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Germline variant of CTC1 gene in a patient with pulmonary fibrosis and myelodysplastic syndrome

The result's identifiers

  • Result code in IS VaVaI

    <a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F65269705%3A_____%2F23%3A00078121" target="_blank" >RIV/65269705:_____/23:00078121 - isvavai.cz</a>

  • Alternative codes found

    RIV/00216224:14110/23:00131222

  • Result on the web

    <a href="https://mrmjournal.org/mrm/article/view/909" target="_blank" >https://mrmjournal.org/mrm/article/view/909</a>

  • DOI - Digital Object Identifier

    <a href="http://dx.doi.org/10.4081/mrm.2023.909" target="_blank" >10.4081/mrm.2023.909</a>

Alternative languages

  • Result language

    angličtina

  • Original language name

    Germline variant of CTC1 gene in a patient with pulmonary fibrosis and myelodysplastic syndrome

  • Original language description

    Introduction: Telomeropathies are associated with a wide range of diseases and less common combinations of various pulmonary and extrapulmonary disorders.Case presentation: In proband with high-risk myelodysplastic syndrome and interstitial pulmonary fibrosis, whole exome sequencing revealed a germline heterozygous variant of CTC1 gene (c.1360delG). This &quot;frameshift&quot; variant results in a premature stop codon and is classified as likely pathogenic/pathogenic. So far, this gene variant has been described in a heterozygous state in adult patients with hematological diseases such as idiopathic aplastic anemia or paroxysmal nocturnal hemoglobinuria, but also in interstitial pulmonary fibrosis. Described CTC1 gene variant affects telomere length and leads to telomeropathies.Conclusions: In our case report, we describe a rare case of coincidence of pulmonary fibrosis and hematological malignancy caused by a germline gene mutation in CTC1. Lung diseases and hematologic malignancies associated with short telomeres do not respond well to standard treatment.

  • 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

    30203 - Respiratory systems

Result continuities

  • Project

  • Continuities

    I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Others

  • Publication year

    2023

  • Confidentiality

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

Data specific for result type

  • Name of the periodical

    Multidisciplinary Respiratory Medicine

  • ISSN

    1828-695X

  • e-ISSN

    2049-6958

  • Volume of the periodical

    18

  • Issue of the periodical within the volume

    JUN 2023

  • Country of publishing house

    IT - ITALY

  • Number of pages

    5

  • Pages from-to

    909

  • UT code for WoS article

    001009940900001

  • EID of the result in the Scopus database

    2-s2.0-85166355988