ArticlePloS one2010
Neural and synaptic defects in slytherin, a zebrafish model for human congenital disorders of glycosylation.
Article in PloS one, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers, 1 of them a synthesis that pooled it.
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Who cites it
23 citing papers in PubMed, 1 synthesis or guideline pooled it.
- Zebrafish models for congenital disorders of glycosylation (CDG): a systematic review.Orphanet journal of rare diseases · 2025Pooled it
- INHAND: Non-proliferative and proliferative lesions of fish - Chapter 8. Fish nervous system.Journal of toxicologic pathology · 2026Article
- Mutation of the GDP-Fucose Biosynthesis GeneInternational journal of molecular sciences · 2025Article
- Review
- Candidate SNP Markers Significantly Altering the Affinity of the TATA-Binding Protein for the Promoters of Human Genes Associated with Primary Open-Angle Glaucoma.International journal of molecular sciences · 2024Article
- Genetic diversity, demographic history, and selective signatures of Silkie chicken.BMC genomics · 2024Article
- Altered mannose metabolism in chronic stress and depression is rapidly reversed by vitamin B12.Frontiers in nutrition · 2022Article
- Effect of NTN and Lmx1Parkinson's disease · 2021Article
- Post-Developmental Roles of Notch Signaling in the Nervous System.Biomolecules · 2020Review
- Systems glycomics of adult zebrafish identifies organ-specific sialylation and glycosylation patterns.Nature communications · 2018Article
- CDG Therapies: From Bench to Bedside.International journal of molecular sciences · 2018Review
- Notch1 Signaling Activation Contributes to Adult Hippocampal Neurogenesis Following Traumatic Brain Injury.Medical science monitor : international medical journal of experimental and clinical research · 2017Article
- Molecular cytogenetic characterisation of a novelMolecular cytogenetics · 2017Article
- Molecular characterization of a novel ring 6 chromosome using next generation sequencing.Molecular cytogenetics · 2016Article
- Disruptions in a cluster of computationally identified enhancers near FOXC1 and GMDS may influence brain development.Neurogenetics · 2016Article
- Article
- In silico analysis of the fucosylation-associated genome of the human blood fluke Schistosoma mansoni: cloning and characterization of the enzymes involved in GDP-L-fucose synthesis and Golgi import.Parasites & vectors · 2013Article
- "Casting" light on the role of glycosylation during embryonic development: insights from zebrafish.Glycoconjugate journal · 2013Review
- Toward developmental models of psychiatric disorders in zebrafish.Frontiers in neural circuits · 2013Review
- Therapies and therapeutic approaches in Congenital Disorders of Glycosylation.Glycoconjugate journal · 2013Review
Corrections and comments
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Authors and funding
9 authors.
Funding
Abstract
Congenital disorder of glycosylation type IIc (CDG IIc) is characterized by mental retardation, slowed growth and severe immunodeficiency, attributed to the lack of fucosylated glycoproteins. While impaired Notch signaling has been implicated in some aspects of CDG IIc pathogenesis, the molecular and cellular mechanisms remain poorly understood. We have identified a zebrafish mutant slytherin (srn), which harbors a missense point mutation in GDP-mannose 4,6 dehydratase (GMDS), the rate-limiting enzyme in protein fucosylation, including that of Notch. Here we report that some of the mechanisms underlying the neural phenotypes in srn and in CGD IIc are Notch-dependent, while others are Notch-independent. We show, for the first time in a vertebrate in vivo, that defects in protein fucosylation leads to defects in neuronal differentiation, maintenance, axon branching, and synapse formation. Srn is thus a useful and important vertebrate model for human CDG IIc that has provided new insights into the neural phenotypes that are hallmarks of the human disorder and has also highlighted the role of protein fucosylation in neural development.
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Registered trials
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