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Impaired maturation and differentiation of oligodendrocytes in the vacuole formation myelin mutant rat.

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Τίτλος: Impaired maturation and differentiation of oligodendrocytes in the vacuole formation myelin mutant rat.
Συγγραφείς: Tanaka M; Osaka Metropolitan University, Izumisano, Japan.; Kyoto University, Kyoto, Japan., Morita C; Osaka Metropolitan University, Izumisano, Japan., Izawa T; Osaka Metropolitan University, Izumisano, Japan., Yamate J; Osaka Metropolitan University, Izumisano, Japan., Franklin RJM; University of Cambridge, Addenbrookes Hospital, Cambridge, UK., Kuramoto T; Kyoto University, Kyoto, Japan.; Tokyo University of Agriculture, Atsugi, Japan., Serikawa T; Kyoto University, Kyoto, Japan., Kuwamura M; Osaka Metropolitan University, Izumisano, Japan.
Πηγή: Veterinary pathology [Vet Pathol] 2026 Mar; Vol. 63 (2), pp. 315-324. Date of Electronic Publication: 2025 Oct 09.
Τύπος έκδοσης: Journal Article
Γλώσσα: English
Στοιχεία περιοδικού: Publisher: Sage Country of Publication: United States NLM ID: 0312020 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1544-2217 (Electronic) Linking ISSN: 03009858 NLM ISO Abbreviation: Vet Pathol Subsets: MEDLINE
Imprint Name(s): Publication: Jan. 2010- : Thousand Oaks, CA : Sage
Original Publication: 1971- : Basel : New York : Karger.
Ιατρικοί όροι (MeSH): Oligodendroglia*/pathology , Oligodendroglia*/metabolism , Oligodendroglia*/cytology , Myelin Sheath*/pathology , Myelin Sheath*/genetics , Myelin Sheath*/metabolism , Vacuoles*/pathology , Cell Differentiation*, Myelin Basic Protein/genetics ; Myelin Basic Protein/metabolism ; Spinal Cord/pathology ; Spinal Cord/metabolism ; Animals ; Rats ; Rats, Mutant Strains ; Male
Περίληψη: The vacuole formation (VF) rat is an autosomal recessive myelin mutant characterized by generalized tremor, hypomyelination, and periaxonal VF of the central nervous system. We previously identified a nonsense mutation in the DOP1 leucine zipper-like protein A (Dop1a, also known as Dopey1) gene located on rat chromosome 8. In this study, we investigated the kinetics of oligodendrocyte progenitor cells (OPCs) and oligodendrocytes by assessing the expression patterns of transcription factors that are involved in oligodendrocyte development. The number of small, round oligodendrocytes lacking distinct cellular processes, immunolabeled with anti-adenomatous polyposis coli (APC, clone CC1) antibody, was increased in the spinal cords of the VF rats, suggesting a disrupted maturation of oligodendrocytes. In addition, the terminal differentiation and maturation of OPCs into myelinating mature oligodendrocytes may be impaired and compensatory myelination largely could fail in the VF rat. Our findings also demonstrated that the DOP1A protein is expressed in OPCs as well as mature oligodendrocytes. Finally, the intracellular trafficking of myelin basic protein (Mbp) mRNAs may be disrupted in oligodendrocytes of the VF rats. Our data suggested that DOP1A dysfunction causes impaired differentiation and maturation of oligodendrocyte-lineage cells, resulting in hypomyelination and an impaired compensatory myelination.
Competing Interests: Declaration of Conflicting InterestsThe authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: Authors Takeshi Izawa and Mitsuru Kuwamura are members of the Editorial Board of Veterinary Pathology and have no further conflicts to declare. The authors did not take part in the peer review or decision-making process for this submission.
Contributed Indexing: Keywords: Dop1a; hypomyelination; mutant; myelin vacuole; oligodendrocyte progenitor cells; oligodendrocytes; rats
Substance Nomenclature: 0 (Myelin Basic Protein)
Entry Date(s): Date Created: 20251009 Date Completed: 20260209 Latest Revision: 20260209
Update Code: 20260209
DOI: 10.1177/03009858251379486
PMID: 41067867
Βάση Δεδομένων: MEDLINE
FullText Links:
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Header DbId: cmedm
DbLabel: MEDLINE
An: 41067867
AccessLevel: 3
PubType: Academic Journal
PubTypeId: academicJournal
PreciseRelevancyScore: 0
IllustrationInfo
Items – Name: Title
  Label: Title
  Group: Ti
  Data: Impaired maturation and differentiation of oligodendrocytes in the vacuole formation myelin mutant rat.
