Literature DB >> 31985473

Clinical and Genetic Features in a Series of Eight Unrelated Patients with Neuropathy Due to Glycyl-tRNA Synthetase (GARS) Variants.

Natalie Forrester1, Rohini Rattihalli2, Rita Horvath3, Lorenzo Maggi4, Adnan Manzur5, Geraint Fuller6, Nicholas Gutowski7, Julia Rankin7, David Dick8, Christopher Buxton1, Mark Greenslade1, Anirban Majumdar9.   

Abstract

Pathogenic variants in the Glycyl-tRNA synthetase gene cause the allelic disorders Charcot-Marie-Tooth disease type 2D and distal hereditary motor neuropathy type V. We describe clinical features in 8 unrelated patients found to have Glycyl-tRNA synthetase variants by Next Generation Sequencing. In addition to upper limb predominant symptoms, other presentations included failure to thrive, feeding difficulties and lower limb dominant symptoms. Variability in the age at testing ranged from 14 months to 59 years. The youngest being symptomatic from 3 months and ventilator-dependent. Sequence variants were reported as pathogenic, p.(Glu125Lys), p.(His472Arg); likely pathogenic, p.(His216Arg), p.(Gly327Arg), p.(Lys510Gln), p.(Met555Val); and of uncertain significance, p.(Arg27Pro). Our case series describes novel Glycyl-tRNA synthetase variants and demonstrates the clinical utility of Next Generation Sequencing testing for patients with hereditary neuropathy. Identification of novel variants by Next Generation Sequencing illustrates that there exists a wide spectrum of clinical features and supports the newer simplified classification of neuropathies.

Entities:  

Keywords:  Aminoacyl-tRNA synthetases; Charcot-Marie-Tooth disease; Glycyl-tRNA synthetase; Next Generation Sequencing; distal hereditary motor neuropathy; peripheral neuropathies

Year:  2020        PMID: 31985473     DOI: 10.3233/JND-200472

Source DB:  PubMed          Journal:  J Neuromuscul Dis


  5 in total

1.  tRNA overexpression rescues peripheral neuropathy caused by mutations in tRNA synthetase.

Authors:  Amila Zuko; Moushami Mallik; Robin Thompson; Emily L Spaulding; Anne R Wienand; Marije Been; Abigail L D Tadenev; Nick van Bakel; Céline Sijlmans; Leonardo A Santos; Julia Bussmann; Marica Catinozzi; Sarada Das; Divita Kulshrestha; Robert W Burgess; Zoya Ignatova; Erik Storkebaum
Journal:  Science       Date:  2021-09-01       Impact factor: 63.714

2.  Screening for SH3TC2 variants in Charcot-Marie-Tooth disease in a cohort of Chinese patients.

Authors:  Bo Sun; Zheng-Qing He; Yan-Ran Li; Jiong-Ming Bai; Hao-Ran Wang; Hong-Fen Wang; Fang Cui; Fei Yang; Xu-Sheng Huang
Journal:  Acta Neurol Belg       Date:  2021-02-15       Impact factor: 2.471

3.  Pathogenic missense variants altering codon 336 of GARS1 lead to divergent dominant phenotypes.

Authors:  Alayne P Meyer; Megan E Forrest; Stefan Nicolau; Wojciech Wiszniewski; Mary Pat Bland; Chang-Yong Tsao; Anthony Antonellis; Nicolas J Abreu
Journal:  Hum Mutat       Date:  2022-04-21       Impact factor: 4.700

4.  Altered Sensory Neuron Development in CMT2D Mice Is Site-Specific and Linked to Increased GlyRS Levels.

Authors:  James N Sleigh; Aleksandra M Mech; Tahmina Aktar; Yuxin Zhang; Giampietro Schiavo
Journal:  Front Cell Neurosci       Date:  2020-08-11       Impact factor: 5.505

5.  De Novo and Bi-allelic Pathogenic Variants in NARS1 Cause Neurodevelopmental Delay Due to Toxic Gain-of-Function and Partial Loss-of-Function Effects.

Authors:  Andreea Manole; Stephanie Efthymiou; Emer O'Connor; Marisa I Mendes; Matthew Jennings; Reza Maroofian; Indran Davagnanam; Kshitij Mankad; Maria Rodriguez Lopez; Vincenzo Salpietro; Ricardo Harripaul; Lauren Badalato; Jagdeep Walia; Christopher S Francklyn; Alkyoni Athanasiou-Fragkouli; Roisin Sullivan; Sonal Desai; Kristin Baranano; Faisal Zafar; Nuzhat Rana; Muhammed Ilyas; Alejandro Horga; Majdi Kara; Francesca Mattioli; Alice Goldenberg; Helen Griffin; Amelie Piton; Lindsay B Henderson; Benyekhlef Kara; Ayca Dilruba Aslanger; Joost Raaphorst; Rolph Pfundt; Ruben Portier; Marwan Shinawi; Amelia Kirby; Katherine M Christensen; Lu Wang; Rasim O Rosti; Sohail A Paracha; Muhammad T Sarwar; Dagan Jenkins; Jawad Ahmed; Federico A Santoni; Emmanuelle Ranza; Justyna Iwaszkiewicz; Cheryl Cytrynbaum; Rosanna Weksberg; Ingrid M Wentzensen; Maria J Guillen Sacoto; Yue Si; Aida Telegrafi; Marisa V Andrews; Dustin Baldridge; Heinz Gabriel; Julia Mohr; Barbara Oehl-Jaschkowitz; Sylvain Debard; Bruno Senger; Frédéric Fischer; Conny van Ravenwaaij; Annemarie J M Fock; Servi J C Stevens; Jürg Bähler; Amina Nasar; John F Mantovani; Adnan Manzur; Anna Sarkozy; Desirée E C Smith; Gajja S Salomons; Zubair M Ahmed; Shaikh Riazuddin; Saima Riazuddin; Muhammad A Usmani; Annette Seibt; Muhammad Ansar; Stylianos E Antonarakis; John B Vincent; Muhammad Ayub; Mona Grimmel; Anne Marie Jelsig; Tina Duelund Hjortshøj; Helena Gásdal Karstensen; Marybeth Hummel; Tobias B Haack; Yalda Jamshidi; Felix Distelmaier; Rita Horvath; Joseph G Gleeson; Hubert Becker; Jean-Louis Mandel; David A Koolen; Henry Houlden
Journal:  Am J Hum Genet       Date:  2020-07-31       Impact factor: 11.025

  5 in total

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