{"doi":"10.1101/2022.08.06.502363","title":"Dual and Opposing Roles for the Kinesin-2 Motor, KIF17, in Hedgehog-dependent Cerebellar Development","abstract":"Abstract While the kinesin-2 motors KIF3A and KIF3B have essential roles in ciliogenesis and Hedgehog (HH) signal transduction, potential role(s) for another kinesin-2 motor, KIF17, in HH signaling have yet to be explored. Here, we investigated the contribution of KIF17 to HH-dependent cerebellar development, where Kif17 is expressed in both HH-producing Purkinje cells and HH-responding cerebellar granule neuron progenitors (CGNPs). Germline Kif17 deletion in mice results in cerebellar hypoplasia due to reduced CGNP proliferation, a consequence of decreased HH pathway activity mediated through decreased Sonic HH (SHH) protein. Notably, Purkinje cell-specific Kif17 deletion phenocopies Kif17 germline mutants. Surprisingly, CGNP-specific Kif17 deletion results in the opposite phenotype– increased CGNP proliferation and HH target gene expression due to altered GLI transcription factor processing. Together these data identify KIF17 as a key regulator of HH-dependent cerebellar development, with dual and opposing roles in HH-producing Purkinje cells and HH-responding CGNPS. Teaser During cerebellar development, the KIF17 microtubule motor performs opposing roles in HH-producing and HH-responding cells.","journal":"bioRxiv (Cold Spring Harbor Laboratory)","year":2022,"id":301665,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":2,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9541,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2022-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":577055,"name":"Brandon S. Carpenter","orcid":"0000-0001-9939-1926","position":1,"is_corresponding":false},{"id":993113,"name":"Olivia Q. Merchant","orcid":null,"position":2,"is_corresponding":false},{"id":281104,"name":"Kristen J. Verhey","orcid":"0000-0001-9329-4981","position":3,"is_corresponding":false},{"id":225418,"name":"Benjamin L. Allen","orcid":"0000-0003-2323-8313","position":4,"is_corresponding":false},{"id":992765,"name":"Bridget Waas","orcid":"0000-0003-1581-5985","position":0,"is_corresponding":true}],"reference_count":72,"raw_metadata":null,"created_at":"2026-07-19T00:32:02.193874Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}