Literature DB >> 29584714

Clemastine effects in rat models of a myelination disorder.

Christopher A Turski1, Gabrielle N Turski2, Bingming Chen3, Hauhui Wang4, Moones Heidari5, Lingjun Li3, Kevin K Noguchi4, Cara Westmark1, Ian Duncan5, Chrysanthy Ikonomidou1.   

Abstract

BackgroundPelizaeus Merzbacher disease (PMD) is a dysmyelinating disorder of the central nervous system caused by impaired differentiation of oligodendrocytes. This study was prompted by findings that antimuscarinic compounds enhance oligodendrocyte differentiation and remyelination in vitro. One of these compounds, clemastine fumarate, is licensed for treatment of allergic conditions. We tested whether clemastine fumarate can promote myelination in two rodent PMD models, the myelin-deficient and the PLP transgenic rat.MethodsPups were treated with daily injections of clemastine (10-30 mg/kg/day) on postnatal days 1-21. Neurologic phenotypes and myelination patterns in the brain, optic nerves, and spinal cords were assessed using histological techniques.ResultsNo changes in neurological phenotype or survival were observed even at the highest dose of clemastine. Postmortem staining with Luxol fast blue and myelin basic protein immunohistochemistry revealed no evidence for improved myelination in the CNS of treated rats compared to vehicle-treated littermates. Populations of mature oligodendrocytes were unaffected by the treatment.ConclusionThese results demonstrate lack of therapeutic effect of clemastine in two rat PMD models. Both models have rapid disease progression consistent with the connatal form of the disease. Further studies are necessary to determine whether clemastine bears a therapeutic potential in milder forms of PMD.

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Year:  2018        PMID: 29584714      PMCID: PMC6621548          DOI: 10.1038/pr.2018.45

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  29 in total

1.  Splice site selection in the proteolipid protein (PLP) gene transcript and primary structure of the DM-20 protein of central nervous system myelin.

Authors:  K A Nave; C Lai; F E Bloom; R J Milner
Journal:  Proc Natl Acad Sci U S A       Date:  1987-08       Impact factor: 11.205

Review 2.  Proteolipid proteins: structure and genetic expression in normal and myelin-deficient mutant mice.

Authors:  K A Nave; R J Milner
Journal:  Crit Rev Neurobiol       Date:  1989

3.  Mammalian target of rapamycin promotes oligodendrocyte differentiation, initiation and extent of CNS myelination.

Authors:  Stacey E Wahl; Lauren E McLane; Kathryn K Bercury; Wendy B Macklin; Teresa L Wood
Journal:  J Neurosci       Date:  2014-03-26       Impact factor: 6.167

Review 4.  The myelin mutants as models to study myelin repair in the leukodystrophies.

Authors:  Ian D Duncan; Yoichi Kondo; Su-Chun Zhang
Journal:  Neurotherapeutics       Date:  2011-10       Impact factor: 7.620

Review 5.  The proteolipid protein gene and myelin disorders in man and animal models.

Authors:  D A Yool; J M Edgar; P Montague; S Malcolm
Journal:  Hum Mol Genet       Date:  2000-04-12       Impact factor: 6.150

Review 6.  PLP1-related inherited dysmyelinating disorders: Pelizaeus-Merzbacher disease and spastic paraplegia type 2.

Authors:  Ken Inoue
Journal:  Neurogenetics       Date:  2004-12-31       Impact factor: 2.660

7.  Maturation-dependent apoptotic cell death of oligodendrocytes in myelin-deficient rats.

Authors:  J B Grinspan; M Coulalaglou; J S Beesley; D F Carpio; S S Scherer
Journal:  J Neurosci Res       Date:  1998-12-01       Impact factor: 4.164

8.  Clemastine Enhances Myelination in the Prefrontal Cortex and Rescues Behavioral Changes in Socially Isolated Mice.

Authors:  Jia Liu; Jeffrey L Dupree; Mar Gacias; Rebecca Frawley; Tamjeed Sikder; Payal Naik; Patrizia Casaccia
Journal:  J Neurosci       Date:  2016-01-20       Impact factor: 6.167

Review 9.  mTOR: a link from the extracellular milieu to transcriptional regulation of oligodendrocyte development.

Authors:  Teresa L Wood; Kathryn K Bercury; Stacey E Cifelli; Lauren E Mursch; Jungsoo Min; Jinxiang Dai; Wendy B Macklin
Journal:  ASN Neuro       Date:  2013-03-19       Impact factor: 4.146

Review 10.  Inherited and acquired disorders of myelin: The underlying myelin pathology.

Authors:  Ian D Duncan; Abigail B Radcliff
Journal:  Exp Neurol       Date:  2016-04-09       Impact factor: 5.330

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  1 in total

1.  Clemastine Induces an Impairment in Developmental Myelination.

Authors:  Ana Palma; Juan Carlos Chara; Alejandro Montilla; Amaia Otxoa-de-Amezaga; Francisca Ruíz-Jaén; Anna M Planas; Carlos Matute; Alberto Pérez-Samartín; María Domercq
Journal:  Front Cell Dev Biol       Date:  2022-03-17
  1 in total

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