Mutations in <i>SYNGAP1</i> in Autosomal Nonsyndromic Mental Retardation

Fadi F. Hamdan(Centre Hospitalier Universitaire Sainte-Justine), Julie Gauthier(Centre Hospitalier de l’Université de Montréal), Dan Spiegelman(Centre Hospitalier de l’Université de Montréal), Anne Noreau(Centre Hospitalier de l’Université de Montréal), Yanlian Yang(Centre Hospitalier de l’Université de Montréal), Stéphanie Pellerin(Centre Hospitalier Universitaire Sainte-Justine), Sylvia Dobrzeniecka(Centre Hospitalier de l’Université de Montréal), Mélanie Côté(Centre Hospitalier de l’Université de Montréal), Elizabeth Perreau-Linck, Lionel Carmant(Centre Hospitalier Universitaire Sainte-Justine), Guy D’Anjou(Centre Hospitalier Universitaire Sainte-Justine), Éric Fombonne, Anjené Addington(National Institute of Mental Health), Judith L. Rapoport(National Institute of Mental Health), Lynn E. DeLisi(Nathan Kline Institute for Psychiatric Research), Marie‐Odile Krebs(Délégation Paris 5), Fayçal Mouaffak(Délégation Paris 5), Ridha Joober(Douglas Mental Health University Institute), Laurent Mottron(Hôpital Rivière-des-Prairies), Pierre Drapeau(Nerve Centre), Claude Marineau(Centre Hospitalier de l’Université de Montréal), Ronald G. Lafrenière(Centre Hospitalier de l’Université de Montréal), Jean Claude Lacaille, Guy A. Rouleau(Centre Hospitalier de l’Université de Montréal), Jacques L. Michaud(Centre Hospitalier Universitaire Sainte-Justine)
New England Journal of Medicine
February 4, 2009
Cited by 350Open Access
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Abstract

Although autosomal forms of nonsyndromic mental retardation account for the majority of cases of mental retardation, the genes that are involved remain largely unknown. We sequenced the autosomal gene SYNGAP1, which encodes a ras GTPase-activating protein that is critical for cognition and synapse function, in 94 patients with nonsyndromic mental retardation. We identified de novo truncating mutations (K138X, R579X, and L813RfsX22) in three of these patients. In contrast, we observed no de novo or truncating mutations in SYNGAP1 in samples from 142 subjects with autism spectrum disorders, 143 subjects with schizophrenia, and 190 control subjects. These results indicate that SYNGAP1 disruption is a cause of autosomal dominant nonsyndromic mental retardation.


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