Progranulin mutation causes frontotemporal dementia in the Swedish Karolinska family

Alzheimers Dement. 2008 Nov;4(6):414-20. doi: 10.1016/j.jalz.2008.09.001.

Abstract

Background: Frontotemporal dementia (FTD) is a neurodegenerative disease characterized by cognitive impairment, language dysfunction, and/or changes in personality. Recently it has been shown that progranulin (GRN) mutations can cause FTD as well as other neurodegenerative phenotypes.

Methods: DNA from 30 family members, of whom seven were diagnosed with FTD, in the Karolinska family was available for GRN sequencing. Fibroblast cell mRNA from one affected family member and six control individuals was available for relative quantitative real-time polymerase chain reaction to investigate the effect of the mutation. Furthermore, the cDNA of an affected individual was sequenced.

Results: Clinical and neuropathologic findings of a previously undescribed family branch are presented. A frameshift mutation in GRN (g.102delC) was detected in all affected family members and absent in four unaffected family members older than 70 years. Real-time polymerase chain reaction data showed an approximately 50% reduction of GRN fibroblast mRNA in an affected individual. The mutated mRNA transcripts were undetectable by cDNA sequencing.

Conclusions: Segregation and RNA analyses showed that the g.102delC mutation, previously reported, causes FTD in the Karolinska family. Our findings add further support to the significance of GRN in FTD etiology and the presence of modifying genes, which emphasize the need for further studies into the mechanisms of clinical heterogeneity. However, the results already call for attention to the complexity of predictive genetic testing of GRN mutations.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age of Onset
  • Aged
  • Cognition Disorders / genetics
  • Cognition Disorders / pathology
  • Cognition Disorders / physiopathology
  • DNA Mutational Analysis
  • Dementia / genetics*
  • Dementia / pathology
  • Dementia / psychology
  • Frameshift Mutation
  • Frontal Lobe
  • Genetic Predisposition to Disease
  • Genetic Testing
  • Humans
  • Intercellular Signaling Peptides and Proteins / genetics*
  • Intercellular Signaling Peptides and Proteins / metabolism
  • Magnetic Resonance Imaging
  • Middle Aged
  • Neurodegenerative Diseases / genetics
  • Neurodegenerative Diseases / pathology
  • Neurodegenerative Diseases / physiopathology
  • Neuropsychological Tests
  • Pedigree
  • Phenotype
  • Progranulins
  • Sweden
  • Temporal Lobe
  • White People / genetics*

Substances

  • GRN protein, human
  • Intercellular Signaling Peptides and Proteins
  • Progranulins