Literature DB >> 31274575

Expansion of phenotype of DDX3X syndrome: six new cases.

Bryony Beal1, Ian Hayes2, Julie McGaughran3, David J Amor4,5, Christina Miteff6, Victoria Jackson7, Olivia van Reyk4, Gopinath Subramanian6, Michael S Hildebrand8, Angela T Morgan4,5, Himanshu Goel1,9.   

Abstract

Pathogenic variants in DDX3X have recently been identified to be a relatively common cause of intellectual disability in females. In this study, we describe six female probands, from five unrelated families, with five novel heterozygous variants in DDX3X, and the identification of potential germline mosaicism. Consistent features between this cohort and previously described cases include developmental delay or intellectual disability, growth disturbance and movement disorder. Common facial dysmorphism within the cohort include short palpebral fissures, micrognathia, bulbous nasal tip, protruding ears, high arched palate, thin upper vermillion and smooth philtrum. Novel clinical features identified from this cohort include facial dysmorphisms, perinatal complications, valgus feet deformity, lipoatrophy, dystonic episodes, and cutaneous mastocytosis. This case series attempts to expand the phenotype of the DDX3X syndrome; however, it remains heterogeneous. Description of further cases is required to more accurately identify the significance of novel phenotypes within this cohort.

Entities:  

Year:  2019        PMID: 31274575     DOI: 10.1097/MCD.0000000000000289

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  8 in total

1.  Pathogenic DDX3X Mutations Impair RNA Metabolism and Neurogenesis during Fetal Cortical Development.

Authors:  Ashley L Lennox; Mariah L Hoye; Ruiji Jiang; Bethany L Johnson-Kerner; Lindsey A Suit; Srivats Venkataramanan; Charles J Sheehan; Fernando C Alsina; Brieana Fregeau; Kimberly A Aldinger; Ching Moey; Iryna Lobach; Alexandra Afenjar; Dusica Babovic-Vuksanovic; Stéphane Bézieau; Patrick R Blackburn; Jens Bunt; Lydie Burglen; Philippe M Campeau; Perrine Charles; Brian H Y Chung; Benjamin Cogné; Cynthia Curry; Maria Daniela D'Agostino; Nataliya Di Donato; Laurence Faivre; Delphine Héron; A Micheil Innes; Bertrand Isidor; Boris Keren; Amy Kimball; Eric W Klee; Paul Kuentz; Sébastien Küry; Dominique Martin-Coignard; Ghayda Mirzaa; Cyril Mignot; Noriko Miyake; Naomichi Matsumoto; Atsushi Fujita; Caroline Nava; Mathilde Nizon; Diana Rodriguez; Lot Snijders Blok; Christel Thauvin-Robinet; Julien Thevenon; Marie Vincent; Alban Ziegler; William Dobyns; Linda J Richards; A James Barkovich; Stephen N Floor; Debra L Silver; Elliott H Sherr
Journal:  Neuron       Date:  2020-03-04       Impact factor: 17.173

2.  Genetic aetiologies for childhood speech disorder: novel pathways co-expressed during brain development.

Authors:  Antony Kaspi; Angela T Morgan; Michael S Hildebrand; Victoria E Jackson; Ruth Braden; Olivia van Reyk; Tegan Howell; Simone Debono; Mariana Lauretta; Lottie Morison; Matthew J Coleman; Richard Webster; David Coman; Himanshu Goel; Mathew Wallis; Gabriel Dabscheck; Lilian Downie; Emma K Baker; Bronwyn Parry-Fielder; Kirrie Ballard; Eva Harrold; Shaun Ziegenfusz; Mark F Bennett; Erandee Robertson; Longfei Wang; Amber Boys; Simon E Fisher; David J Amor; Ingrid E Scheffer; Melanie Bahlo
Journal:  Mol Psychiatry       Date:  2022-09-18       Impact factor: 13.437

3.  Aberrant cortical development is driven by impaired cell cycle and translational control in a DDX3X syndrome model.

Authors:  Mariah L Hoye; Lorenzo Calviello; Abigail J Poff; Nna-Emeka Ejimogu; Carly R Newman; Maya D Montgomery; Jianhong Ou; Stephen N Floor; Debra L Silver
Journal:  Elife       Date:  2022-06-28       Impact factor: 8.713

4.  Prospective and detailed behavioral phenotyping in DDX3X syndrome.

Authors:  Lara Tang; Tess Levy; Sylvia Guillory; Danielle Halpern; Jessica Zweifach; Ivy Giserman-Kiss; Jennifer H Foss-Feig; Yitzchak Frank; Reymundo Lozano; Puneet Belani; Christina Layton; Bonnie Lerman; Emanuel Frowner; Michael S Breen; Silvia De Rubeis; Ana Kostic; Alexander Kolevzon; Joseph D Buxbaum; Paige M Siper; Dorothy E Grice
Journal:  Mol Autism       Date:  2021-05-16       Impact factor: 7.509

5.  Social and emotional characteristics of girls and young women with DDX3X-associated intellectual disability: a descriptive and comparative study.

Authors:  Elise Ng-Cordell; Anna Kolesnik-Taylor; Sinéad O'Brien; Duncan Astle; Gaia Scerif; Kate Baker
Journal:  J Autism Dev Disord       Date:  2022-05-10

6.  A de novo DDX3X Variant Is Associated With Syndromic Intellectual Disability: Case Report and Literature Review.

Authors:  Yun Chen; Kai-Yu Liu; Zai-Lan Yang; Xiao-Huan Li; Rui Xu; Hao Zhou
Journal:  Front Pediatr       Date:  2020-06-30       Impact factor: 3.418

7.  Proteomic Analysis of Brain Region and Sex-Specific Synaptic Protein Expression in the Adult Mouse Brain.

Authors:  Ute Distler; Sven Schumann; Hans-Georg Kesseler; Rainer Pielot; Karl-Heinz Smalla; Malte Sielaff; Michael J Schmeisser; Stefan Tenzer
Journal:  Cells       Date:  2020-01-28       Impact factor: 6.600

8.  A novel de novo DDX3X missense variant in a female with brachycephaly and intellectual disability: a case report.

Authors:  Giada Moresco; Jole Costanza; Carlo Santaniello; Ornella Rondinone; Federico Grilli; Elisabetta Prada; Simona Orcesi; Ilaria Coro; Anna Pichiecchio; Paola Marchisio; Monica Miozzo; Laura Fontana; Donatella Milani
Journal:  Ital J Pediatr       Date:  2021-03-31       Impact factor: 2.638

  8 in total

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