Advanced search
Publication Cover
Autophagy Volume 15, 2019 - Issue 5
Submit an article Journal homepage
4,678
Views
46
CrossRef citations to date
0
Altmetric
Research Paper

The ALS-FTD-linked gene product, C9orf72, regulates neuronal morphogenesis via autophagy

Wan Yun HoDepartment of Physiology, National University of Singapore, Singapore, SingaporeView further author information
,
Yee Kit TaiDepartment of Physiology, National University of Singapore, Singapore, SingaporeORCID Iconhttp://orcid.org/0000-0003-0798-3575View further author information
ORCID Icon,
Jer-Cherng ChangDepartment of Physiology, National University of Singapore, Singapore, SingaporeView further author information
,
Jason LiangLudwig Institute for Cancer Research, University of California at San Diego, La Jolla, CA, USA;Department of Cellular and Molecular Medicine, University of California at San Diego, La Jolla, CA, USAView further author information
,
Sheue-Houy TyanDepartment of Medicine, National University of Singapore, Singapore, SingaporeView further author information
,
Song ChenDepartment of Cancer Biology, University of Cincinnati College of Medicine, Cincinnati, OH, USAView further author information
,
Jun-Lin GuanDepartment of Cancer Biology, University of Cincinnati College of Medicine, Cincinnati, OH, USAView further author information
,
Huilin ZhouLudwig Institute for Cancer Research, University of California at San Diego, La Jolla, CA, USA;Department of Cellular and Molecular Medicine, University of California at San Diego, La Jolla, CA, USAView further author information
,
Han-Ming ShenDepartment of Physiology, National University of Singapore, Singapore, SingaporeView further author information
,
Edward KooDepartment of Medicine, National University of Singapore, Singapore, Singapore;Department of Neurosciences, University of California at San Diego, La Jolla, CA, USAView further author information
&
Shuo-Chien LingDepartment of Physiology, National University of Singapore, Singapore, Singapore;Neurobiology/Ageing Programme, National University of Singapore, Singapore, Singapore;Neuroscience and Behavioural Disorders Programme, Duke-NUS Medical School, Singapore, SingaporeCorrespondence[email protected]
ORCID Iconhttp://orcid.org/0000-0002-0300-8812View further author information
ORCID Icon show all
Pages 827-842 | Received 13 Nov 2017, Accepted 18 Dec 2018, Published online: 28 Jan 2019

Citations (46)

Keep up to date with the latest research on this topic with citation updates for this article.

Subscribe to citation updates

Read on this site (4)

Lan Jiang, Tizhong Zhang, Kefeng Lu & Shiqian Qi. (2022) The progress in C9orf72 research: ALS/FTD pathogenesis, functions and structure. Small GTPases 13:1, pages 56-76.
Read now
Jimmy Beckers, Arun Kumar Tharkeshwar & Philip Van Damme. (2021) C9orf72 ALS-FTD: recent evidence for dysregulation of the autophagy-lysosome pathway at multiple levels. Autophagy 17:11, pages 3306-3322.
Read now
Alexander G Thompson, Patrick Oeckl, Emily Feneberg, Robert Bowser, Markus Otto, Roman Fischer, Benedikt Kessler & Martin R Turner. (2021) Advancing mechanistic understanding and biomarker development in amyotrophic lateral sclerosis. Expert Review of Proteomics 18:11, pages 977-994.
Read now
Patricia Richard, Shuang Feng, Yueh-Lin Tsai, Wencheng Li, Paola Rinchetti, Ubayed Muhith, Juan Irizarry-Cole, Katharine Stolz, Lionel A. Sanz, Stella Hartono, Mainul Hoque, Saba Tadesse, Hervé Seitz, Francesco Lotti, Michio Hirano, Frédéric Chédin, Bin Tian & James L. Manley. (2021) SETX (senataxin), the helicase mutated in AOA2 and ALS4, functions in autophagy regulation. Autophagy 17:8, pages 1889-1906.
Read now

Articles from other publishers (42)

