Paulo Martin, João Bosco Pesquero

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In one paragraph

Article in Metabolic brain disease, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed, 1 pooled it
0.7field-weighted citation impact, top 29% 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, 1 synthesis or guideline pooled it, 7 citations in OpenAlex.

  1. Pooled it
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  3. Review
  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

4 authors at 3 institutions in 2 countries.

Patrícia Varela *Center for Research and Molecular Diagnostic of Genetic Diseases - Department of Biophysics, Universidade Federal de São Paulo, Rua Pedro de Toledo, 669 - 9o andar, São Paulo, 04039-032, Brazil.
Gerson Carvalho *Medical Genetics Unit, Hospital de Apoio de Brasília, Brasília, DF, Brazil.
Renan Paulo MartinCenter for Research and Molecular Diagnostic of Genetic Diseases - Department of Biophysics, Universidade Federal de São Paulo, Rua Pedro de Toledo, 669 - 9o andar, São Paulo, 04039-032, Brazil.
João Bosco PesqueroCenter for Research and Molecular Diagnostic of Genetic Diseases - Department of Biophysics, Universidade Federal de São Paulo, Rua Pedro de Toledo, 669 - 9o andar, São Paulo, 04039-032, Brazil. jbpesquero@gmail.com.ORCID 0000-0002-4507-632X
Universidade Federal de São Paulo · BRHospital Universitário de Brasília · BRJohns Hopkins University · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Fabry disease (FD) is a rare X-linked glycosphingolipidosis caused by mutations in GLA, a gene responsible for encoding α-galactosidase A, an enzyme required for degradation of glycosphingolipids, mainly globotriaosylceramide (Gb3) in all cells of the body. FD patients present a broad spectrum of clinical phenotype and many symptoms are shared with other diseases, making diagnosis challenging. Here we describe a novel GLA variant located in the 5' splice site of the intron 3, in four members of a family with neuropsychiatric symptoms. Analysis of the RNA showed the variant promotes alteration of the wild type donor site, affecting splicing and producing two aberrant transcripts. The functional characterization showed absence of enzymatic activity in cells expressing both transcripts, confirming their pathogenicity. The family presents mild signs of FD, as angiokeratoma, cornea verticillata, acroparesthesia, tinnitus, vertigo, as well as accumulation of plasma lyso-Gb3 and urinary Gb3. Interestingly, the man and two women present psychiatric symptoms, as depression or schizophrenia. Although psychiatric illnesses, especially depression, are frequently reported in patients with FD and studies have shown that the hippocampus is an affected brain structure in these patients, it is not clear whether the Gb3 accumulation in the brain is responsible for these symptoms or they are secondary. Therefore, new studies are needed to understand whether the accumulation of Gb3 could produce neuronal alterations leading to psychiatric symptoms.

Indexed as

MutationAdolescentalpha-GalactosidaseBrainFabry DiseaseFemaleHumansMaleMiddle AgedPhenotypeYoung Adultalpha-GalactosidaseGLA protein, humanDepressionFabry diseaseNeurological manifestationsPsychiatric symptoms, GLA geneSchizophreniaSplice site mutationα-Galactosidase A

Identifiers

PMID33156427
OpenAlexW3096621921

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.