| Literature DB >> 29653316 |
Eline Wauters1, Sara Van Mossevelde2, Kristel Sleegers1, Julie van der Zee1, Sebastiaan Engelborghs3, Anne Sieben4, Rik Vandenberghe5, Stéphanie Philtjens1, Marleen Van den Broeck1, Karin Peeters1, Ivy Cuijt1, Wouter De Coster1, Tim Van Langenhove6, Patrick Santens6, Adrian Ivanoiu7, Patrick Cras8, Jan L De Bleecker6, Jan Versijpt9, Roeland Crols10, Nina De Klippel11, Jean-Jacques Martin12, Peter P De Deyn13, Marc Cruts1, Christine Van Broeckhoven14.
Abstract
We previously reported a granulin (GRN) null mutation, originating from a common founder, in multiple Belgian families with frontotemporal dementia. Here, we used data of a 10-year follow-up study to describe in detail the clinical heterogeneity observed in this extended founder pedigree. We identified 85 patients and 40 unaffected mutation carriers, belonging to 29 branches of the founder pedigree. Most patients (74.4%) were diagnosed with frontotemporal dementia, while others had a clinical diagnosis of unspecified dementia, Alzheimer's dementia or Parkinson's disease. The observed clinical heterogeneity can guide clinical diagnosis, genetic testing, and counseling of mutation carriers. Onset of initial symptomatology is highly variable, ranging from age 45 to 80 years. Analysis of known modifiers, suggested effects of GRN rs5848, microtubule-associated protein tau H1/H2, and chromosome 9 open reading frame 72 G4C2 repeat length on onset age but explained only a minor fraction of the variability. Contrary, the extended GRN founder family is a valuable source for identifying other onset age modifiers based on exome or genome sequences. These modifiers might be interesting targets for developing disease-modifying therapies.Entities:
Keywords: Clinical heterogeneity; Founder pedigree; Frontotemporal dementia; GRN; Modifiers
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Year: 2018 PMID: 29653316 DOI: 10.1016/j.neurobiolaging.2018.03.007
Source DB: PubMed Journal: Neurobiol Aging ISSN: 0197-4580 Impact factor: 4.673