ArticleHuman molecular genetics2010
KBP interacts with SCG10, linking Goldberg-Shprintzen syndrome to microtubule dynamics and neuronal differentiation.
Article in Human molecular genetics, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
23 citing papers in PubMed, 41 citations in OpenAlex.
- A systems-level proteomic analysis identifies kinesin targets of KIFBP during neuronal development.bioRxiv : the preprint server for biology · 2026Article
- Rules of the road: how to turn off kinesin motors.The Biochemical journal · 2026Review
- Morpholino Knockdown in Zebrafish: A Tool to Investigate the Functional Impact of Variants of Unknown Significance in Mitochondrial Diseases.Neuromolecular medicine · 2025Review
- Microtubules, Membranes, and Movement: New Roles for Stathmin-2 in Axon Integrity.Journal of neuroscience research · 2024Review
- Review
- Cryo-EM of kinesin-binding protein: challenges and opportunities from protein-surface interactions.Acta crystallographica. Section D, Structural biology · 2021Article
- The Emerging Genetic Landscape of Hirschsprung Disease and Its Potential Clinical Applications.Frontiers in pediatrics · 2021Review
- Article
- Goldberg-Shprintzen syndrome is determined by the absence, or reduced expression levels, of KIFBP.Human mutation · 2020Article
- Scoliosis in Shprintzen-Goldberg Syndrome.Case reports in orthopedics · 2020Article
- Genetic parameters and genome-wide association study regarding feed efficiency and slaughter traits in Charolais cows.Journal of animal science · 2019Article
- Kinesin-binding protein ensures accurate chromosome segregation by buffering KIF18A and KIF15.The Journal of cell biology · 2019Article
- Multiple Functions of KBP in Neural Development Underlie Brain Anomalies in Goldberg-Shprintzen Syndrome.Frontiers in molecular neuroscience · 2019Article
- Kif1bp loss in mice leads to defects in the peripheral and central nervous system and perinatal death.Scientific reports · 2017Article
- Association of a Novel Nonsense Mutation in KIAA1279 with Goldberg-Shprintzen Syndrome.Iranian journal of child neurology · 2017Article
- Kif1B Interacts with KBP to Promote Axon Elongation by Localizing a Microtubule Regulator to Growth Cones.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2016Article
- Article
- Dynamic expression of Notch-dependent neurogenic markers in the chick embryonic nervous system.Frontiers in neuroanatomy · 2014Article
- Homozygous truncating mutation of the KBP gene, encoding a KIF1B-binding protein, in a familial case of fetal polymicrogyria.Neurogenetics · 2013Article
- Development and developmental disorders of the enteric nervous system.Nature reviews. Gastroenterology & hepatology · 2013Review
Corrections and comments
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Authors and funding
13 authors at 4 institutions in 2 countries.
Funding
Abstract
Goldberg-Shprintzen syndrome (GOSHS) is a rare clinical disorder characterized by central and enteric nervous system defects. This syndrome is caused by inactivating mutations in the Kinesin Binding Protein (KBP) gene, which encodes a protein of which the precise function is largely unclear. We show that KBP expression is up-regulated during neuronal development in mouse cortical neurons. Moreover, KBP-depleted PC12 cells were defective in nerve growth factor-induced differentiation and neurite outgrowth, suggesting that KBP is required for cell differentiation and neurite development. To identify KBP interacting proteins, we performed a yeast two-hybrid screen and found that KBP binds almost exclusively to microtubule associated or related proteins, specifically SCG10 and several kinesins. We confirmed these results by validating KBP interaction with one of these proteins: SCG10, a microtubule destabilizing protein. Zebrafish studies further demonstrated an epistatic interaction between KBP and SCG10 in vivo. To investigate the possibility of direct interaction between KBP and microtubules, we undertook co-localization and in vitro binding assays, but found no evidence of direct binding. Thus, our data indicate that KBP is involved in neuronal differentiation and that the central and enteric nervous system defects seen in GOSHS are likely caused by microtubule-related defects.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.