Xingfeng Xu, Mao Li, Yan Su, Qi Wang, Peifang Qin, Haitao Huang, Yuting Zhang, Yali Zhou & Jianguo Yan. (2023) Cdk5 phosphorylation‐dependent C9orf72 degradation promotes neuronal death in Parkinson's disease models. CNS Neuroscience & Therapeutics 29:12, pages 3952-3966.
Crossref
Shen Zhang, Mindan Tong, Denghao Zheng, Huiying Huang, Linsen Li, Christian Ungermann, Yi Pan, Hanyan Luo, Ming Lei, Zaiming Tang, Wan Fu, She Chen, Xiaoxia Liu & Qing Zhong. (2023) C9orf72-catalyzed GTP loading of Rab39A enables HOPS-mediated membrane tethering and fusion in mammalian autophagy. Nature Communications 14:1.
Crossref
Fulya Akçimen, Elia R. Lopez, John E. Landers, Avindra Nath, Adriano Chiò, Ruth Chia & Bryan J. Traynor. (2023) Amyotrophic lateral sclerosis: translating genetic discoveries into therapies. Nature Reviews Genetics 24:9, pages 642-658.
Crossref
Zhe Wang, Xing‐Chen Wang, Ya‐Fei Chen, Chuan‐Li Wang, Le Chen, Ming‐Yue Jiang, Xi‐Wei Liu, Xiao‐Xuan Zhang, Yong‐Zhen Feng & Jin‐Hui Xu. (2023) Loss and recovery of myocardial mitochondria in mice under different tail suspension time: Apoptosis and mitochondrial fission, fusion and autophagy. Experimental Physiology.
Crossref
Poulomi Banerjee, Arpan R. Mehta, Raja S. Nirujogi, James Cooper, Owen G. James, Jyoti Nanda, James LongdenKaren BurrKarina McDadeAndrea SalzingerEvdokia Paza, Judith Newton, David StorySuvankar PalColin SmithDario R. Alessi, Bhuvaneish T. Selvaraj, Josef Priller & Siddharthan Chandran. (2023) Cell-autonomous immune dysfunction driven by disrupted autophagy in C9orf72 -ALS iPSC-derived microglia contributes to neurodegeneration . Science Advances 9:16.
Crossref
Rim Diab, Federica Pilotto & Smita Saxena. (2023) Autophagy and neurodegeneration: Unraveling the role of C9ORF72 in the regulation of autophagy and its relationship to ALS-FTD pathology. Frontiers in Cellular Neuroscience 17.
Crossref
Philip McGoldrick, Agnes Lau, Zhipeng You, Thomas M. Durcan & Janice Robertson. (2023) Loss of C9orf72 perturbs the Ran-GTPase gradient and nucleocytoplasmic transport, generating compositionally diverse Importin β-1 granules. Cell Reports 42:3, pages 112134.
Crossref
Mira Seidel, Sandeep Rajkumar, Christina Steffke, Vivien Noeth, Shreya Agarwal, Kevin Roger, Joanna Lipecka, Albert Ludolph, Chiara Ida Guerrera, Tobias Boeckers & Alberto Catanese. (2023) Propranolol reduces the accumulation of cytotoxic aggregates in C9orf72-ALS/FTD in vitro models. Current Research in Neurobiology 5, pages 100105.
Crossref
Pauline A. Gelon, Paul A. Dutchak & Chantelle F. Sephton. (2022) Synaptic dysfunction in ALS and FTD: anatomical and molecular changes provide insights into mechanisms of disease. Frontiers in Molecular Neuroscience 15.
Crossref
Ling Zou, Minru Liao, Yongqi Zhen, Shiou Zhu, Xiya Chen, Jin Zhang, Yue Hao & Bo Liu. (2022) Autophagy and beyond: Unraveling the complexity of UNC-51-like kinase 1 (ULK1) from biological functions to therapeutic implications. Acta Pharmaceutica Sinica B 12:10, pages 3743-3782.
Crossref
Layla T. Ghaffari, Davide Trotti, Aaron R. Haeusler & Brigid K. Jensen. (2022) Breakdown of the central synapses in C9orf72-linked ALS/FTD. Frontiers in Molecular Neuroscience 15.
Crossref
