ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience1999
Insertion of a retrotransposon in Mbp disrupts mRNA splicing and myelination in a new mutant rat.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 1999. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.
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Who cites it
28 citing papers in PubMed, 67 citations in OpenAlex.
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- Neonatal lethality of mouse A/J-7Mammalian genome : official journal of the International Mammalian Genome Society · 2023Article
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- The Pathogenesis of Neurotrauma Indicates Targets for Neuroprotective Therapies.Current neuropharmacology · 2021Article
- The Role of Astrogliosis in Formation of the Syrinx in Spinal Cord Injury.Current neuropharmacology · 2021Review
- Rat models of human diseases and related phenotypes: a systematic inventory of the causative genes.Journal of biomedical science · 2020Review
- Article
- Myelination of Purkinje axons is critical for resilient synaptic transmission in the deep cerebellar nucleus.Scientific reports · 2018Article
- The Axon-Myelin Unit in Development and Degenerative Disease.Frontiers in neuroscience · 2018Review
- The impact of erdosteine on cisplatin-induced ototoxicity: a proteomics approach.European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery · 2017Article
- Functional and structural properties of ion channels at the nerve terminal depends on compact myelin.The Journal of physiology · 2016Article
- Inherited and acquired disorders of myelin: The underlying myelin pathology.Experimental neurology · 2016Review
- Enhanced Expression of Trib3 during the Development of Myelin Breakdown in dmy Myelin Mutant Rats.PloS one · 2016Article
- q-Space diffusion MRI (QSI) of the disease progression in the spinal cords of the Long Evans shaker: diffusion time and apparent anisotropy.NMR in biomedicine · 2013Article
- Central dysmyelination reduces the temporal fidelity of synaptic transmission and the reliability of postsynaptic firing during high-frequency stimulation.Journal of neurophysiology · 2013Article
- White matter maturation in the brains of Long Evans shaker myelin mutant rats by ex-vivo QSI and DTI.Magnetic resonance imaging · 2013Article
- Dysmyelination of auditory afferent axons increases the jitter of action potential timing during high-frequency firing.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2013Article
- Autophagy promotes oligodendrocyte survival and function following dysmyelination in a long-lived myelin mutant.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2013Article
Corrections and comments
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Authors and funding
6 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Our understanding of myelination has been greatly enhanced via the study of spontaneous mutants that harbor a defect in a gene encoding one of the major myelin proteins (myelin mutants). In this study, we describe a unique genetic defect in a new myelin mutant called the Long Evans shaker (les) rat that causes severe dysmyelination of the CNS. Myelin deficits result from disruption of the myelin basic protein (Mbp) gene caused by the insertion of an endogenous retrotransposon [early transposons (ETn) element] into a noncoding region (intron 3) of the gene. The ETn element alters the normal splicing dynamics of MBP mRNA, leading to a dramatic reduction in the levels of full-length isoforms (<5% of normal) and the appearance of improperly spliced, chimeric transcripts. Although these aberrant transcripts contain proximal coding regions of the MBP gene (exons 1-3), they are unable to encode functional proteins required to maintain the structural integrity of the myelin sheath. These chimeric transcripts seem capable, however, of producing the necessary signal to initiate and coordinate myelin gene expression because normal numbers of mature oligodendrocytes synthesizing abundant levels of other myelin proteins are present in the mutant CNS. The les rat is thus an excellent model to study alternative functions of MBP beyond its well characterized role in myelin compaction.
Indexed as
Identifiers
10212300PMC6782241W1908867576What OpenQuestion holds
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.