A Novel Bradycardia-Associated Variant in <i>HCN4</i> as a Candidate Modifier in Type 3 Long QT Syndrome: Case Report and Deep In Silico Analysis

<b>Background:</b> Genetic testing for long QT syndrome (LQTS) is straightforward in many families; however, in severe and complex cases, a single disease-causing variant may not be enough to explain all clinical features. In such cases, the search for genetic modifiers may be beneficial...

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Main Authors: Anna A. Bukaeva, Anastasia V. Blokhina, Maria S. Kharlap, Marija Zaicenoka, Evgenia D. Zotova, Anna V. Petukhova, Elizaveta V. Garbuzova, Anastasia A. Zharikova, Mikhail G. Divashuk, Anna V. Kiseleva, Alexandra I. Ershova, Alexey N. Meshkov, Oxana M. Drapkina
Format: Article
Language:English
Published: MDPI AG 2025-04-01
Series:Biomedicines
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Online Access:https://www.mdpi.com/2227-9059/13/4/1008
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Summary:<b>Background:</b> Genetic testing for long QT syndrome (LQTS) is straightforward in many families; however, in severe and complex cases, a single disease-causing variant may not be enough to explain all clinical features. In such cases, the search for genetic modifiers may be beneficial for precise diagnosis and management. <b>Case presentation:</b> We describe a three-generational family affected with clinically heterogeneous LQTS type 3 and bradycardia in which a novel missense variant p.V642M in <i>HCN4</i> was identified in addition to the known pathogenic variant p.E1784K in <i>SCN5A</i>. We performed the detailed clinical investigation of the family and a deep in silico analysis of the discovered variants, showing the causal role of a new <i>HCN4</i> variant in sinus bradycardia and its possible contribution to the phenotypic heterogeneity of LQTS type 3. <b>Conclusions:</b> This case is the first description of a functional variant in <i>HCN4</i> as a candidate modifier in LQTS type 3 and demonstrates the importance of analyzing additional genetic variations in families with complex LQTS phenotypes.
ISSN:2227-9059