Literature DB >> 25102098

A girl with West syndrome and autistic features harboring a de novo TBL1XR1 mutation.

Hirotomo Saitsu1, Jun Tohyama2, Tom Walsh3, Mitsuhiro Kato4, Yu Kobayashi2, Ming Lee3, Yoshinori Tsurusaki1, Noriko Miyake1, Yu-Ichi Goto5, Ichizo Nishino6, Akira Ohtake7, Mary-Claire King3, Naomichi Matsumoto1.   

Abstract

Recently, de novo mutations in TBL1XR1 were found in two patients with autism spectrum disorders. Here, we report on a Japanese girl presenting with West syndrome, Rett syndrome-like and autistic features. Her initial development was normal until she developed a series of spasms at 5 months of age. Electroencephalogram at 7 months showed a pattern of hypsarrhythmia, which led to a diagnosis of West syndrome. Stereotypic hand movements appeared at 8 months of age, and autistic features such as deficits in communication, hyperactivity and excitability were observed later, at 4 years and 9 months. Whole exome sequencing of the patient and her parents revealed a de novo TBL1XR1 mutation [c.209 G>A (p.Gly70Asp)] occurring at an evolutionarily conserved amino acid in an F-box-like domain. Our report expands the clinical spectrum of TBL1XR1 mutations to West syndrome with Rett-like features, together with autistic features.

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Year:  2014        PMID: 25102098     DOI: 10.1038/jhg.2014.71

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  11 in total

1.  Reversible SUMOylation of TBL1-TBLR1 regulates β-catenin-mediated Wnt signaling.

Authors:  Hyo-Kyoung Choi; Kyung-Chul Choi; Jung-Yoon Yoo; Meiying Song; Suk Jin Ko; Chul Hoon Kim; Jin-Hyun Ahn; Kyung-Hee Chun; Jong In Yook; Ho-Geun Yoon
Journal:  Mol Cell       Date:  2011-07-22       Impact factor: 17.970

2.  Detection of inherited mutations for breast and ovarian cancer using genomic capture and massively parallel sequencing.

Authors:  Tom Walsh; Ming K Lee; Silvia Casadei; Anne M Thornton; Sunday M Stray; Christopher Pennil; Alex S Nord; Jessica B Mandell; Elizabeth M Swisher; Mary-Claire King
Journal:  Proc Natl Acad Sci U S A       Date:  2010-06-28       Impact factor: 11.205

Review 3.  Genes of early-onset epileptic encephalopathies: from genotype to phenotype.

Authors:  Mario Mastrangelo; Vincenzo Leuzzi
Journal:  Pediatr Neurol       Date:  2012-01       Impact factor: 3.372

4.  Mutations in TCF4, encoding a class I basic helix-loop-helix transcription factor, are responsible for Pitt-Hopkins syndrome, a severe epileptic encephalopathy associated with autonomic dysfunction.

Authors:  Jeanne Amiel; Marlene Rio; Loic de Pontual; Richard Redon; Valerie Malan; Nathalie Boddaert; Perrine Plouin; Nigel P Carter; Stanislas Lyonnet; Arnold Munnich; Laurence Colleaux
Journal:  Am J Hum Genet       Date:  2007-03-23       Impact factor: 11.025

5.  Haploinsufficiency of TCF4 causes syndromal mental retardation with intermittent hyperventilation (Pitt-Hopkins syndrome).

Authors:  Christiane Zweier; Maarit M Peippo; Juliane Hoyer; Sergio Sousa; Armand Bottani; Jill Clayton-Smith; William Reardon; Jorge Saraiva; Alexandra Cabral; Ina Gohring; Koen Devriendt; Thomy de Ravel; Emilia K Bijlsma; Raoul C M Hennekam; Alfredo Orrico; Monika Cohen; Alexander Dreweke; Andre Reis; Peter Nurnberg; Anita Rauch
Journal:  Am J Hum Genet       Date:  2007-03-23       Impact factor: 11.025

6.  TBL1-TBLR1 and beta-catenin recruit each other to Wnt target-gene promoter for transcription activation and oncogenesis.

Authors:  Jiong Li; Cun-Yu Wang
Journal:  Nat Cell Biol       Date:  2008-01-13       Impact factor: 28.824

7.  Multiplex targeted sequencing identifies recurrently mutated genes in autism spectrum disorders.

Authors:  Brian J O'Roak; Laura Vives; Wenqing Fu; Jarrett D Egertson; Ian B Stanaway; Ian G Phelps; Gemma Carvill; Akash Kumar; Choli Lee; Katy Ankenman; Jeff Munson; Joseph B Hiatt; Emily H Turner; Roie Levy; Diana R O'Day; Niklas Krumm; Bradley P Coe; Beth K Martin; Elhanan Borenstein; Deborah A Nickerson; Heather C Mefford; Dan Doherty; Joshua M Akey; Raphael Bernier; Evan E Eichler; Jay Shendure
Journal:  Science       Date:  2012-11-15       Impact factor: 47.728

8.  Sporadic autism exomes reveal a highly interconnected protein network of de novo mutations.

Authors:  Brian J O'Roak; Laura Vives; Santhosh Girirajan; Emre Karakoc; Niklas Krumm; Bradley P Coe; Roie Levy; Arthur Ko; Choli Lee; Joshua D Smith; Emily H Turner; Ian B Stanaway; Benjamin Vernot; Maika Malig; Carl Baker; Beau Reilly; Joshua M Akey; Elhanan Borenstein; Mark J Rieder; Deborah A Nickerson; Raphael Bernier; Jay Shendure; Evan E Eichler
Journal:  Nature       Date:  2012-04-04       Impact factor: 49.962

9.  TBLR1 regulates the expression of nuclear hormone receptor co-repressors.

Authors:  Xin-Min Zhang; Qing Chang; Lin Zeng; Judy Gu; Stuart Brown; Ross S Basch
Journal:  BMC Cell Biol       Date:  2006-08-07       Impact factor: 4.241

10.  Targeted gene disruption demonstrates that P-selectin glycoprotein ligand 1 (PSGL-1) is required for P-selectin-mediated but not E-selectin-mediated neutrophil rolling and migration.

