Literature DB >> 31602190

GATAD2B Gene Microdeletion Causing Intellectual Disability Autosomal Dominant Type 18: Case Report and Review of the Literature.

Milana Trubnykova1, Jeny Bazalar Montoya2, Jorge La Serna-Infantes3, Flor Vásquez Sotomayor1, María Del Carmen Castro Mujica4, Hugo Hernán Abarca Barriga1,4,5.   

Abstract

Pathogenic variants of the GATAD2B gene (1q21.3) are linked to intellectual disability autosomal dominant type 18 (MRD18; MIM 615074), characterized by dysmorphic features, psychomotor and language delay. We present an 11-year-old female patient with intellectual disability and typical clinical characteristics of MRD18. Chromosomal microarray analysis (CMA) revealed a novel CNV, approximately 200 kb in size and showed that the INTS3 and SLC27A3 genes are completely deleted along with the first 10 exons of the GATAD2B gene. INTS3 encodes the integrator complex subunit 3 and is part of the complex that maintains genome stability; SLC27A3 encodes a fatty acid transporter and has been associated with autism spectrum disorder. GATAD2B haploinsufficiency is associated with the phenotype. Furthermore, the girl had other clinical characteristics not previously described, such as emotional instability, calf hypotrophy, hypoplastic digit pads, tapered thumbs, and anterior earlobe crease. This study highlights the importance of the phenotype-genotype correlation using molecular diagnostic techniques, such as CMA, and its impact on precise diagnosis, treatment, prognosis, and genetic counseling for patients and their families.
Copyright © 2019 by S. Karger AG, Basel.

Entities:  

Keywords:  Chromosomal microarray analysis; Copy number variation; GATAD2B; Intellectual disability; MRD18

Year:  2019        PMID: 31602190      PMCID: PMC6738330          DOI: 10.1159/000499209

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


  24 in total

Review 1.  1q21.3 deletion involving GATAD2B: An emerging recurrent microdeletion syndrome.

Authors:  Thipwimol Tim-Aroon; Natini Jinawath; Weerin Thammachote; Praweena Sinpitak; Anchalee Limrungsikul; Chaiyos Khongkhatithum; Duangrurdee Wattanasirichaigoon
Journal:  Am J Med Genet A       Date:  2017-03       Impact factor: 2.802

2.  Novel GATAD2B loss-of-function mutations cause intellectual disability in two unrelated cases.

Authors:  Xiaomei Luo; Yongyi Zou; Bo Tan; Yue Zhang; Jing Guo; Lanlan Zeng; Rui Zhang; Hu Tan; Xianda Wei; Yiqiao Hu; Yu Zheng; Desheng Liang; Lingqian Wu
Journal:  J Hum Genet       Date:  2017-01-12       Impact factor: 3.172

3.  Adaptive and maladaptive functioning in Kleefstra syndrome compared to other rare genetic disorders with intellectual disabilities.

Authors:  Karlijn Vermeulen; Anneke de Boer; Joost G E Janzing; David A Koolen; Charlotte W Ockeloen; Marjolein H Willemsen; Floor M Verhoef; Patricia A M van Deurzen; Linde van Dongen; Hans van Bokhoven; Jos I M Egger; Wouter G Staal; Tjitske Kleefstra
Journal:  Am J Med Genet A       Date:  2017-05-12       Impact factor: 2.802

Review 4.  Consensus statement: chromosomal microarray is a first-tier clinical diagnostic test for individuals with developmental disabilities or congenital anomalies.

Authors:  David T Miller; Margaret P Adam; Swaroop Aradhya; Leslie G Biesecker; Arthur R Brothman; Nigel P Carter; Deanna M Church; John A Crolla; Evan E Eichler; Charles J Epstein; W Andrew Faucett; Lars Feuk; Jan M Friedman; Ada Hamosh; Laird Jackson; Erin B Kaminsky; Klaas Kok; Ian D Krantz; Robert M Kuhn; Charles Lee; James M Ostell; Carla Rosenberg; Stephen W Scherer; Nancy B Spinner; Dimitri J Stavropoulos; James H Tepperberg; Erik C Thorland; Joris R Vermeesch; Darrel J Waggoner; Michael S Watson; Christa Lese Martin; David H Ledbetter
Journal:  Am J Hum Genet       Date:  2010-05-14       Impact factor: 11.025

Review 5.  Structure and function insights into the NuRD chromatin remodeling complex.

Authors:  Morgan P Torchy; Ali Hamiche; Bruno P Klaholz
Journal:  Cell Mol Life Sci       Date:  2015-03-22       Impact factor: 9.261

6.  Characterising and predicting haploinsufficiency in the human genome.

Authors:  Ni Huang; Insuk Lee; Edward M Marcotte; Matthew E Hurles
Journal:  PLoS Genet       Date:  2010-10-14       Impact factor: 5.917

7.  [Intellectual development disorders in Latin America: a framework for setting policy priorities for research and care].

Authors:  Eduardo Lazcano-Ponce; Gregorio Katz; Betania Allen-Leigh; Laura Magaña Valladares; Guillermina Rangel-Eudave; Alberto Minoletti; Ernesto Wahlberg; Armando Vásquez; Luis Salvador-Carulla
Journal:  Rev Panam Salud Publica       Date:  2013-09

Review 8.  The NuRD architecture.

Authors:  Hillary F Allen; Paul A Wade; Tatiana G Kutateladze
Journal:  Cell Mol Life Sci       Date:  2013-01-23       Impact factor: 9.261

9.  DECIPHER: Database of Chromosomal Imbalance and Phenotype in Humans Using Ensembl Resources.

Authors:  Helen V Firth; Shola M Richards; A Paul Bevan; Stephen Clayton; Manuel Corpas; Diana Rajan; Steven Van Vooren; Yves Moreau; Roger M Pettett; Nigel P Carter
Journal:  Am J Hum Genet       Date:  2009-04-02       Impact factor: 11.025

10.  De novo mutations in moderate or severe intellectual disability.

Authors:  Fadi F Hamdan; Myriam Srour; Jose-Mario Capo-Chichi; Hussein Daoud; Christina Nassif; Lysanne Patry; Christine Massicotte; Amirthagowri Ambalavanan; Dan Spiegelman; Ousmane Diallo; Edouard Henrion; Alexandre Dionne-Laporte; Anne Fougerat; Alexey V Pshezhetsky; Sunita Venkateswaran; Guy A Rouleau; Jacques L Michaud
Journal:  PLoS Genet       Date:  2014-10-30       Impact factor: 5.917

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  1 in total

1.  Defining the Dynamic Regulation of O-GlcNAc Proteome in the Mouse Cortex---the O-GlcNAcylation of Synaptic and Trafficking Proteins Related to Neurodegenerative Diseases.

Authors:  Van N Huynh; Sheng Wang; Xiaosen Ouyang; Willayat Y Wani; Michelle S Johnson; Balu K Chacko; Anil G Jegga; Wei-Jun Qian; John C Chatham; Victor M Darley-Usmar; Jianhua Zhang
Journal:  Front Aging       Date:  2021-09-29
  1 in total

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