ArticleThe American journal of pathology1988
The twitcher mouse. An alteration of the unmyelinated fibers in the PNS.
Article in The American journal of pathology, 1988. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 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.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
9 citing papers in PubMed, 26 citations in OpenAlex.
- Long-Term Improvement of Neurological Signs and Metabolic Dysfunction in a Mouse Model of Krabbe's Disease after Global Gene Therapy.Molecular therapy : the journal of the American Society of Gene Therapy · 2018Article
- Ultrastructural Characterization of the Lower Motor System in a Mouse Model of Krabbe Disease.Scientific reports · 2016Article
- Long-term Improvements in Lifespan and Pathology in CNS and PNS After BMT Plus One Intravenous Injection of AAVrh10-GALC in Twitcher Mice.Molecular therapy : the journal of the American Society of Gene Therapy · 2015Article
- Effect of vitamin D3 intake on the onset of disease in a murine model of human Krabbe disease.Journal of neuroscience research · 2015Article
- Early axonal loss accompanied by impaired endocytosis, abnormal axonal transport, and decreased microtubule stability occur in the model of Krabbe's disease.Neurobiology of disease · 2014Article
- Missense mutation in mouse GALC mimics human gene defect and offers new insights into Krabbe disease.Human molecular genetics · 2013Article
- Psychosine induces the dephosphorylation of neurofilaments by deregulation of PP1 and PP2A phosphatases.Neurobiology of disease · 2012Article
- Peripheral neuropathy in the Twitcher mouse involves the activation of axonal caspase 3.ASN neuro · 2011Review
- Molecular characterization of mutations that cause globoid cell leukodystrophy and pharmacological rescue using small molecule chemical chaperones.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2010Article
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
The twitcher is an authentic murine model of globoid cell leukodystrophy (GLD) in man. Extensive demyelination of the central and peripheral nervous systems (CNS and PNS) characterizes the neuropathologic features of GLD. In the common peroneal nerve of the twitcher, where demyelination was extensive, pronounced morphologic and quantitative alterations were noted in the unmyelinated fibers. They were 1) a large number of long and attenuated cellular processes of Schwann cells, which often enclosed only one or two axons; and 2) a threefold increase in the number of Schwann cell-axon units with reduced numbers of axons per unit. These results suggested increased branching of unmyelinated Schwann cells. Mild increase in unmyelinated fibers and mild decrease in myelinated fibers were additional features. In contrast, the sympathetic nerve trunk, which had only small numbers of myelinated and rare or no demyelinated fibers, showed much milder alterations in the unmyelinated fibers. Thus, the results of our study suggest that the alterations of the Schwann cells of the unmyelinated fibers in the twitcher are secondary to or in association with the chronic demyelinating process.
Indexed as
Identifiers
3358457PMC1880603W161813810What 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.