Literature DB >> 33408686

Case Report: Early-Onset Behavioral Variant Frontotemporal Dementia in Patient With Retrotransposed Full-Length Transcript of Matrin-3 Variant 5.

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.
Copyright © 2020 Castro, Venkateswaran, Peters, Deyle, Bower, Koob, Boeve and Vossel.

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


  3 in total

Review 1.  Multisystem Proteinopathy Due to VCP Mutations: A Review of Clinical Heterogeneity and Genetic Diagnosis.

Authors:  Gerald Pfeffer; Grace Lee; Carly S Pontifex; Roberto D Fanganiello; Allison Peck; Conrad C Weihl; Virginia Kimonis
Journal:  Genes (Basel)       Date:  2022-05-27       Impact factor: 4.141

2.  Duplication of exons 15 and 16 in Matrin-3: a phenotype bridging amyotrophic lateral sclerosis and immune-mediated disorders.

Authors:  Maria Caputo; Elisabetta Zucchi; Ilaria Martinelli; Giulia Gianferrari; Cecilia Simonini; Amedeo Amedei; Elena Niccolai; Cinzia Gellera; Viviana Pensato; Jessica Mandrioli
Journal:  Neurol Sci       Date:  2021-10-19       Impact factor: 3.830

3.  An Indonesian elderly with primary progressive aphasia and behavioral variant of frontotemporal dementia: A case report and review article.

Authors:  Aditya Kusumo Riswanto; Wendy Amelia Sihombing; Yudha Haryono
Journal:  Ann Med Surg (Lond)       Date:  2022-09-02
  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.