ArticleThe Journal of biological chemistry2013
Inactivation of ceramide synthase 6 in mice results in an altered sphingolipid metabolism and behavioral abnormalities.
Article in The Journal of biological chemistry, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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Who cites it
41 citing papers in PubMed, 90 citations in OpenAlex.
- Sphingolipids in Emotional Well-Being.Journal of neurochemistry · 2026Review
- IGFBP2-Ceramide Pathway Mediates Divergent Myelin Breakdown in the PNS and CNS Following Injury.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- How ceramides affect the development of colon cancer: from normal colon to carcinoma.Pflugers Archiv : European journal of physiology · 2024Review
- Lack of ceramide synthase 5 protects retinal ganglion cells from ocular hypertensive injury.Experimental eye research · 2024Article
- Abnormal plasma ceramides refine high-risk patients with worsening heart failure.Frontiers in cardiovascular medicine · 2023Article
- Mysterious sphingolipids: metabolic interrelationships at the center of pathophysiology.Frontiers in physiology · 2023Review
- The Role of Ceramide in Inherited Retinal Disease Pathology.Advances in experimental medicine and biology · 2023Article
- Neuronal Ganglioside and Glycosphingolipid (GSL) Metabolism and Disease : Cascades of Secondary Metabolic Errors Can Generate Complex Pathologies (in LSDs).Advances in neurobiology · 2023Article
- Ceramide synthase 6 impacts T-cell allogeneic response and graft-versus-host disease through regulating N-RAS/ERK pathway.Leukemia · 2022Article
- Article
- Comprehensive analysis of LASS6 expression and prognostic value in ovarian cancer.Journal of ovarian research · 2021Article
- Bioactive Sphingolipids as Major Regulators of Coronary Artery Disease.Biomolecules & therapeutics · 2021Review
- CERTAlzheimer's research & therapy · 2021Article
- Mutations in sphingolipid metabolism genes are associated with ADHD.Translational psychiatry · 2020Article
- The role of ceramides in metabolic disorders: when size and localization matters.Nature reviews. Endocrinology · 2020Review
- Article
- Ceramide Synthases Are Attractive Drug Targets for Treating Metabolic Diseases.Frontiers in endocrinology · 2020Review
- Sphingolipids as prognostic biomarkers of neurodegeneration, neuroinflammation, and psychiatric diseases and their emerging role in lipidomic investigation methods.Advanced drug delivery reviews · 2020Review
- Biosynthesis of long chain base in sphingolipids in animals, plants and fungi.Future science OA · 2019Review
- Alteration of Sphingolipids in Biofluids: Implications for Neurodegenerative Diseases.International journal of molecular sciences · 2019Review
Corrections and comments
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Authors and funding
14 authors at 6 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The N-acyl chain length of ceramides is determined by the specificity of different ceramide synthases (CerS). The CerS family in mammals consists of six members with different substrate specificities and expression patterns. We have generated and characterized a mouse line harboring an enzymatically inactive ceramide synthase 6 (CerS6KO) gene and lacz reporter cDNA coding for β-galactosidase directed by the CerS6 promoter. These mice display a decrease in C16:0 containing sphingolipids. Relative to wild type tissues the amount of C16:0 containing sphingomyelin in kidney is ∼35%, whereas we find a reduction of C16:0 ceramide content in the small intestine to about 25%. The CerS6KO mice show behavioral abnormalities including a clasping abnormality of their hind limbs and a habituation deficit. LacZ reporter expression in the brain reveals CerS6 expression in hippocampus, cortex, and the Purkinje cell layer of the cerebellum. Using newly developed antibodies that specifically recognize the CerS6 protein we show that the endogenous CerS6 protein is N-glycosylated and expressed in several tissues of mice, mainly kidney, small and large intestine, and brain.
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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.