Claudia S. Bauer, Rebecca N. Cohen, Francesca Sironi, Matthew R. Livesey, Thomas H. Gillingwater, J. Robin Highley, Daniel J. Fillingham, Ian Coldicott, Emma F. Smith, Yolanda B. Gibson, Christopher P. Webster, Andrew J. Grierson, Caterina Bendotti & Kurt J. De Vos. (2022) An interaction between synapsin and C9orf72 regulates excitatory synapses and is impaired in ALS/FTD. Acta Neuropathologica 144:3, pages 437-464.
Crossref
Mei Pu, Yusi Tai, Luyang Yuan, Yu Zhang, Huijie Guo, Zongbing Hao, Jing Chen, Xinming Qi, Guanghui Wang, Zhouteng Tao & Jin Ren. (2022) The contribution of proteasomal impairment to autophagy activation by C9orf72 poly-GA aggregates. Cellular and Molecular Life Sciences 79:9.
Crossref
David K. Sidibe, Maria C. Vogel & Sandra Maday. (2022) Organization of the autophagy pathway in neurons. Current Opinion in Neurobiology 75, pages 102554.
Crossref
Jared S. Katzeff, Fiona Bright, Katherine Phan, Jillian J. Kril, Lars M. Ittner, Michael Kassiou, John R. Hodges, Olivier Piguet, Matthew C. Kiernan, Glenda M. Halliday & Woojin Scott Kim. (2022) Biomarker discovery and development for frontotemporal dementia and amyotrophic lateral sclerosis. Brain 145:5, pages 1598-1609.
Crossref
Romina Moavero, Angelika Mühlebner, Mark Jasper Luinenburg, Dana Craiu, Eleonora Aronica & Paolo Curatolo. (2022) Genetic pathogenesis of the epileptogenic lesions in Tuberous Sclerosis Complex: Therapeutic targeting of the mTOR pathway. Epilepsy & Behavior 131, pages 107713.
Crossref
O. H. Houghton, S. Mizielinska & P. Gomez-Suaga. (2022) The Interplay Between Autophagy and RNA Homeostasis: Implications for Amyotrophic Lateral Sclerosis and Frontotemporal Dementia. Frontiers in Cell and Developmental Biology 10.
Crossref
Sean S. Keating, Rebecca San Gil, Molly E.V. Swanson, Emma L. Scotter & Adam K. Walker. (2022) TDP-43 pathology: From noxious assembly to therapeutic removal. Progress in Neurobiology 211, pages 102229.
Crossref
Fei‐Yang Tzou, Jung‐Kun Wen, Jui‐Yu Yeh, Shu‐Yi Huang, Guang‐Chao Chen & Chih‐Chiang Chan. (2021) Drosophila as a model to study autophagy in neurodegenerative diseases and digestive tract . IUBMB Life 74:4, pages 339-360.
Crossref
Artur Schumacher-Schuh, Andrei Bieger, Wyllians V. Borelli, Makayla K. Portley, Paula Saffie Awad & Sara Bandres-Ciga. (2022) Advances in Proteomic and Metabolomic Profiling of Neurodegenerative Diseases. Frontiers in Neurology 12.
Crossref
Xiaomin Cang, Yu Wang, Jia Zeng, Jingwen Gao, Qianqian Yu, Chunfeng Lu, Feng Xu, Jiaxi Lin, Jinzhou Zhu & Xueqin Wang. (2022) C9orf72 knockdown alleviates hepatic insulin resistance by promoting lipophagy. Biochemical and Biophysical Research Communications 588, pages 15-22.
Crossref
Wan Yun Ho & Shuo-Chien Ling. 2022. Imaging and Quantifying Neuronal Autophagy. Imaging and Quantifying Neuronal Autophagy 115 133 .
Esteban Quezada, Claudio Cappelli, Iván Diaz, Nur Jury, Nicholas Wightman, Robert H. BrownJr.Jr., Martín Montecino & Brigitte van Zundert. (2021) BET bromodomain inhibitors PFI-1 and JQ1 are identified in an epigenetic compound screen to enhance C9ORF72 gene expression and shown to ameliorate C9ORF72-associated pathological and behavioral abnormalities in a C9ALS/FTD model. Clinical Epigenetics 13:1.
Crossref
