Abstract
Mutation screening and phenotypic profiling of 2 amyotrophic lateral sclerosis-(ALS) and frontotemporal dementia-(FTD) associated genes, CHCHD10 and TUBA4A, were performed in a Belgian cohort of 459 FTD, 28 FTD-ALS, and 429 ALS patients. In CHCHD10, we identified a novel nonsense mutation (p.Gln108*) in a patient with atypical clinical FTD and pathology-confirmed Parkinson's disease (1/459, 0.22%) leading to loss of transcript. We further observed 3 previously described missense variants (p.Pro34Ser, p.Pro80Leu, and p.Pro96Thr) that were also present in the matched control series. In TUBA4A, we detected a novel frameshift mutation (p.Arg64Glyfs*90) leading to a truncated protein in 1 FTD patient (1/459 of 0.22%) with family history of Parkinson's disease and cognitive impairment, and a novel missense mutation (p.Thr381Met) in 2 sibs with familial ALS and memory problems (1 index patient/429, 0.23%) in whom we previously identified a pathogenic Chromosome 9 open reading frame 72 repeat expansion mutation. The present study confirms the role of CHCHD10 and TUBA4A in the FTD-ALS spectrum, although genetic variations in these 2 genes are extremely rare in the Belgian population and often associated with symptomatology of related neurodegenerative diseases including Parkinson's disease and Alzheimer's disease.
| Original language | English |
|---|---|
| Pages (from-to) | 177.e9-177.e16 |
| Number of pages | 8 |
| Journal | Neurobiology of Aging |
| Volume | 51 |
| DOIs | |
| Publication status | Published - Mar 2017 |
Bibliographical note
Copyright © 2017 Elsevier Inc. All rights reserved.Keywords
- Aged
- Aged, 80 and over
- Amyotrophic Lateral Sclerosis/genetics
- Belgium
- Cohort Studies
- Female
- Frontotemporal Dementia/genetics
- Genetic Association Studies
- Humans
- Male
- Middle Aged
- Mitochondrial Proteins/genetics
- Mutation
- Tubulin/genetics
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