| Literature DB >> 27891590 |
N Orenstein1,2, K Weiss3, S N Oprescu4, R Shapira2, D Kidron2,5, L Vanagaite-Basel1,2,6,7, A Antonellis4, M Muenke3.
Abstract
Recently, bi-allelic mutations in cytosolic isoleucyl-tRNA synthetase (IARS) have been described in three individuals with growth delay, hepatic dysfunction, and neurodevelopmental disabilities. Here we report an additional subject with this condition identified by whole-exome sequencing. Our findings support the association between this disorder and neonatal cholestasis with distinct liver pathology. Furthermore, we provide functional data on two novel missense substitutions and expand the phenotype to include mild developmental delay, skin hyper-elasticity, and hypervitaminosis D.Entities:
Keywords: IARS; cholestasis; connective tissue; development; growth; vitamin D
Mesh:
Substances:
Year: 2017 PMID: 27891590 PMCID: PMC5639925 DOI: 10.1111/cge.12930
Source DB: PubMed Journal: Clin Genet ISSN: 0009-9163 Impact factor: 4.438