Cheng Y, Zhang S, Shang H. Latest advances on new promising molecular-based therapeutic approaches for Huntington’s disease. J Transl Int Med. 2024;12(2):134–47. https://doi.org/10.2478/jtim-2023-0142.

Article  PubMed  PubMed Central  Google Scholar 

Caron NS, Wright GEB, Hayden MR. Huntington Disease. In: Adam MP, Mirzaa GM, Pagon RA, Wallace SE, Bean LJH, Gripp KW, Amemiya A, editors. GeneReviews. Seattle (WA)2020.

Finkbeiner S. Huntington’s Disease. Cold Spring Harb Perspect Biol. 2011;3(6):a007476. https://doi.org/10.1101/cshperspect.a007476.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Pengo M, Squitieri F. Beyond CAG Repeats: The Multifaceted Role of Genetics in Huntington Disease. Genes (Basel). 2024;15(6):807. https://doi.org/10.3390/genes15060807.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Jumper J, Evans R, Pritzel A, Green T, Figurnov M, Ronneberger O, et al. Highly accurate protein structure prediction with AlphaFold. Nature. 2021;596(7873):583–9. https://doi.org/10.1038/s41586-021-03819-2.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Jumper J, Hassabis D. Protein structure predictions to atomic accuracy with AlphaFold. Nat Methods. 2022;19(1):11–2. https://doi.org/10.1038/s41592-021-01362-6.

Article  CAS  PubMed  Google Scholar 

Guo Q, Bin H, Cheng J, Seefelder M, Engler T, Pfeifer G, et al. The cryo-electron microscopy structure of huntingtin. Nature. 2018;555(7694):117–20. https://doi.org/10.1038/nature25502.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Harding RJ, Deme JC, Hevler JF, Tamara S, Lemak A, Cantle JP, et al. Huntingtin structure is orchestrated by HAP40 and shows a polyglutamine expansion-specific interaction with exon 1. Commun Biol. 2021;4(1):1374. https://doi.org/10.1038/s42003-021-02895-4.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Magana Gomez PG, Kovalevskiy O. Confidence scores in AlphaFold-Multimer. Hinxton, United Kingdom: Eur Mol Biol Lab. 2023 [cited 2024 August 8].

Richard E, Michael ON, Alexander P, Natasha A, Andrew S, Tim G, et al. Protein complex prediction with AlphaFold-Multimer. bioRxiv. 2022:2021.10.04.463034. https://doi.org/10.1101/2021.10.04.463034.

Pettersen EF, Goddard TD, Huang CC, Couch GS, Greenblatt DM, Meng EC, Ferrin TE. UCSF Chimera–a visualization system for exploratory research and analysis. J Comput Chem. 2004;25(13):1605–12. https://doi.org/10.1002/jcc.20084.

Article  CAS  PubMed  Google Scholar 

Sehnal D, Bittrich S, Deshpande M, Svobodova R, Berka K, Bazgier V, et al. Mol* Viewer: modern web app for 3D visualization and analysis of large biomolecular structures. Nucleic Acids Res. 2021;49(W1):W431–7. https://doi.org/10.1093/nar/gkab314.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Brennan P. drawProteins: a Bioconductor/R package for reproducible and programmatic generation of protein schematics. F1000Res. 2018;7:1105. https://doi.org/10.12688/f1000research.14541.1.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Wickham H. ggplot2 Elegant Graphics for Data Analysis. In: Use R!,. Springer International Publishing Imprint: Springer. 2016.

R Core Team. R: A language and environment for statistical computing. Vienna, Austria: R Foundation for Statistical Computing. 2017.

Cock PJ, Antao T, Chang JT, Chapman BA, Cox CJ, Dalke A, et al. Biopython: freely available Python tools for computational molecular biology and bioinformatics. Bioinformatics. 2009;25(11):1422–3. https://doi.org/10.1093/bioinformatics/btp163.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Harris CR, Millman KJ, van der Walt SJ, Gommers R, Virtanen P, Cournapeau D, et al. Array programming with NumPy. Nature. 2020;585(7825):357–62. https://doi.org/10.1038/s41586-020-2649-2.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Caron NS, Desmond CR, Xia J, Truant R. Polyglutamine domain flexibility mediates the proximity between flanking sequences in huntingtin. Proc Natl Acad Sci U S A. 2013;110(36):14610–5. https://doi.org/10.1073/pnas.1301342110.

Article  PubMed  PubMed Central  Google Scholar 

Totzeck F, Andrade-Navarro MA, Mier P. The Protein Structure Context of PolyQ Regions. PLoS ONE. 2017;12(1):e0170801. https://doi.org/10.1371/journal.pone.0170801.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Chen S, Ferrone FA, Wetzel R. Huntington’s disease age-of-onset linked to polyglutamine aggregation nucleation. Proc Natl Acad Sci U S A. 2002;99(18):11884–9. https://doi.org/10.1073/pnas.182276099.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Bugg CW, Isas JM, Fischer T, Patterson PH, Langen R. Structural features and domain organization of huntingtin fibrils. J Biol Chem. 2012;287(38):31739–46. https://doi.org/10.1074/jbc.M112.353839.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Sivanandam VN, Jayaraman M, Hoop CL, Kodali R, Wetzel R, van der Wel PC. The aggregation-enhancing huntingtin N-terminus is helical in amyloid fibrils. J Am Chem Soc. 2011;133(12):4558–66. https://doi.org/10.1021/ja110715f.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Michalek M, Salnikov ES, Bechinger B. Structure and topology of the huntingtin 1–17 membrane anchor by a combined solution and solid-state NMR approach. Biophys J. 2013;105(3):699–710. https://doi.org/10.1016/j.bpj.2013.06.030.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Falk AS, Bravo-Arredondo JM, Varkey J, Pacheco S, Langen R, Siemer AB. Structural Model of the Proline-Rich Domain of Huntingtin Exon-1 Fibrils. Biophys J. 2020;119(10):2019–28. https://doi.org/10.1016/j.bpj.2020.10.010.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Kim MW, Chelliah Y, Kim SW, Otwinowski Z, Bezprozvanny I. Secondary structure of Huntingtin amino-terminal region. Structure. 2009;17(9):1205–12. https://doi.org/10.1016/j.str.2009.08.002.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Williamson MP. The structure and function of proline-rich regions in proteins. Biochem J. 1994;297(2):249–60. https://doi.org/10.1042/bj2970249.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Liu YF, Deth RC, Devys D. SH3 domain-dependent association of huntingtin with epidermal growth factor receptor signaling complexes. J Biol Chem. 1997;272(13):8121–4. https://doi.org/10.1074/jbc.272.13.8121.

Article  CAS  PubMed  Google Scholar 

Ceccon A, Tugarinov V, Clore GM. Quantitative Exchange NMR-Based Analysis of Huntingtin-SH3 Interactions Suggests an Allosteric Mechanism of Inhibition of Huntingtin Aggregation. J Am Chem Soc. 2021;143(25):9672–81. https://doi.org/10.1021/jacs.1c04786.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Xu S, Li G, Ye X, Chen D, Chen Z, Xu Z, et al. HAP40 is a conserved central regulator of Huntingtin and a potential modulator of Huntington’s disease pathogenesis. PLoS Genet. 2022;18(7):e1010302. https://doi.org/10.1371/journal.pgen.1010302.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Wu Y, Wang Y, Lu Y, Yan J, Zhao H, Yang R, Pan J. Research advances in huntingtin-associated protein 1 and its application prospects in disease