| Literature DB >> 31985473 |
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