Evidence map›Paper›PMID 30341396›Full record

ArticleScientific reports2018

A novel homozygous mutation in GAD1 gene described in a schizophrenic patient impairs activity and dimerization of GAD67 enzyme.

Chiara Magri, Edoardo Giacopuzzi, Luca La Via, Daniela Bonini, Viola Ravasio, Mohammed E A Elhussiny, Flavia Orizio, Fabrizio Gangemi, Paolo Valsecchi, Roberto Bresciani and 3 more

Erratum issuedOpen access · goldAbstract read
In one paragraph

Article in Scientific reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
4.4field-weighted citation impact, top 6% 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

16 citing papers in PubMed, 26 citations in OpenAlex.

  1. Review
  2. Article
  3. Review
  4. Sigma-1 Receptor Activation by Fluvoxamine Ameliorates ER Stress, Synaptic Dysfunction and Behavioral Deficits in a Ketamine Model of Schizophrenia.Journal of neuroimmune pharmacology : the official journal of the Society on NeuroImmune Pharmacology · 2025
    Article
  5. Review
  6. Review
  7. The Healthy and Diseased Retina Seen through Neuron-Glia Interactions.International journal of molecular sciences · 2024
    Review
  8. Beyond the γ-aminobutyric acid hypothesis of schizophrenia.Frontiers in cellular neuroscience · 2023
    Review
  9. Article
  10. Review
  11. Article
  12. Article
  13. Article
  14. Article
  15. Neurological Syndromes Associated with Anti-GAD Antibodies.International journal of molecular sciences · 2020
    Review
  16. Rare compound heterozygous missenseNeuropsychiatric disease and treatment · 2019
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

13 authors at 3 institutions in 1 country.

Chiara MagriUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy. chiara.magri@unibs.it.ORCID http://orcid.org/0000-0001-9933-8666
Edoardo GiacopuzziUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.ORCID http://orcid.org/0000-0002-5692-366X
Luca La ViaUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Daniela BoniniUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Viola RavasioUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Mohammed E A ElhussinyUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Flavia OrizioUnit of Biotechnology, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Fabrizio GangemiUnit of Physics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
Paolo ValsecchiNeuroscience Section, Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.
Roberto BrescianiUnit of Biotechnology, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.ORCID http://orcid.org/0000-0002-7881-7550
Alessandro BarbonUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.ORCID http://orcid.org/0000-0002-1127-7764
Antonio VitaNeuroscience Section, Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.
Massimo GennarelliUnit of Biology and Genetics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy.
University of Brescia · ITAzienda Socio Sanitaria Territoriale degli Spedali Civili di Brescia · ITCentro San Giovanni di Dio Fatebenefratelli · IT

Funding

A Large-Scale Schizophrenia Association Study in SwedenR01MH077139 · NIMH · UNIV OF NORTH CAROLINA CHAPEL HILL · PI SULLIVAN, PATRICK F · 2007 to 2018
$9.9M
Brain and Behavior Research Foundation (Brain & Behavior Research Foundation) 20751NIMH NIH HHS R01 MH077139
6 · The paper itself

Abstract

Recently, by whole exome sequencing of schizophrenia (SCZ) patients, we identified a subject that was homozygous for a novel missense substitution (c.391 A > G) in the glutamate acid decarboxylase 1 (GAD1) gene. GAD1 encodes for GAD67 enzyme, catalyzing the production of gamma-aminobutyric acid (GABA) from L-glutamic acid. Here, we studied the impact of this mutation on GAD67 activity, dimerization and subcellular localization. Biochemical assay revealed that c.391 A > G reduces GAD67 enzymatic activity by ~30%, probably due to the impaired homodimerization of homozygous mutants as highlighted by proximity ligation assays. The mutational screening of 120 genes of the "GABAergic system" in a cohort of 4,225 SCZ cases and 5,834 controls (dbGaP: phs000473.v1.p2), did not identify other cases that were homozygous for ultra-rare variants in GAD1, but highlighted an increased frequency of cases that were homozygous for rare variants in genes of the GABA system (SCZ: 0.14% vs. Controls: 0.00%; p-value = 0.0055). In conclusion, this study demonstrates the functional impact of c.391 A > G variant and its biological effect makes it a good candidate as risk variant for SCZ. This study also supports an involvement of ultra-rare variants in GABAergic genes in the etiopathogenesis of SCZ.

Indexed as

Mutation, MissenseProtein MultimerizationAdultgamma-Aminobutyric AcidGlutamate DecarboxylaseGlutamic AcidHomozygoteHumansMaleMutant ProteinsSchizophreniagamma-Aminobutyric AcidGlutamate Decarboxylaseglutamate decarboxylase 1Glutamic AcidMutant Proteins

Identifiers

PMID30341396
PMCPMC6195539
OpenAlexW2896268215

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.