Childhood onset in familial prion disease with a novel mutation in the PRNP gene

Arch Neurol. 2006 Jul;63(7):1016-21. doi: 10.1001/archneur.63.7.1016.

Abstract

Background: Up to 15% of cases of prion diseases are due to the autosomal dominant inheritance of coding PRNP mutations.

Objective: To describe the unique clinical and genetic findings in a family of East Indian origin with autosomal dominant inheritance of a novel PRNP mutation.

Design: Detailed neurological examination and sequencing analysis of the MAPT and PRNP genes.

Setting: Toronto Western Hospital, Toronto, Ontario.

Patients: Five available members of a family of East Indian origin with a rapidly progressive neurodegenerative disorder characterized by dementia, motor decline, and ataxia.

Results: We identified a novel Pro105Thr mutation in the PRNP gene in all of the 3 clinically affected family members but not in their unaffected relatives or normal controls. Although 5 of 6 affected family members had a relatively homogeneous phenotype and age at onset (range, 33-41 years), 1 of the 6 patients developed the disease at age 13 years.

Conclusions: A novel mutation in the PRNP gene was identified in all of the available, clinically affected members of this family with a rapidly progressive neurodegenerative disease. To our knowledge, the propositus represents the youngest individual with inherited prion disease described to date.

Publication types

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

MeSH terms

  • Adolescent
  • Adult
  • Age of Onset
  • Amyloid / genetics*
  • Base Sequence
  • DNA Mutational Analysis
  • Family Health
  • Female
  • Humans
  • Male
  • Molecular Sequence Data
  • Pedigree
  • Point Mutation*
  • Prion Diseases / genetics*
  • Prion Proteins
  • Prions
  • Protein Precursors / genetics*

Substances

  • Amyloid
  • PRNP protein, human
  • Prion Proteins
  • Prions
  • Protein Precursors