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  Label: Authors
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  Data: <searchLink fieldCode="AU" term="%22Tanaka+M%22">Tanaka M</searchLink>; Osaka Metropolitan University, Izumisano, Japan.; Kyoto University, Kyoto, Japan.<br /><searchLink fieldCode="AU" term="%22Morita+C%22">Morita C</searchLink>; Osaka Metropolitan University, Izumisano, Japan.<br /><searchLink fieldCode="AU" term="%22Izawa+T%22">Izawa T</searchLink>; Osaka Metropolitan University, Izumisano, Japan.<br /><searchLink fieldCode="AU" term="%22Yamate+J%22">Yamate J</searchLink>; Osaka Metropolitan University, Izumisano, Japan.<br /><searchLink fieldCode="AU" term="%22Franklin+RJM%22">Franklin RJM</searchLink>; University of Cambridge, Addenbrookes Hospital, Cambridge, UK.<br /><searchLink fieldCode="AU" term="%22Kuramoto+T%22">Kuramoto T</searchLink>; Kyoto University, Kyoto, Japan.; Tokyo University of Agriculture, Atsugi, Japan.<br /><searchLink fieldCode="AU" term="%22Serikawa+T%22">Serikawa T</searchLink>; Kyoto University, Kyoto, Japan.<br /><searchLink fieldCode="AU" term="%22Kuwamura+M%22">Kuwamura M</searchLink>; Osaka Metropolitan University, Izumisano, Japan.
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  Data: <searchLink fieldCode="JN" term="%220312020%22">Veterinary pathology</searchLink> [Vet Pathol] 2026 Mar; Vol. 63 (2), pp. 315-324. <i>Date of Electronic Publication: </i>2025 Oct 09.
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  Data: Journal Article
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  Data: English
– Name: TitleSource
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  Data: <i>Publisher: </i><searchLink fieldCode="PB" term="%22Sage%22">Sage </searchLink><i>Country of Publication: </i>United States <i>NLM ID: </i>0312020 <i>Publication Model: </i>Print-Electronic <i>Cited Medium: </i>Internet <i>ISSN: </i>1544-2217 (Electronic) <i>Linking ISSN: </i><searchLink fieldCode="IS" term="%2203009858%22">03009858 </searchLink><i>NLM ISO Abbreviation: </i>Vet Pathol <i>Subsets: </i>MEDLINE
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  Data: <i>Publication</i>: Jan. 2010- : Thousand Oaks, CA : Sage<br /><i>Original Publication</i>: 1971- : Basel : New York : Karger.
– Name: SubjectMESH
  Label: MeSH Terms
  Group: Su
  Data: <searchLink fieldCode="MM" term="%22Oligodendroglia%22">Oligodendroglia*</searchLink>/<searchLink fieldCode="MM" term="%22Oligodendroglia+pathology%22">pathology</searchLink> <br /><searchLink fieldCode="MM" term="%22Oligodendroglia%22">Oligodendroglia*</searchLink>/<searchLink fieldCode="MM" term="%22Oligodendroglia+metabolism%22">metabolism</searchLink> <br /><searchLink fieldCode="MM" term="%22Oligodendroglia%22">Oligodendroglia*</searchLink>/<searchLink fieldCode="MM" term="%22Oligodendroglia+cytology%22">cytology</searchLink> <br /><searchLink fieldCode="MM" term="%22Myelin+Sheath%22">Myelin Sheath*</searchLink>/<searchLink fieldCode="MM" term="%22Myelin+Sheath+pathology%22">pathology</searchLink> <br /><searchLink fieldCode="MM" term="%22Myelin+Sheath%22">Myelin Sheath*</searchLink>/<searchLink fieldCode="MM" term="%22Myelin+Sheath+genetics%22">genetics</searchLink> <br /><searchLink fieldCode="MM" term="%22Myelin+Sheath%22">Myelin Sheath*</searchLink>/<searchLink fieldCode="MM" term="%22Myelin+Sheath+metabolism%22">metabolism</searchLink> <br /><searchLink fieldCode="MM" term="%22Vacuoles%22">Vacuoles*</searchLink>/<searchLink fieldCode="MM" term="%22Vacuoles+pathology%22">pathology</searchLink> <br /><searchLink fieldCode="MM" term="%22Cell+Differentiation%22">Cell Differentiation*</searchLink><br /><searchLink fieldCode="MH" term="%22Myelin+Basic+Protein%22">Myelin Basic Protein</searchLink>/<searchLink fieldCode="MH" term="%22Myelin+Basic+Protein+genetics%22">genetics</searchLink> ; <searchLink fieldCode="MH" term="%22Myelin+Basic+Protein%22">Myelin Basic Protein</searchLink>/<searchLink fieldCode="MH" term="%22Myelin+Basic+Protein+metabolism%22">metabolism</searchLink> ; <searchLink fieldCode="MH" term="%22Spinal+Cord%22">Spinal Cord</searchLink>/<searchLink fieldCode="MH" term="%22Spinal+Cord+pathology%22">pathology</searchLink> ; <searchLink fieldCode="MH" term="%22Spinal+Cord%22">Spinal Cord</searchLink>/<searchLink fieldCode="MH" term="%22Spinal+Cord+metabolism%22">metabolism</searchLink> ; <searchLink fieldCode="MH" term="%22Animals%22">Animals</searchLink> ; <searchLink fieldCode="MH" term="%22Rats%22">Rats</searchLink> ; <searchLink fieldCode="MH" term="%22Rats%2C+Mutant+Strains%22">Rats, Mutant Strains</searchLink> ; <searchLink fieldCode="MH" term="%22Male%22">Male</searchLink>
