Hereditary Myoclonus Dystonia: A Novel SGCE Variant and Phenotype Including Intellectual Disability

Tremor Other Hyperkinet Mov (N Y). 2018 Mar 28:8:547. doi: 10.7916/D8J11FRZ. eCollection 2018.

Abstract

Background: Hereditary myoclonus dystonia is often due to changes in the SGCE gene. Dystonia (DYT)-SGCE has a variable phenotype that can involve focal or generalized myoclonus and various forms of task-specific, segmental, or generalized dystonia. Psychiatric comorbidities are common.

Case report: We report a case of a young woman with generalized myoclonus, dystonia, and intellectual disability. She was found to have a novel SGCE splice site variant.

Discussion: This novel variant is very likely pathogenic by in silico analysis and has not been previously reported. Additionally, her intellectual disability may constitute a novel phenotype for patients with SGCE variants.

Keywords: DYT11; SGCE; intellectual disability; myoclonus dystonia; sarcoglycan.

Publication types

  • Case Reports
  • Video-Audio Media

MeSH terms

  • Dystonic Disorders / genetics*
  • Dystonic Disorders / physiopathology*
  • Dystonic Disorders / therapy
  • Female
  • Genetic Variation
  • Humans
  • Intellectual Disability / genetics
  • Intellectual Disability / physiopathology
  • Phenotype
  • Sarcoglycans / genetics*
  • Young Adult

Substances

  • SGCE protein, human
  • Sarcoglycans

Supplementary concepts

  • Myoclonic dystonia