| Literature DB >> 33408686 |
Madelyn Castro1, Nisha Venkateswaran1,2, Samuel T Peters1, David R Deyle3, Matthew Bower4,5, Michael D Koob6, Bradley F Boeve7, Keith Vossel1,2,8.
Abstract
Frontotemporal dementia (FTD) rarely occurs in individuals under the age of 30, and genetic causes of early-onset FTD are largely unknown. The current report follows a 27 year-old patient with no significant past medical history presenting with two years of progressive changes in behavior, rushed speech, verbal aggression, and social withdrawal. MRI and FDG-PET imaging of the brain revealed changes maximally in the frontal and temporal lobes, which along with the clinical features, are consistent with behavioral variant FTD. Next generation sequencing of a panel of 28 genes associated with dementia and amyotrophic lateral sclerosis (ALS) initially revealed a duplication of exon 15 in Matrin-3 (MATR3). Whole genome sequencing determined that this genetic anomaly was, in fact, a sequence corresponding with full-length MATR3 variant 5 inserted into chromosome 12, indicating retrotransposition from a messenger RNA intermediate. To our knowledge, this is a novel mutation of MATR3, as the majority of mutations in MATR3 linked to FTD-ALS are point mutations. Genomic DNA analysis revealed that this mutation is also present in one unaffected first-degree relative and one unaffected second-degree relative. This suggests that the mutation is either a disease-causing mutation with incomplete penetrance, which has been observed in heritable FTD, or a benign variant. Retrotransposons are not often implicated in neurodegenerative diseases; thus, it is crucial to clarify the potential role of this MATR3 variant 5 retrotransposition in early-onset FTD.Entities:
Keywords: Matrin 3; case report; frontotemporal dementia; retrotransposons; whole genome sequencing
Year: 2020 PMID: 33408686 PMCID: PMC7779795 DOI: 10.3389/fneur.2020.600468
Source DB: PubMed Journal: Front Neurol ISSN: 1664-2295 Impact factor: 4.003