Human genetics · 2011
Truncating mutations in NRXN2 and NRXN1 in autism spectrum disorders and schizophrenia.
Сравнительное исследованиеСтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Gauthier Julie, Siddiqui Tabrez J, Huashan Peng, Yokomaku Daisaku, Hamdan Fadi F, Champagne Nathalie, Lapointe Mathieu, Spiegelman Dan, Noreau Anne, Lafrenière Ronald G, Fathalli Ferid, Joober Ridha, Krebs Marie-Odile, DeLisi Lynn E, Mottron Laurent, Fombonne Eric, Michaud Jacques L, Drapeau Pierre, Carbonetto Salvatore, Craig Ann Marie, Rouleau Guy A
Аннотация
Growing genetic evidence is converging in favor of common pathogenic mechanisms for autism spectrum disorders (ASD), intellectual disability (ID or mental retardation) and schizophrenia (SCZ), three neurodevelopmental disorders affecting cognition and behavior. Copy number variations and deleterious mutations in synaptic organizing proteins including NRXN1 have been associated with these neurodevelopmental disorders, but no such associations have been reported for NRXN2 or NRXN3. From resequencing the three neurexin genes in individuals affected by ASD (n = 142), SCZ (n = 143) or non-syndromic ID (n = 94), we identified a truncating mutation in NRXN2 in a patient with ASD inherited from a father with severe language delay and family history of SCZ. We also identified a de novo truncating mutation in NRXN1 in a patient with SCZ, and other potential pathogenic ASD mutations. These truncating mutations result in proteins that fail to promote synaptic differentiation in neuron coculture and fail to bind either of the established postsynaptic binding partners LRRTM2 or NLGN2 in cell binding assays. Our findings link NRXN2 disruption to the pathogenesis of ASD for the first time and further strengthen the involvement of NRXN1 in SCZ, supporting the notion of a common genetic mechanism in these disorders.
Предметные рубрики MeSH
Amino Acid SequenceAnimalsCOS CellsCalcium-Binding ProteinsCase-Control StudiesCell Adhesion Molecules, NeuronalCells, CulturedChildChild Development Disorders, PervasiveChlorocebus aethiopsCohort StudiesFemaleGene DosageGenetic Predisposition to DiseaseHumansLanguage Development DisordersMaleMembrane ProteinsMiceMolecular Sequence DataMutationNerve Tissue ProteinsNeural Cell Adhesion MoleculesNeuronsPedigreeSchizophreniaSequence Homology, Amino Acid