Evidence map›Paper›PMID 31285529›Full record

ArticleEuropean journal of human genetics : EJHG2019

Report on three additional patients and genotype-phenotype correlation in SLC25A22-related disorders group.

Camille Lemattre, Marion Imbert-Bouteille, Vincent Gatinois, Paule Benit, Elodie Sanchez, Thomas Guignard, Frédéric Tran Mau-Them, Emmanuelle Haquet, François Rivier, Emilie Carme and 13 more

Abstract read
In one paragraph

Article in European journal of human genetics : EJHG, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
–field-weighted citation impact
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

8 citing papers in PubMed.

  1. Article
  2. Review
  3. Article
  4. Review
  5. Review
  6. Review
  7. Amino Acid Transport Defects in Human Inherited Metabolic Disorders.International journal of molecular sciences · 2019
    Review
  8. Review
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

23 authors.

Camille LemattreDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Marion Imbert-BouteilleDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Vincent GatinoisDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Paule BenitInserm UMR 1141 - PROTECT, Hôpital Robert Debré, 48, Boulevard Sérurier, 75019, Paris, France.
Elodie SanchezDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Thomas GuignardDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.ORCID http://orcid.org/0000-0001-7228-3925
Frédéric Tran Mau-ThemDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Emmanuelle HaquetDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
François RivierService de Neuropédiatrie, Université de Montpellier, CHU de Montpellier, France.
Emilie CarmeService de Neuropédiatrie, Université de Montpellier, CHU de Montpellier, France.
Agathe RoubertieService de Neuropédiatrie, Université de Montpellier, CHU de Montpellier, France.
Anne BolandCentre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, F-91057, Evry, France.
Doris LechnerCentre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, F-91057, Evry, France.
Vincent MeyerCentre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, F-91057, Evry, France.
Julien ThevenonDépartement de Génétique et Procréation, Hôpital Couple-Enfant, Université de Grenoble, CHU de Grenoble, France.ORCID http://orcid.org/0000-0001-9271-3961
Yannis DuffourdLaboratoire de Génétique Moléculaire, Plateau Technique de Biologie, Université de Bourgogne, CHRU Dijon, France.
Jean-Baptiste RivièreLaboratoire de Génétique Moléculaire, Plateau Technique de Biologie, Université de Bourgogne, CHRU Dijon, France.ORCID http://orcid.org/0000-0001-9144-4210
Jean-François DeleuzeCentre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, F-91057, Evry, France.
Constance WellsDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
Florence MolinariINSERM, INMED, Aix Marseille University, Marseille, France.
Pierre RustinInserm UMR 1141 - PROTECT, Hôpital Robert Debré, 48, Boulevard Sérurier, 75019, Paris, France.
Patricia BlanchetDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France.
David GenevièveDépartement de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du Développement et Syndromes Malformatifs, Université de Montpellier, CHU de Montpellier, France. d-genevieve@chu-montpellier.fr.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Early infantile epileptic encephalopathy (EIEE) is a heterogeneous group of severe forms of age-related developmental and epileptic encephalopathies with onset during the first weeks or months of life. The interictal electroencephalogram (EEG) shows a "suppression burst" (SB) pattern. The prognosis is usually poor and most children die within the first two years or survive with very severe intellectual disabilities. EIEE type 3 is caused by variants affecting function, in SLC25A22, which is also responsible for epilepsy of infancy with migrating focal seizures (EIMFS). We report a family with a less severe phenotype of EIEE type 3. We performed exome sequencing and identified two unreported variants in SLC25A22 in the compound heterozygous state: NM_024698.4: c.[813_814delTG];[818 G>A] (p.[Ala272Glnfs*144];[Arg273Lys]). Functional studies in cultured skin fibroblasts from a patient showed that glutamate oxidation was strongly defective, based on a literature review. We clustered the 18 published patients (including those from this family) into three groups according to the severity of the SLC25A22-related disorders. In an attempt to identify genotype-phenotype correlations, we compared the variants according to the location depending on the protein domains. We observed that patients with two variants located in helical transmembrane domains presented a severe phenotype, whereas patients with at least one variant outside helical transmembrane domains presented a milder phenotype. These data are suggestive of a continuum of disorders related to SLC25A22 that could be called SLC25A22-related disorders. This might be a first clue to enable geneticists to outline a prognosis based on genetic molecular data regarding the SLC25A22 gene.

Indexed as

AdolescentBase SequenceChildChild, PreschoolElectroencephalographyExomeFemaleFibroblastsGenetic Association StudiesGenetic Predisposition to DiseaseHumansMaleMitochondrial Membrane Transport ProteinsPedigreePhenotypeSkinMitochondrial Membrane Transport ProteinsSLC25A22 protein, human

Identifiers

PMID31285529
PMCPMC6871179

What OpenQuestion holds

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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.