Literature DB >> 30296593

A new case confirming and expanding the phenotype spectrum of ADAT3-related intellectual disability syndrome.

Rajech Sharkia1, Abdelnaser Zalan2, Azhar Jabareen-Masri2, Hazar Zahalka3, Muhammad Mahajnah4.   

Abstract

The present study describes two patients with clinical diagnosis of ID, from a consanguineous family in Israel. Whole exome sequencing identified a homozygous missense mutation in the ADAT3 gene. The clinical features of our patients were compared with several cases described in two recently published studies that documented clinical manifestation of this same mutation. Both affected siblings in our study expressed the previously described clinical features such as intellectual disability, strabismus, FTT/underweight, microcephaly and hypotonia. Interestingly, our patients suffered from additional clinical manifestations that were not detailed in the previous two studies, such as: gait difficulties, instability, teeth abnormalities, neuropathy and contractures of the hand wrist and fingers. We conclude that the ADAT3 gene mutation is responsible for ADAT3-related ID syndrome, which induces the variety clinical manifestations exhibited by our patients. Further studies aimed at identifying and characterizing additional afflicted families worldwide will be required to obtain a more comprehensive understanding of this syndrome.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  ADAT3 gene mutation; Arabs in Israel; Clinical manifestations; Intellectual disability syndrome

Mesh:

Substances:

Year:  2018        PMID: 30296593     DOI: 10.1016/j.ejmg.2018.10.001

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  8 in total

1.  Novel Missense Variants in ADAT3 as a Cause of Syndromic Intellectual Disability.

Authors:  Elizabeth Thomas; Andrea M Lewis; Yaping Yang; Sirisak Chanprasert; Lorraine Potocki; Daryl A Scott
Journal:  J Pediatr Genet       Date:  2019-07-09

Review 2.  Modifications of the human tRNA anticodon loop and their associations with genetic diseases.

Authors:  Jing-Bo Zhou; En-Duo Wang; Xiao-Long Zhou
Journal:  Cell Mol Life Sci       Date:  2021-10-04       Impact factor: 9.261

3.  The structure of the mouse ADAT2/ADAT3 complex reveals the molecular basis for mammalian tRNA wobble adenosine-to-inosine deamination.

Authors:  Elizabeth Ramos-Morales; Efil Bayam; Jordi Del-Pozo-Rodríguez; Thalia Salinas-Giegé; Martin Marek; Peggy Tilly; Philippe Wolff; Edouard Troesch; Eric Ennifar; Laurence Drouard; Juliette D Godin; Christophe Romier
Journal:  Nucleic Acids Res       Date:  2021-06-21       Impact factor: 16.971

4.  Matching tRNA modifications in humans to their known and predicted enzymes.

Authors:  Valérie de Crécy-Lagard; Pietro Boccaletto; Carl G Mangleburg; Puneet Sharma; Todd M Lowe; Sebastian A Leidel; Janusz M Bujnicki
Journal:  Nucleic Acids Res       Date:  2019-03-18       Impact factor: 16.971

5.  Formation of tRNA Wobble Inosine in Humans Is Disrupted by a Millennia-Old Mutation Causing Intellectual Disability.

Authors:  Jillian Ramos; Lu Han; Yan Li; Felix Hagelskamp; Stefanie M Kellner; Fowzan S Alkuraya; Eric M Phizicky; Dragony Fu
Journal:  Mol Cell Biol       Date:  2019-09-11       Impact factor: 4.272

Review 6.  Inosine in Biology and Disease.

Authors:  Sundaramoorthy Srinivasan; Adrian Gabriel Torres; Lluís Ribas de Pouplana
Journal:  Genes (Basel)       Date:  2021-04-19       Impact factor: 4.096

7.  A Rare Case of Adenosine Deaminase tRNA-Specific 3 Mutation, Adrenal Insufficiency, and Rhabdomyolysis.

Authors:  Waheeb AlDhalaan; Faaezuddin Syed; Haroon A Javaid; Afaf AlSagheir; Sami Almustanyir
Journal:  Cureus       Date:  2021-11-23

8.  Identification and rescue of a tRNA wobble inosine deficiency causing intellectual disability disorder.

Authors:  Jillian Ramos; Melissa Proven; Jonatan Halvardson; Felix Hagelskamp; Ekaterina Kuchinskaya; Benjamin Phelan; Ryan Bell; Stefanie M Kellner; Lars Feuk; Ann-Charlotte Thuresson; Dragony Fu
Journal:  RNA       Date:  2020-08-06       Impact factor: 4.942

  8 in total

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