دورية أكاديمية
أعرض في EDS

Genome-wide synthetic lethal CRISPR screen identifies FIS1 as a genetic interactor of ALS-linked C9ORF72.

التفاصيل البيبلوغرافية
العنوان: Genome-wide synthetic lethal CRISPR screen identifies FIS1 as a genetic interactor of ALS-linked C9ORF72.
المؤلفون: Chai N; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA; Stanford Neurosciences Graduate Program, Stanford University School of Medicine, Stanford, CA, USA., Haney MS; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Couthouis J; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Morgens DW; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Benjamin A; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Wu K; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA; Stanford Neurosciences Graduate Program, Stanford University School of Medicine, Stanford, CA, USA., Ousey J; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Fang S; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Finer S; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Bassik MC; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA., Gitler AD; Department of Genetics, Stanford University School of Medicine, Stanford, CA, USA. Electronic address: agitler@stanford.edu.
المصدر: Brain research [Brain Res] 2020 Feb 01; Vol. 1728, pp. 146601. Date of Electronic Publication: 2019 Dec 13.
نوع المنشور: Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
اللغة: English
بيانات الدورية: Publisher: Elsevier/North-Holland Biomedical Press Country of Publication: Netherlands NLM ID: 0045503 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1872-6240 (Electronic) Linking ISSN: 00068993 NLM ISO Abbreviation: Brain Res Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Amsterdam Elsevier/North-Holland Biomedical Press.
مواضيع طبية MeSH: Amyotrophic Lateral Sclerosis/*genetics , C9orf72 Protein/*genetics , Membrane Proteins/*genetics , Mitochondrial Proteins/*genetics, Clustered Regularly Interspaced Short Palindromic Repeats ; Gene Knockout Techniques/methods ; Genetic Testing ; Humans ; RNA-Seq ; Synthetic Lethal Mutations/genetics ; U937 Cells
مستخلص: Mutations in the C9ORF72 gene are the most common cause of amyotrophic lateral sclerosis (ALS). Both toxic gain of function and loss of function pathogenic mechanisms have been proposed. Accruing evidence from mouse knockout studies point to a role for C9ORF72 as a regulator of immune function. To provide further insight into its cellular function, we performed a genome-wide synthetic lethal CRISPR screen in human myeloid cells lacking C9ORF72. We discovered a strong synthetic lethal genetic interaction between C9ORF72 and FIS1, which encodes a mitochondrial membrane protein involved in mitochondrial fission and mitophagy. Mass spectrometry experiments revealed that in C9ORF72 knockout cells, FIS1 strongly bound to a class of immune regulators that activate the receptor for advanced glycation end (RAGE) products and trigger inflammatory cascades. These findings present a novel genetic interactor for C9ORF72 and suggest a compensatory role for FIS1 in suppressing inflammatory signaling in the absence of C9ORF72.
(Copyright © 2019 Elsevier B.V. All rights reserved.)
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معلومات مُعتمدة: R35 NS097263 United States NS NINDS NIH HHS; T32 HG000044 United States HG NHGRI NIH HHS; T32 MH020016 United States MH NIMH NIH HHS
فهرسة مساهمة: Keywords: C9ORF72; CRISPR; FIS1; Synthetic lethal screen
المشرفين على المادة: 0 (C9orf72 Protein)
0 (FIS1 protein, human)
0 (Membrane Proteins)
0 (Mitochondrial Proteins)
تواريخ الأحداث: Date Created: 20191218 Date Completed: 20210423 Latest Revision: 20230331
رمز التحديث: 20240628
مُعرف محوري في PubMed: PMC7539795
DOI: 10.1016/j.brainres.2019.146601
PMID: 31843624
قاعدة البيانات: MEDLINE
الوصف
تدمد:1872-6240
DOI:10.1016/j.brainres.2019.146601