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: The vacuole formation (VF) rat is an autosomal recessive myelin mutant characterized by generalized tremor, hypomyelination, and periaxonal VF of the central nervous system. We previously identified a nonsense mutation in the DOP1 leucine zipper-like protein A (Dop1a, also known as Dopey1) gene located on rat chromosome 8. In this study, we investigated the kinetics of oligodendrocyte progenitor cells (OPCs) and oligodendrocytes by assessing the expression patterns of transcription factors that are involved in oligodendrocyte development. The number of small, round oligodendrocytes lacking distinct cellular processes, immunolabeled with anti-adenomatous polyposis coli (APC, clone CC1) antibody, was increased in the spinal cords of the VF rats, suggesting a disrupted maturation of oligodendrocytes. In addition, the terminal differentiation and maturation of OPCs into myelinating mature oligodendrocytes may be impaired and compensatory myelination largely could fail in the VF rat. Our findings also demonstrated that the DOP1A protein is expressed in OPCs as well as mature oligodendrocytes. Finally, the intracellular trafficking of myelin basic protein (Mbp) mRNAs may be disrupted in oligodendrocytes of the VF rats. Our data suggested that DOP1A dysfunction causes impaired differentiation and maturation of oligodendrocyte-lineage cells, resulting in hypomyelination and an impaired compensatory myelination.
– Name: Abstract
  Label: Competing Interests
  Group: Ab
  Data: Declaration of Conflicting InterestsThe authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: Authors Takeshi Izawa and Mitsuru Kuwamura are members of the Editorial Board of Veterinary Pathology and have no further conflicts to declare. The authors did not take part in the peer review or decision-making process for this submission.
– Name: SubjectMinor
  Label: Contributed Indexing
  Group:
  Data: <i>Keywords: </i>Dop1a; hypomyelination; mutant; myelin vacuole; oligodendrocyte progenitor cells; oligodendrocytes; rats
– Name: NumberCAS
  Label: Substance Nomenclature
  Group: ID
  Data: 0 (Myelin Basic Protein)
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  Data: <i>Date Created: </i>20251009 <i>Date Completed: </i>20260209 <i>Latest Revision: </i>20260209
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  Data: 10.1177/03009858251379486
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  Data: 41067867
PLink https://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=cmedm&AN=41067867
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        Value: 10.1177/03009858251379486
    Languages:
      – Code: eng
        Text: English
    PhysicalDescription:
      Pagination:
        StartPage: 315
    Subjects:
      – SubjectFull: Myelin Basic Protein genetics
        Type: general
      – SubjectFull: Myelin Basic Protein metabolism
        Type: general
      – SubjectFull: Spinal Cord pathology
        Type: general
      – SubjectFull: Spinal Cord metabolism
        Type: general
      – SubjectFull: Animals
        Type: general
      – SubjectFull: Rats
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      – SubjectFull: Rats, Mutant Strains
        Type: general
      – SubjectFull: Male
        Type: general
      – SubjectFull: Oligodendroglia pathology
        Type: general
      – SubjectFull: Oligodendroglia metabolism
        Type: general
      – SubjectFull: Oligodendroglia cytology
        Type: general
      – SubjectFull: Myelin Sheath pathology
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      – SubjectFull: Myelin Sheath genetics
        Type: general
      – SubjectFull: Myelin Sheath metabolism
        Type: general
      – SubjectFull: Vacuoles pathology
        Type: general
      – SubjectFull: Cell Differentiation
        Type: general
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      – TitleFull: Impaired maturation and differentiation of oligodendrocytes in the vacuole formation myelin mutant rat.
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              Text: 2026 Mar
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              Y: 2026
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