Authors:  J Yang; T Hirata; K Croce; G Merrill-Skoloff; B Tchernychev; E Williams; R Flaumenhaft; B C Furie; B Furie
Journal:  J Exp Med       Date:  1999-12-20       Impact factor: 14.307

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  17 in total

Review 1.  TBL1XR1 in physiological and pathological states.

Authors:  Jian Yi Li; Garrett Daniels; Jing Wang; Xinmin Zhang
Journal:  Am J Clin Exp Urol       Date:  2015-04-25

2.  Monogenic disorders that mimic the phenotype of Rett syndrome.

Authors:  Siddharth Srivastava; Sonal Desai; Julie Cohen; Constance Smith-Hicks; Kristin Barañano; Ali Fatemi; SakkuBai Naidu
Journal:  Neurogenetics       Date:  2018-01-10       Impact factor: 2.660

3.  Understanding the Landscape of X-linked Variants Causing Intellectual Disability in Females Through Extreme X Chromosome Inactivation Skewing.

Authors:  Evelyn Quintanilha Vianna; Rafael Mina Piergiorge; Andressa Pereira Gonçalves; Jussara Mendonça Dos Santos; Veluma Calassara; Carla Rosenberg; Ana Cristina Victorino Krepischi; Raquel Tavares Boy da Silva; Suely Rodrigues Dos Santos; Márcia Gonçalves Ribeiro; Filipe Brum Machado; Enrique Medina-Acosta; Márcia Mattos Gonçalves Pimentel; Cíntia Barros Santos-Rebouças
Journal:  Mol Neurobiol       Date:  2020-06-20       Impact factor: 5.590

4.  Genetic heterogeneity in infantile spasms.

Authors:  Alison M Muir; Candace T Myers; Nancy T Nguyen; Julia Saykally; Dana Craiu; Peter De Jonghe; Ingo Helbig; Dorota Hoffman-Zacharska; Renzo Guerrini; Anna-Elina Lehesjoki; Carla Marini; Rikke S Møller; Jose Serratosa; Katalin Štěrbová; Pasquale Striano; Sarah von Spiczak; Sarah Weckhuysen; Heather C Mefford
Journal:  Epilepsy Res       Date:  2019-07-29       Impact factor: 3.045

Review 5.  Rare variant of TBL1XR1 in West syndrome: A case report.

Authors:  Yajun Shen; Meng Yuan; Huan Luo; Zuozhen Yang; Mengmeng Liang; Jing Gan
Journal:  Mol Genet Genomic Med       Date:  2022-05-25       Impact factor: 2.473

6.  De novo non-synonymous TBL1XR1 mutation alters Wnt signaling activity.

Authors:  Akira Nishi; Shusuke Numata; Atsushi Tajima; Xiaolei Zhu; Koki Ito; Atsushi Saito; Yusuke Kato; Makoto Kinoshita; Shinji Shimodera; Shinji Ono; Shinichiro Ochi; Akira Imamura; Naohiro Kurotaki; Shu-Ichi Ueno; Nakao Iwata; Kiyoshi Fukui; Issei Imoto; Atsushi Kamiya; Tetsuro Ohmori
Journal:  Sci Rep       Date:  2017-06-06       Impact factor: 4.379

7.  A specific mutation in TBL1XR1 causes Pierpont syndrome.

Authors:  Charlotte A Heinen; Aldo Jongejan; Peter J Watson; Bert Redeker; Anita Boelen; Olga Boudzovitch-Surovtseva; Francesca Forzano; Roel Hordijk; Richard Kelley; Ann H Olney; Mary Ella Pierpont; G Bradley Schaefer; Fiona Stewart; A S Paul van Trotsenburg; Eric Fliers; John W R Schwabe; Raoul C Hennekam
Journal:  J Med Genet       Date:  2016-01-14       Impact factor: 6.318

8.  Complex intrachromosomal rearrangement in 1q leading to 1q32.2 microdeletion: a potential role of SRGAP2 in the gyrification of cerebral cortex.

Authors:  Martina Rincic; Milan Rados; Zeljka Krsnik; Kristina Gotovac; Fran Borovecki; Thomas Liehr; Lukrecija Brecevic
Journal:  Mol Cytogenet       Date:  2016-02-20       Impact factor: 2.009

9.  Integrating population variation and protein structural analysis to improve clinical interpretation of missense variation: application to the WD40 domain.

Authors:  Roman A Laskowski; Nidhi Tyagi; Diana Johnson; Shelagh Joss; Esther Kinning; Catherine McWilliam; Miranda Splitt; Janet M Thornton; Helen V Firth; Caroline F Wright
Journal:  Hum Mol Genet       Date:  2016-01-05       Impact factor: 6.150

10.  Histone deacetylase 3 associates with MeCP2 to regulate FOXO and social behavior.

Authors:  Alexi Nott; Jemmie Cheng; Fan Gao; Yuan-Ta Lin; Elizabeta Gjoneska; Tak Ko; Paras Minhas; Alicia Viridiana Zamudio; Jia Meng; Feiran Zhang; Peng Jin; Li-Huei Tsai
Journal:  Nat Neurosci       Date:  2016-07-18       Impact factor: 24.884

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