New insights into the phenotype of FARS2 deficiency

Mol Genet Metab. 2017 Dec;122(4):172-181. doi: 10.1016/j.ymgme.2017.10.004. Epub 2017 Oct 12.

Abstract

Mutations in FARS2 are known to cause dysfunction of mitochondrial translation due to deficient aminoacylation of the mitochondrial phenylalanine tRNA. Here, we report three novel mutations in FARS2 found in two patients in a compound heterozygous state. The missense mutation c.1082C>T (p.Pro361Leu) was detected in both patients. The mutations c.461C>T (p.Ala154Val) and c.521_523delTGG (p.Val174del) were each detected in one patient. We report abnormal in vitro aminoacylation assays as a functional validation of the molecular genetic findings. Based on the phenotypic data of previously reported subjects and the two subjects reported here, we conclude that FARS2 deficiency can be associated with two phenotypes: (i) an epileptic phenotype, and (ii) a spastic paraplegia phenotype.

Keywords: Early-onset epileptic encephalopathy; FARS2; Hereditary spastic paraplegia; Mitochondria; Mitochondrial aminoacyl-tRNA synthetase; Neurogenic bladder.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Amino Acyl-tRNA Synthetases / metabolism
  • Aminoacylation
  • Brain / diagnostic imaging
  • Cells, Cultured
  • Epilepsy / genetics*
  • Exome
  • Female
  • Fibroblasts / metabolism
  • Heterozygote
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Mitochondria / enzymology
  • Mitochondria / metabolism
  • Mitochondrial Proteins / deficiency*
  • Mitochondrial Proteins / genetics*
  • Muscle, Skeletal / pathology
  • Mutation, Missense / genetics
  • Oxygen Consumption
  • Phenotype*
  • Phenylalanine-tRNA Ligase / deficiency*
  • Phenylalanine-tRNA Ligase / genetics*
  • RNA, Transfer / metabolism
  • Sequence Analysis, DNA
  • Spastic Paraplegia, Hereditary / genetics*

Substances

  • Mitochondrial Proteins
  • RNA, Transfer
  • Amino Acyl-tRNA Synthetases
  • FARS2 protein, human
  • Phenylalanine-tRNA Ligase