ArticleExperimental neurobiology2016
Drosophila Homolog of Human KIF22 at the Autism-Linked 16p11.2 Loci Influences Synaptic Connectivity at Larval Neuromuscular Junctions.
Article in Experimental neurobiology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 27 citations in OpenAlex.
- Research progress of kinesin family in neurological diseases.Frontiers in cellular neuroscience · 2025Review
- Dynamics of cognitive variability with age and its genetic underpinning in NIHR BioResource Genes and Cognition cohort participants.Nature medicine · 2024Article
- Understanding copy number variations through their genes: a molecular view on 16p11.2 deletion and duplication syndromes.Frontiers in pharmacology · 2024Review
- GBP2 facilitates the progression of glioma via regulation of KIF22/EGFR signaling.Cell death discovery · 2022Article
- 16pdel lipid changes in iPSC-derived neurons and function of FAM57B in lipid metabolism and synaptogenesis.iScience · 2022Article
- KIF22 promotes bladder cancer progression by activating the expression of CDCA3.International journal of molecular medicine · 2021Article
- KIF22 promotes progress of esophageal squamous cell carcinoma cells and is negatively regulated by miR-122.American journal of translational research · 2021Article
- Allele-specific open chromatin in human iPSC neurons elucidates functional disease variants.Science (New York, N.Y.) · 2020Article
- Anterograde Axonal Transport in Neuronal Homeostasis and Disease.Frontiers in molecular neuroscience · 2020Review
- Autism Spectrum Disorder-Related Syndromes: Modeling withInternational journal of molecular sciences · 2019Review
- Recessive gene disruptions in autism spectrum disorder.Nature genetics · 2019Article
- An interaction-based model for neuropsychiatric features of copy-number variants.PLoS genetics · 2019Article
- High Expression of KIF22/Kinesin-Like DNA Binding Protein (Kid) as a Poor Prognostic Factor in Prostate Cancer Patients.Medical science monitor : international medical journal of experimental and clinical research · 2018Article
- A method for estimating relative changes in the synaptic density in Drosophila central nervous system.BMC neuroscience · 2018Article
- KIF22 coordinates CAR and EGFR dynamics to promote cancer cell proliferation.Science signaling · 2018Article
- Drosophila Studies on Autism Spectrum Disorders.Neuroscience bulletin · 2017Review
- The 16p11.2 homologs fam57ba and doc2a generate certain brain and body phenotypes.Human molecular genetics · 2017Article
- MAPK3 at the Autism-Linked Human 16p11.2 Locus Influences Precise Synaptic Target Selection atMolecules and cells · 2017Article
Corrections and comments
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Authors and funding
4 authors at 3 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Copy number variations at multiple chromosomal loci, including 16p11.2, have recently been implicated in the pathogenesis of autism spectrum disorder (ASD), a neurodevelopmental disease that affects 1~3% of children worldwide. The aim of this study was to investigate the roles of human genes at the 16p11.2 loci in synaptic development using Drosophila larval neuromuscular junctions (NMJ), a well-established model synapse with stereotypic innervation patterns. We conducted a preliminary genetic screen based on RNA interference in combination with the GAL4-UAS system, followed by mutational analyses. Our result indicated that disruption of klp68D, a gene closely related to human KIF22, caused ectopic innervations of axon branches forming type III boutons in muscle 13, along with less frequent re-routing of other axon branches. In addition, mutations in klp64D, of which gene product forms Kinesin-2 complex with KLP68D, led to similar targeting errors of type III axons. Mutant phenotypes were at least partially reproduced by knockdown of each gene via RNA interference. Taken together, our data suggest the roles of Kinesin-2 proteins, including KLP68D and KLP64D, in ensuring proper synaptic wiring.
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