Compound heterozygosity for novel variations of the NHLRC1 Gene in a family with Lafora disease

Clin Neurol Neurosurg. 2022 Jul:218:107255. doi: 10.1016/j.clineuro.2022.107255. Epub 2022 Apr 25.

Abstract

Purpose: NHLRC1 gene mutations are present in a varied proportion of patients with Lafora disease (LD). Compound heterozygosity for novel variations of the gene has been reported in progressive Lafora myoclonic epilepsy of Lafora pedigree.

Methods: The clinical data of the cases were collected for diagnosis, and the genetic spectrum of the family was confirmed. For molecular diagnosis, whole-exome sequencing (WES) of the pedigree was performed.

Results: A novel biallelic compound heterozygous c.333dupC chr6-18122504 (p.(Gly112ArgfsTer44)) and c.612dupT chr6-18122225 (p.(Gly205Trpfs*29)) mutation in the NHLRC1 gene was identified in our progressive myoclonic epilepsy of Lafora pedigree.

Conclusions: The genetic analysis was useful for the diagnosis of LD. Genetic analysis is recommended for patients and close relatives, and tissue biopsy is an alternative.

Keywords: Epilepsy; Heredity; Heterozygous variant; Lafora disease; NHLRC1 gene.

Publication types

  • Case Reports

MeSH terms

  • Carrier Proteins / genetics
  • Humans
  • Lafora Disease* / diagnosis
  • Lafora Disease* / genetics
  • Lafora Disease* / pathology
  • Mutation
  • Pedigree
  • Protein Tyrosine Phosphatases, Non-Receptor / genetics
  • Protein Tyrosine Phosphatases, Non-Receptor / metabolism
  • Ubiquitin-Protein Ligases / genetics

Substances

  • Carrier Proteins
  • NHLRC1 protein, human
  • Ubiquitin-Protein Ligases
  • Protein Tyrosine Phosphatases, Non-Receptor