Literature DB >> 33871358

Downregulation of glial genes involved in synaptic function mitigates Huntington's disease pathogenesis.

Tarik Seref Onur1,2,3, Andrew Laitman2,4,5, He Zhao2, Ryan Keyho2, Hyemin Kim2, Jennifer Wang2, Megan Mair1,2,3, Huilan Wang6, Lifang Li1,2, Alma Perez2, Maria de Haro1,2, Ying-Wooi Wan2, Genevera Allen2,7, Boxun Lu6, Ismael Al-Ramahi1,2, Zhandong Liu2,4,5, Juan Botas1,2,3,4.   

Abstract

Most research on neurodegenerative diseases has focused on neurons, yet glia help form and maintain the synapses whose loss is so prominent in these conditions. To investigate the contributions of glia to Huntington's disease (HD), we profiled the gene expression alterations of Drosophila expressing human mutant Huntingtin (mHTT) in either glia or neurons and compared these changes to what is observed in HD human and HD mice striata. A large portion of conserved genes are concordantly dysregulated across the three species; we tested these genes in a high-throughput behavioral assay and found that downregulation of genes involved in synapse assembly mitigated pathogenesis and behavioral deficits. To our surprise, reducing dNRXN3 function in glia was sufficient to improve the phenotype of flies expressing mHTT in neurons, suggesting that mHTT's toxic effects in glia ramify throughout the brain. This supports a model in which dampening synaptic function is protective because it attenuates the excitotoxicity that characterizes HD.
© 2021, Onur et al.

Entities:  

Keywords:  D. melanogaster; Huntington's disease; computational biology; excitotoxicity; genetics; genomics; glia; high-throughput experimentation; human; mouse; neurodegeneration; synaptic biology; systems biology

Year:  2021        PMID: 33871358     DOI: 10.7554/eLife.64564

Source DB:  PubMed          Journal:  Elife        ISSN: 2050-084X            Impact factor:   8.140


  5 in total

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Journal:  Biomedicines       Date:  2022-05-23

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Journal:  Cell Genom       Date:  2022-07-26

3.  Dynamics of huntingtin protein interactions in the striatum identifies candidate modifiers of Huntington disease.

Authors:  Todd M Greco; Christopher Secker; Eduardo Silva Ramos; Joel D Federspiel; Jeh-Ping Liu; Alma M Perez; Ismael Al-Ramahi; Jeffrey P Cantle; Jeffrey B Carroll; Juan Botas; Scott O Zeitlin; Erich E Wanker; Ileana M Cristea
Journal:  Cell Syst       Date:  2022-02-10       Impact factor: 11.091

4.  Genes and Diseases: Insights from Transcriptomics Studies.

Authors:  Dmitry S Kolobkov; Darya A Sviridova; Serikbai K Abilev; Artem N Kuzovlev; Lyubov E Salnikova
Journal:  Genes (Basel)       Date:  2022-06-28       Impact factor: 4.141

5.  Discovery of levodopa-induced dyskinesia-associated genes using genomic studies in patients and Drosophila behavioral analyses.

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  5 in total

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