Literature DB >> 32021600

A Novel DLG3 Mutation Expanding the Phenotype of X-Linked Intellectual Disability Caused by DLG3 Nonsense Variants.

Anna Sandestig1, Anna Green1, Johan Aronsson2, Katarina Ellnebo1, Margarita Stefanova1.   

Abstract

The DLG3 gene is located at Xq13.1 and encodes SAP102, a member of the MAGUK protein family, extensively expressed in the brain and involved in synaptic function. Mutations in DLG3 are associated with a rare nonsyndromic form of X-linked intellectual disability (XLID) and have been described in 11 families to date. All affected males presented with intellectual disability, and some showed additional clinical features. The majority of female carriers were reported asymptomatic or mildly affected, due to skewed X-inactivation, rarely severely affected. We report a family, a boy and his mother, with a novel nonsense mutation in the DLG3 gene, c.1720C>T; p.Arg574*. The boy, hemizygous for the variant, showed intellectual disability, short stature due to growth hormone deficiency, dysmorphic features, and pectus excavatum. The mother, who presented with learning disabilities and borderline cognitive development, is a heterozygous carrier of the variant, which had arisen de novo. X-inactivation test was noninformative. This case report broadens the phenotypic spectrum of XLID caused by DLG3 nonsense variants. The dysmorphic features of the affected males may be more frequent than previously thought.
Copyright © 2019 by S. Karger AG, Basel.

Entities:  

Keywords:  Clinical heterogeneity; DLG3 nonsense mutation; Dysmorphology; Exome sequencing; Facial dysmorphology; X-linked intellectual disability

Year:  2019        PMID: 32021600      PMCID: PMC6997794          DOI: 10.1159/000502601

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  14 in total

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Journal:  Eur J Med Genet       Date:  2012-05-30       Impact factor: 2.708

Review 2.  The postsynaptic architecture of excitatory synapses: a more quantitative view.

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Review 3.  Synaptic trafficking of glutamate receptors by MAGUK scaffolding proteins.

Authors:  Guillermo M Elias; Roger A Nicoll
Journal:  Trends Cell Biol       Date:  2007-07-20       Impact factor: 20.808

4.  Next-generation sequencing in X-linked intellectual disability.

Authors:  Andreas Tzschach; Ute Grasshoff; Stefanie Beck-Woedl; Claudia Dufke; Claudia Bauer; Martin Kehrer; Christina Evers; Ute Moog; Barbara Oehl-Jaschkowitz; Nataliya Di Donato; Robert Maiwald; Christine Jung; Alma Kuechler; Solveig Schulz; Peter Meinecke; Stephanie Spranger; Jürgen Kohlhase; Jörg Seidel; Silke Reif; Manuela Rieger; Angelika Riess; Marc Sturm; Julia Bickmann; Christopher Schroeder; Andreas Dufke; Olaf Riess; Peter Bauer
Journal:  Eur J Hum Genet       Date:  2015-02-04       Impact factor: 4.246

Review 5.  Membrane-associated guanylate kinases regulate adhesion and plasticity at cell junctions.

Authors:  Lars Funke; Srikanth Dakoji; David S Bredt
Journal:  Annu Rev Biochem       Date:  2005       Impact factor: 23.643

6.  Origin of PDZ (DHR, GLGF) domains.

Authors:  M B Kennedy
Journal:  Trends Biochem Sci       Date:  1995-09       Impact factor: 13.807

Review 7.  MAGUKs: multifaceted synaptic organizers.

Authors:  Sehoon Won; Jon M Levy; Roger A Nicoll; Katherine W Roche
Journal:  Curr Opin Neurobiol       Date:  2017-02-23       Impact factor: 6.627

8.  Postsynaptic density scaffold SAP102 regulates cortical synapse development through EphB and PAK signaling pathway.

Authors:  Yasunobu Murata; Martha Constantine-Paton
Journal:  J Neurosci       Date:  2013-03-13       Impact factor: 6.167

9.  A novel mutation in the DLG3 gene encoding the synapse-associated protein 102 (SAP102) causes non-syndromic mental retardation.

Authors:  Ginevra Zanni; Hilde van Esch; Anissa Bensalem; Yoann Saillour; Karine Poirier; Laetitia Castelnau; Hans Hilger Ropers; Arjan P M de Brouwer; Fréderic Laumonnier; Jean-Pierre Fryns; Jamel Chelly
Journal:  Neurogenetics       Date:  2009-10-01       Impact factor: 2.660

10.  X-exome sequencing in Finnish families with intellectual disability--four novel mutations and two novel syndromic phenotypes.

Authors:  Anju K Philips; Auli Sirén; Kristiina Avela; Mirja Somer; Maarit Peippo; Minna Ahvenainen; Fatma Doagu; Maria Arvio; Helena Kääriäinen; Hilde Van Esch; Guy Froyen; Stefan A Haas; Hao Hu; Vera M Kalscheuer; Irma Järvelä
Journal:  Orphanet J Rare Dis       Date:  2014-04-11       Impact factor: 4.123

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