Literature DB >> 26842963

ADAT3-related intellectual disability: Further delineation of the phenotype.

Ayman W El-Hattab1, Mohammed A Saleh2, Amal Hashem3,4, Mohammed Al-Owain3,5, Ali Al Asmari2, Hala Rabei1, Hanem Abdelraouf4, Mais Hashem6, Anas M Alazami6, Nisha Patel6, Ranad Shaheen6, Eissa A Faqeih2, Fowzan S Alkuraya3,6,7.   

Abstract

ADAT3-related intellectual disability has been recently described in 24 individuals from eight Saudi families who had cognitive impairment and strabismus. Other common features included growth failure, microcephaly, tone abnormalities, epilepsy, and nonspecific brain abnormalities. A single homozygous founder mutation (c.382G>A:p.(V128M)) in the ADAT3 gene, which encodes a protein that functions in tRNA editing, was identified in all affected individuals. In this report, we present additional 15 individuals from 11 families (10 Saudis and 1 Emirati) who are homozygous for the same founder mutation. In addition to the universal findings of intellectual disability and strabismus, the majority exhibited microcephaly and growth failure. Additional features not reported in the original cohort include dysmorphic facial features (prominent forehead, up-slanted palpebral fissures, epicanthus, and depressed nasal bridge), behavioral problems (hyperactivity and aggressiveness), recurrent otitis media, and growth hormone deficiency. ADAT3-related intellectual disability is an important recognizable cause of intellectual disability in Arabia.
© 2016 Wiley Periodicals, Inc.

Entities:  

Keywords:  failure to thrive; growth hormone; intellectual disability; strabismus

Mesh:

Substances:

Year:  2016        PMID: 26842963     DOI: 10.1002/ajmg.a.37578

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  13 in total

1.  A homozygous truncating mutation in PUS3 expands the role of tRNA modification in normal cognition.

Authors:  Ranad Shaheen; Lu Han; Eissa Faqeih; Nour Ewida; Eman Alobeid; Eric M Phizicky; Fowzan S Alkuraya
Journal:  Hum Genet       Date:  2016-04-07       Impact factor: 4.132

2.  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

3.  THUMPD1 bi-allelic variants cause loss of tRNA acetylation and a syndromic neurodevelopmental disorder.

Authors:  Martin Broly; Bogdan V Polevoda; Kamel M Awayda; Ning Tong; Jenna Lentini; Thomas Besnard; Wallid Deb; Declan O'Rourke; Julia Baptista; Sian Ellard; Mohammed Almannai; Mais Hashem; Ferdous Abdulwahab; Hanan Shamseldin; Saeed Al-Tala; Fowzan S Alkuraya; Alberta Leon; Rosa L E van Loon; Alessandra Ferlini; Mariabeatrice Sanchini; Stefania Bigoni; Andrea Ciorba; Hans van Bokhoven; Zafar Iqbal; Almundher Al-Maawali; Fathiya Al-Murshedi; Anuradha Ganesh; Watfa Al-Mamari; Sze Chern Lim; Lynn S Pais; Natasha Brown; Saima Riazuddin; Stéphane Bézieau; Dragony Fu; Bertrand Isidor; Benjamin Cogné; Mitchell R O'Connell
Journal:  Am J Hum Genet       Date:  2022-02-22       Impact factor: 11.043

4.  An intellectual disability-associated missense variant in TRMT1 impairs tRNA modification and reconstitution of enzymatic activity.

Authors:  Kejia Zhang; Jenna M Lentini; Christopher T Prevost; Mais O Hashem; Fowzan S Alkuraya; Dragony Fu
Journal:  Hum Mutat       Date:  2020-01-16       Impact factor: 4.878

5.  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

6.  A novel 8-bp duplication in ADAT3 causes mild intellectual disability.

Authors:  Ahmad Reza Salehi Chaleshtori; Noriko Miyake; Mohammad Ahmadvand; Oranous Bashti; Naomichi Matsumoto; Mehrdad Noruzinia
Journal:  Hum Genome Var       Date:  2018-05-21

Review 7.  tRNA-Derived Small RNAs: Biogenesis, Modification, Function and Potential Impact on Human Disease Development.

Authors:  Vera Oberbauer; Matthias R Schaefer
Journal:  Genes (Basel)       Date:  2018-12-05       Impact factor: 4.096

8.  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

9.  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

10.  Crystal structure of the yeast heterodimeric ADAT2/3 deaminase.

Authors:  Xiwen Liu; Ruoyu Chen; Yujie Sun; Ran Chen; Jie Zhou; Qingnan Tian; Xuan Tao; Zhang Zhang; Guan-Zheng Luo; Wei Xie
Journal:  BMC Biol       Date:  2020-12-03       Impact factor: 7.431

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