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http://purl.uniprot.org/citations/35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#typehttp://purl.uniprot.org/core/Journal_Citation
http://purl.uniprot.org/citations/35132656http://www.w3.org/2000/01/rdf-schema#comment"The mechanochemical coupling of ATPase hydrolysis and conformational dynamics in kinesin motors facilitates intramolecular interaction cycles between the kinesin motor and neck domains, which are essential for microtubule-based motility. Here, we characterized a charge-inverting KIF1A-E239K mutant that we identified in a family with axonal-type Charcot-Marie-Tooth disease and also in 24 cases in human neuropathies including spastic paraplegia and hereditary sensory and autonomic neuropathy. We show that Glu239 in the β7 strand is a key residue of the motor domain that regulates the motor-neck interaction. Expression of the KIF1A-E239K mutation has decreased ability to complement Kif1a+/- neurons, and significantly decreases ATPase activity and microtubule gliding velocity. X-ray crystallography shows that this mutation causes an excess positive charge on β7, which may electrostatically interact with a negative charge on the neck. Quantitative mass spectrometric analysis supports that the mutation hyper-stabilizes the motor-neck interaction at the late ATP hydrolysis stage. Thus, the negative charge of Glu239 dynamically regulates the kinesin motor-neck interaction, promoting release of the neck from the motor domain upon ATP hydrolysis."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.org/dc/terms/identifier"doi:10.15252/embj.2021108899"xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Hirokawa N."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Ogawa T."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Tanaka Y."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Morikawa M."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Zuchner S."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Shy M.E."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Jerath N.U."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/author"Morikawa M.'"xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/date"2022"xsd:gYear
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/name"EMBO J"xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/pages"e108899"xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/title"A neuropathy-associated kinesin KIF1A mutation hyper-stabilizes the motor-neck interaction during the ATPase cycle."xsd:string
http://purl.uniprot.org/citations/35132656http://purl.uniprot.org/core/volume"41"xsd:string
http://purl.uniprot.org/citations/35132656http://www.w3.org/2004/02/skos/core#exactMatchhttp://purl.uniprot.org/pubmed/35132656
http://purl.uniprot.org/citations/35132656http://xmlns.com/foaf/0.1/primaryTopicOfhttps://pubmed.ncbi.nlm.nih.gov/35132656
http://purl.uniprot.org/uniprot/#_A0A3B3IT28-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_A0A3B3ITE5-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_A0A3B3IU40-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_A0A6H0DX63-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_G1UI30-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_Q53T79-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656
http://purl.uniprot.org/uniprot/#_P33173-mappedCitation-35132656http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/35132656