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Knockdown of SIRT2 Rescues YARS-induced Charcot-Marie-Tooth Neuropathy in Drosophila

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References

  1. Catala M, Kubis N. Gross anatomy and development of the peripheral nervous system. Handb Clin Neurol 2013, 115: 29–41.

    Article  PubMed  Google Scholar 

  2. Pipis M, Rossor AM, Laura M, Reilly MM. Next-generation sequencing in Charcot-Marie-Tooth disease: Opportunities and challenges. Nat Rev Neurol 2019, 15: 644–656.

    Article  PubMed  Google Scholar 

  3. Froelich CA, First EA. Dominant intermediate Charcot-Marie-tooth disorder is not due to a catalytic defect in tyrosyl-tRNA synthetase. Biochemistry 2011, 50: 7132–7145.

    Article  CAS  PubMed  Google Scholar 

  4. Blocquel D, Li S, Wei N, Daub H, Sajish M, Erfurth ML. Alternative stable conformation capable of protein misinteraction links tRNA synthetase to peripheral neuropathy. Nucleic Acids Res 2017, 45: 8091–8104.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  5. Matsuyama A, Shimazu T, Sumida Y, Saito A, Yoshimatsu Y, Seigneurin-Berny D, et al. In vivo destabilization of dynamic microtubules by HDAC6-mediated deacetylation. EMBO J 2002, 21: 6820–6831.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  6. Saxena S, Caroni P. Selective neuronal vulnerability in neurodegenerative diseases: From stressor thresholds to degeneration. Neuron 2011, 71: 35–48.

    Article  CAS  PubMed  Google Scholar 

  7. Skoge RH, Dölle C, Ziegler M. Regulation of SIRT2-dependent α-tubulin deacetylation by cellular NAD levels. DNA Repair (Amst) 2014, 23: 33–38.

    Article  CAS  PubMed  Google Scholar 

  8. North BJ, Marshall BL, Borra MT, Denu JM, Verdin E. The human Sir2 ortholog, SIRT2, is an NAD+-dependent tubulin deacetylase. Mol Cell 2003, 11: 437–444.

    Article  CAS  PubMed  Google Scholar 

  9. Outeiro TF, Kontopoulos E, Altmann SM, Kufareva I, Strathearn KE, Amore AM, et al. Sirtuin 2 inhibitors rescue alpha-synuclein-mediated toxicity in models of Parkinson’s disease. Science 2007, 317: 516–519.

    Article  CAS  PubMed  Google Scholar 

  10. Luthi-Carter R, Taylor DM, Pallos J, Lambert E, Amore A, Parker A, et al. SIRT2 inhibition achieves neuroprotection by decreasing sterol biosynthesis. Proc Natl Acad Sci U S A 2010, 107: 7927–7932.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  11. Zhao Y, Xie L, Shen C, Qi Q, Qin Y, Xing J, et al. SIRT2-knockdown rescues GARS-induced Charcot-Marie-tooth neuropathy. Aging Cell 2021, 20: e13391.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  12. Wei N, Zhang Q, Yang XL. Neurodegenerative Charcot-Marie-Tooth disease as a case study to decipher novel functions of aminoacyl-tRNA synthetases. J Biol Chem 2019, 294: 5321–5339.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  13. Storkebaum E, Leitão-Gonçalves R, Godenschwege T, Nangle L, Mejia M, Bosmans I, et al. Dominant mutations in the tyrosyl-tRNA synthetase gene recapitulate in Drosophila features of human Charcot-Marie-Tooth neuropathy. Proc Natl Acad Sci U S A 2009, 106: 11782–11787.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  14. Xu Z, Schaedel L, Portran D, Aguilar A, Gaillard J, Xu Z, et al. Microtubules acquire resistance from mechanical breakage through intralumenal acetylation. Science 2017, 356: 328–332.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  15. Godena VK, Brookes-Hocking N, Moller A, Shaw G, Oswald M, Sancho RM, et al. Increasing microtubule acetylation rescues axonal transport and locomotor deficits caused by LRRK2 Roc-COR domain mutations. Nat Commun 2014, 5: 5245.

    Article  CAS  PubMed  Google Scholar 

  16. Bobrowska A, Donmez G, Weiss A, Guarente L, Bates G. SIRT2 ablation has no effect on tubulin acetylation in brain, cholesterol biosynthesis or the progression of Huntington’s disease phenotypes in vivo. PLoS One 2012, 7: e34805.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  17. Li W, Zhang B, Tang J, Cao Q, Wu Y, Wu C, et al. Sirtuin 2, a mammalian homolog of yeast silent information regulator-2 longevity regulator, is an oligodendroglial protein that decelerates cell differentiation through deacetylating alpha-tubulin. J Neurosci 2007, 27: 2606–2616.

    Article  PubMed  PubMed Central  Google Scholar 

  18. Niu X, Mao CX, Wang S, Wang X, Zhang Y, Hu J, et al. α-Tubulin acetylation at lysine 40 regulates dendritic arborization and larval locomotion by promoting microtubule stability in Drosophila. PLoS One 2023, 18: e0280573.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

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Acknowledgments

We thank members of the Yu Lab for technical support throughout this study. NSC-34 motor neuron cells were gifted from Dr. Yanshan Fang at the Shanghai Institute of Organic Chemistry, CAS. The dYARS transgenic fly lines described in this paper were kindly gifted by Prof. Erik Storkebaum at the Max Planck Institute for Molecular Biomedicine. The tubK40Q transgenic fly lines were kindly gifted by Prof. Liu Zhihua at Hubei University. We thank the facility center of the State Key Laboratory of Genetic Engineering for assistance with enzymatic activity experiments. This work was supported by the National Natural Science Foundation of China (32370825, 92249302, 92049301, and 31821002) and the Hubei Provincial Natural Science Foundation 2020CFB668.

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Author notes

  1. Xuedong Li, Mengrong Wang, and Xiang Gao contributed equally to this work.

Authors and Affiliations

  1. State Key Laboratory of Genetic Engineering, School of Life Sciences, Zhongshan Hospital, Fudan University, Shanghai, 200438, China

    Xuedong Li, Mengrong Wang, Chenyu Li, Chunyu Chen, Yun Qi & Wei Yu

  2. School of Basic Medic Science, Southwest Medical University, Luzhou, 64600, China

    Xuedong Li & Ying Wan

  3. Department of Geriatrics, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, 430030, China

    Xiang Gao

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  1. Xuedong Li
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  4. Chenyu Li
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Correspondence to Ying Wan or Wei Yu.

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Li, X., Wang, M., Gao, X. et al. Knockdown of SIRT2 Rescues YARS-induced Charcot-Marie-Tooth Neuropathy in Drosophila. Neurosci. Bull. 40, 539–543 (2024). https://doi.org/10.1007/s12264-023-01156-0

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