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Mitochondrial membrane protein-associated neurodegeneration: a case report and literature review

The result's identifiers

  • Result code in IS VaVaI

    <a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00216208%3A11110%2F18%3A10380572" target="_blank" >RIV/00216208:11110/18:10380572 - isvavai.cz</a>

  • Alternative codes found

    RIV/61989592:15120/18:73590552 RIV/00064165:_____/18:10380572

  • Result on the web

    <a href="http://dx.doi.org/10.1080/13554794.2018.1506038" target="_blank" >http://dx.doi.org/10.1080/13554794.2018.1506038</a>

  • DOI - Digital Object Identifier

    <a href="http://dx.doi.org/10.1080/13554794.2018.1506038" target="_blank" >10.1080/13554794.2018.1506038</a>

Alternative languages

  • Result language

    angličtina

  • Original language name

    Mitochondrial membrane protein-associated neurodegeneration: a case report and literature review

  • Original language description

    Mitochondrial membrane protein-associated neurodegeneration (MPAN) is an autosomal recessive disorder caused by mutation in the C19orf12 gene. We report a compound heterozygous c.[32C&gt;T];[205G&gt;A;424A&gt;G] (p.[Thr11Met];[Gly69Arg;Lys142Glu]) Czech patient who manifested with right foot dystonia, impaired handwriting, attention deficit, and signs of iron accumulation on brain MRI. Gradually, he developed dysarthria, spastic-dystonic gait, pedes cavi, and atrophy of leg muscles. Additionally, we report demographic parameters, clinical signs, and allelic frequencies of C19orf12 mutations of all published MPAN cases. We compared the most frequent mutations, p.Thr11Met and p.Gly69ArgfsX10; the latter was associated with younger age at onset and more frequent optic atrophy in homozygotes

  • Czech name

  • Czech description

Classification

  • Type

    J<sub>imp</sub> - Article in a specialist periodical, which is included in the Web of Science database

  • CEP classification

  • OECD FORD branch

    30103 - Neurosciences (including psychophysiology)

Result continuities

  • Project

    <a href="/en/project/NV15-25602A" target="_blank" >NV15-25602A: Biomarkers of progression and treatment response in neurodegenerative disorders</a><br>

  • Continuities

    I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Others

  • Publication year

    2018

  • Confidentiality

    S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Data specific for result type

  • Name of the periodical

    Neurocase

  • ISSN

    1355-4794

  • e-ISSN

  • Volume of the periodical

    24

  • Issue of the periodical within the volume

    3

  • Country of publishing house

    GB - UNITED KINGDOM

  • Number of pages

    5

  • Pages from-to

    161-165

  • UT code for WoS article

    000445876300006

  • EID of the result in the Scopus database

    2-s2.0-85052057469