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Mutation in this protein causes Huntington's disease (HD), a fatal inherited neurodegenerative disorder. Identification of Htt\u2010interacting proteins by the traditional approaches including yeast two\u2010hybrid systems and affinity purifications has greatly facilitated the understanding of Htt function. However, these methods eliminated the intracellular spatial information of the Htt interactome during sample preparations. Moreover, the temporal changes of the Htt interactome in response to acute cellular stresses cannot be easily resolved with these approaches. Ascorbate peroxidase (APEX2)\u2010based proximity labeling has been used to spatiotemporally investigate protein\u2013protein interactions in living cells. In this study, we generated stable human SH\u2010SY5Y cell lines expressing full\u2010length Htt23Q and Htt145Q with N\u2010terminus tagged Flag\u2010APEX2 to quantitatively map the spatiotemporal changes of Htt interactome to a mild acute proteotoxic stress. Our data revealed that normal and mutant Htt (muHtt) are associated with distinct intracellular microenvironments. Specifically, mutant Htt is preferentially associated with intermediate filaments and myosin complexes. Furthermore, the dynamic changes of Htt interactomes in response to stress are different between normal and mutant Htt. Vimentin is identified as one of the most significant proteins that preferentially interacts with muHtt in situ. Further functional studies demonstrated that mutant Htt affects the vimentin's function of regulating proteostasis in healthy and HD human neural stem cells. Taken together, our data offer important insights into the molecular functions of normal and mutant Htt by providing a list of Htt\u2010interacting proteins in their natural cellular context for further studies in different HD models.<jats:boxed-text content-type=\"graphic\" position=\"anchor\"><jats:graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" mimetype=\"image\/png\" position=\"anchor\" specific-use=\"enlarged-web-image\" xlink:href=\"graphic\/jnc15726-fig-0007-m.png\"><jats:alt-text>image<\/jats:alt-text><\/jats:graphic><\/jats:boxed-text><\/jats:p>","DOI":"10.1111\/jnc.15726","type":"journal-article","created":{"date-parts":[[2022,11,28]],"date-time":"2022-11-28T07:08:14Z","timestamp":1669619294000},"page":"512-528","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Characterization of huntingtin interactomes and their dynamic responses in living cells by proximity proteomics"],"prefix":"10.1111","volume":"164","author":[{"given":"Hongyuan","family":"Xu","sequence":"first","affiliation":[{"name":"Department of Biomedical Science, Charles E. Schmidt College of Medicine Florida Atlantic University  Boca Raton Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johanna","family":"Bensalel","sequence":"additional","affiliation":[{"name":"Department of Biomedical Science, Charles E. Schmidt College of Medicine Florida Atlantic University  Boca Raton Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sunil","family":"Raju","sequence":"additional","affiliation":[{"name":"Department of Biomedical Science, Charles E. Schmidt College of Medicine Florida Atlantic University  Boca Raton Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Enrico","family":"Capobianco","sequence":"additional","affiliation":[{"name":"The Jackson Laboratory  Farmington Connecticut USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael L.","family":"Lu","sequence":"additional","affiliation":[{"name":"Department of Biomedical Science, Charles E. 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