Daniel J Klionsky, Giulia Petroni, Ravi K Amaravadi, Eric H Baehrecke, Andrea Ballabio, Patricia Boya, José Manuel Bravo‐San Pedro, Ken Cadwell, Francesco Cecconi, Augustine M K Choi, Mary E Choi, Charleen T Chu, Patrice Codogno, Maria Isabel Colombo, Ana Maria Cuervo, Vojo Deretic, Ivan Dikic, Zvulun Elazar, Eeva‐Liisa Eskelinen, Gian Maria Fimia, David A Gewirtz, Douglas R Green, Malene Hansen, Marja Jäättelä, Terje Johansen, Gábor Juhász, Vassiliki Karantza, Claudine Kraft, Guido Kroemer, Nicholas T Ktistakis, Sharad Kumar, Carlos Lopez‐Otin, Kay F Macleod, Frank Madeo, Jennifer Martinez, Alicia Meléndez, Noboru Mizushima, Christian Münz, Josef M Penninger, Rushika M Perera, Mauro Piacentini, Fulvio Reggiori, David C Rubinsztein, Kevin M Ryan, Junichi Sadoshima, Laura Santambrogio, Luca Scorrano, Hans‐Uwe Simon, Anna Katharina Simon, Anne Simonsen, Alexandra Stolz, Nektarios Tavernarakis, Sharon A Tooze, Tamotsu Yoshimori, Junying Yuan, Zhenyu Yue, Qing Zhong, Lorenzo Galluzzi & Federico Pietrocola. (2021) Autophagy in major human diseases. The EMBO Journal 40:19.
Crossref
Junsheng Chen, Arthur Bassot, Fabrizio Giuliani & Thomas Simmen. (2021) Amyotrophic Lateral Sclerosis (ALS): Stressed by Dysfunctional Mitochondria-Endoplasmic Reticulum Contacts (MERCs). Cells 10:7, pages 1789.
Crossref
Deepti Lall, Ileana Lorenzini, Thomas A. Mota, Shaughn Bell, Thomas E. Mahan, Jason D. Ulrich, Hayk Davtyan, Jessica E. Rexach, A.K.M. Ghulam Muhammad, Oksana Shelest, Jesse Landeros, Michael Vazquez, Junwon Kim, Layla Ghaffari, Jacqueline Gire O’Rourke, Daniel H. Geschwind, Mathew Blurton-Jones, David M. Holtzman, Rita Sattler & Robert H. Baloh. (2021) C9orf72 deficiency promotes microglial-mediated synaptic loss in aging and amyloid accumulation. Neuron 109:14, pages 2275-2291.e8.
Crossref
Jessica Root, Paola Merino, Austin Nuckols, Michelle Johnson & Thomas Kukar. (2021) Lysosome dysfunction as a cause of neurodegenerative diseases: Lessons from frontotemporal dementia and amyotrophic lateral sclerosis. Neurobiology of Disease 154, pages 105360.
Crossref
Weilun Pang & Fenghua Hu. (2020) Cellular and physiological functions of C9ORF72 and implications for ALS/FTD. Journal of Neurochemistry 157:3, pages 334-350.
Crossref
Sara Saez-Atienzar, Sara Bandres-CigaRebekah G. Langston, Jonggeol J. KimShing Wan ChoiRegina H. Reynolds, Yevgeniya AbramzonRamita Dewan, Sarah Ahmed, John E. Landers, Ruth Chia, Mina Ryten, Mark R. Cookson, Michael A. NallsAdriano Chiò & Bryan J. Traynor. (2021) Genetic analysis of amyotrophic lateral sclerosis identifies contributing pathways and cell types. Science Advances 7:3.
Crossref
Olubankole Aladesuyi Arogundade, Jie Jiang & John Ravits. 2021. Neurotherapeutics in the Era of Translational Medicine. Neurotherapeutics in the Era of Translational Medicine 19 47 .
Wan Yun Ho, Sheeja Navakkode, Fujia Liu, Tuck Wah Soong & Shuo-Chien Ling. (2020) Deregulated expression of a longevity gene, Klotho, in the C9orf72 deletion mice with impaired synaptic plasticity and adult hippocampal neurogenesis. Acta Neuropathologica Communications 8:1.
Crossref
Garam Kim, Olivia Gautier, Eduardo Tassoni-Tsuchida, X. Rosa Ma & Aaron D. Gitler. (2020) ALS Genetics: Gains, Losses, and Implications for Future Therapies. Neuron 108:5, pages 822-842.
Crossref
Chang Geon Chung, Sung Soon Park, Jeong Hyang Park & Sung Bae Lee. (2020) Dysregulated Plasma Membrane Turnover Underlying Dendritic Pathology in Neurodegenerative Diseases. Frontiers in Cellular Neuroscience 14.
Crossref
