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In mammals, KIF1B\u03b2 is also responsible for the axonal transport of synaptic vesicle precursors. Mutations in KIF1A and KIF1B\u03b2 lead to a wide range of neuropathies. Although previous studies have revealed the biochemical, biophysical and cell biological properties of KIF1A, and its defects in neurological disorders, the fundamental properties of KIF1B\u03b2 remain elusive. In this study, we determined the motile parameters of KIF1B\u03b2 through single-molecule motility assays. We found that the C-terminal region of KIF1B\u03b2 has an inhibitory role in motor activity. AlphaFold2 prediction suggests that the C-terminal region blocks the motor domain. Additionally, we established simple methods for testing the axonal transport activity of human KIF1B\u03b2 using Caenorhabditis elegans genetics. Taking advantage of these methods, we demonstrated that these assays enable the detection of reduced KIF1B\u03b2 activities, both in vitro and in vivo, caused by a Charcot\u2013Marie\u2013Tooth disease-associated Q98L mutation.<\/jats:p>","DOI":"10.1242\/jcs.261783","type":"journal-article","created":{"date-parts":[[2024,9,16]],"date-time":"2024-09-16T08:02:25Z","timestamp":1726473745000},"source":"Crossref","is-referenced-by-count":2,"title":["Characterizing human KIF1B\u03b2 motor activity by single-molecule motility assays and <i>Caenorhabditis elegans<\/i> genetics"],"prefix":"10.1242","volume":"137","author":[{"given":"Rei","family":"Iguchi","sequence":"first","affiliation":[{"name":", Tohoku University 1 Graduate School of Life Sciences , Katahira 2-1, Aoba-ku, Sendai, Miyagi 980-8578 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomoki","family":"Kita","sequence":"additional","affiliation":[{"name":", Tohoku University 1 Graduate School of Life Sciences , Katahira 2-1, Aoba-ku, Sendai, Miyagi 980-8578 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taisei","family":"Watanabe","sequence":"additional","affiliation":[{"name":"Tohoku University 2 Department of Biology, Faculty of Science , , Aramaki-Aoba 6-3, Aoba-ku, Sendai, Miyagi 980-8578 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6919-2782","authenticated-orcid":false,"given":"Kyoko","family":"Chiba","sequence":"additional","affiliation":[{"name":", Tohoku University 3 Frontier Research Institute for Interdisciplinary Sciences (FRIS) , Aramaki-Aoba 6-3, Aoba-ku, Sendai, Miyagi 980-0845 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8367-9228","authenticated-orcid":false,"given":"Shinsuke","family":"Niwa","sequence":"additional","affiliation":[{"name":", Tohoku University 1 Graduate School of Life Sciences , Katahira 2-1, Aoba-ku, Sendai, Miyagi 980-8578 ,","place":["Japan"]},{"name":"Tohoku University 2 Department of Biology, Faculty of Science , , Aramaki-Aoba 6-3, Aoba-ku, Sendai, Miyagi 980-8578 ,","place":["Japan"]},{"name":", Tohoku University 3 Frontier Research Institute for Interdisciplinary Sciences (FRIS) , Aramaki-Aoba 6-3, Aoba-ku, Sendai, Miyagi 980-0845 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,10,10]]},"reference":[{"key":"2024101010072928200_JCS261783C1","doi-asserted-by":"publisher","first-page":"e2113795119","DOI":"10.1073\/pnas.2113795119","article-title":"De novo mutations in KIF1A-associated neuronal disorder (KAND) dominant-negatively inhibit motor activity and axonal transport of synaptic vesicle precursors","volume":"119","author":"Anazawa","year":"2022","journal-title":"Proc. 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