Yifei Xie, Jing Zhang, Bingbing Lu, Zhuo Bao, Jimin Zhao, Xianyu Lu, Yaxing Wei, Ke Yao, Yanan Jiang, Qiang Yuan, Xiaofan Zhang, Bo Li, Xinhuan Chen, Zigang Dong & Kangdong Liu. (2020) Mefloquine Inhibits Esophageal Squamous Cell Carcinoma Tumor Growth by Inducing Mitochondrial Autophagy. Frontiers in Oncology 10.
Crossref
Fiona Limanaqi, Carla Letizia Busceti, Francesca Biagioni, Federica Cantini, Paola Lenzi & Francesco Fornai. (2020) Cell-Clearing Systems Bridging Repeat Expansion Proteotoxicity and Neuromuscular Junction Alterations in ALS and SBMA. International Journal of Molecular Sciences 21:11, pages 4021.
Crossref
Qiang Zhu, Jie Jiang, Tania F. Gendron, Melissa McAlonis-Downes, Lulin Jiang, Amy Taylor, Sandra Diaz Garcia, Somasish Ghosh Dastidar, Maria J. Rodriguez, Patrick King, Yongjie Zhang, Albert R. La Spada, Huaxi Xu, Leonard Petrucelli, John Ravits, Sandrine Da Cruz, Clotilde Lagier-Tourenne & Don W. Cleveland. (2020) Reduced C9ORF72 function exacerbates gain of toxicity from ALS/FTD-causing repeat expansion in C9orf72. Nature Neuroscience 23:5, pages 615-624.
Crossref
Elena Seranova, Adina Maria Palhegyi, Surbhi Verma, Simona Dimova, Rachel Lasry, Moriyah Naama, Congxin Sun, Timothy Barrett, Tatiana Rosado Rosenstock, Dhiraj Kumar, Malkiel A. Cohen, Yosef Buganim & Sovan Sarkar. (2020) Human Induced Pluripotent Stem Cell Models of Neurodegenerative Disorders for Studying the Biomedical Implications of Autophagy. Journal of Molecular Biology 432:8, pages 2754-2798.
Crossref
Manon Boivin, Véronique Pfister, Angeline Gaucherot, Frank Ruffenach, Luc Negroni, Chantal Sellier & Nicolas Charlet‐Berguerand. (2020) Reduced autophagy upon C9ORF72 loss synergizes with dipeptide repeat protein toxicity in G4C2 repeat expansion disorders. The EMBO Journal 39:4.
Crossref
Emiliano Vicencio, Sebastián Beltrán, Luis Labrador, Patricio Manque, Melissa Nassif & Ute Woehlbier. (2020) Implications of Selective Autophagy Dysfunction for ALS Pathology. Cells 9:2, pages 381.
Crossref
Christopher P. Cali, Maribel Patino, Yee Kit Tai, Wan Yun Ho, Catriona A. McLean, Christopher M. Morris, William W. Seeley, Bruce L. Miller, Carles Gaig, Jean Paul G. Vonsattel, Charles L. WhiteIIIIII, Sigrun Roeber, Hans Kretzschmar, Juan C. Troncoso, Claire Troakes, Marla Gearing, Bernardino Ghetti, Vivianna M. Van Deerlin, Virginia M.-Y. Lee, John Q. Trojanowski, Kin Y. Mok, Helen Ling, Dennis W. Dickson, Gerard D. Schellenberg, Shuo-Chien Ling & Edward B. Lee. (2019) C9orf72 intermediate repeats are associated with corticobasal degeneration, increased C9orf72 expression and disruption of autophagy. Acta Neuropathologica 138:5, pages 795-811.
Crossref
Jie Jiang & John Ravits. (2019) Pathogenic Mechanisms and Therapy Development for C9orf72 Amyotrophic Lateral Sclerosis/Frontotemporal Dementia. Neurotherapeutics 16:4, pages 1115-1132.
Crossref
Udaya Geetha Vijayakumar, Vanessa Milla, Mei Yu Cynthia Stafford, Anthony J. Bjourson, William Duddy & Stephanie Marie-Rose Duguez. (2019) A Systematic Review of Suggested Molecular Strata, Biomarkers and Their Tissue Sources in ALS. Frontiers in Neurology 10.
Crossref

Related research

People also read lists articles that other readers of this article have read.

Recommended articles lists articles that we recommend and is powered by our AI driven recommendation engine.

Cited by lists all citing articles based on Crossref citations.
Articles with the Crossref icon will open in a new tab.