Title |
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Congenital atrial standstill associated with coinheritance of a novel SCN5A mutation and connexin 40 polymorphisms
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Creator |
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Groenewegen, W. Antoinette
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Accessrights |
open access |
Subject |
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Other
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Atrial standstill
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Other
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SCN5A
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Other
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Connexin 40
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Other
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Incomplete penetrance
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Other
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Single nucleotide polymorphism
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Other
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Sudden death
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NDC
493.2
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Description |
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Abstract
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The novel SCN5A mutation L212P was identified in the proband (age 11 years) and his father. The father was in normal sinus rhythm. The proband had no P waves on surface ECG, and his right atrium could not be captured by pacing. The recombinant L212P Na channel showed a large hyperpolarizing shift in both the voltage dependence of activation (WT: −48.1 ± 0.9 mV; L212P: −63.5 ± 1.5 mV; P < .001) and inactivation (WT: −86.6 ± 0.9 mV; L212P: −95.6 ± 0.8 mV; P < .001) and delayed recovery from inactivation. Further screenings for genetic variations that might mitigate L212P dysfunction revealed that the proband, but not his father, carries Cx40 polymorphisms inherited from his asymptomatic mother.
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Publisher |
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Heart Rhythm Society Published by Elsevier Inc.
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Date |
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Language |
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Resource Type |
journal article |
Version Type |
AM |
Identifier |
HDL
http://hdl.handle.net/2115/16956
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Relation |
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URI
http://www.sciencedirect.com/science/journal/15475271
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isVersionOf
DOI
https://doi.org/10.1016/j.hrthm.2005.06.032
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PMID
16188595
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Journal |
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Heart Rhythm
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Volume Number2
Issue Number10
Page Start1128
Page End1134
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File |
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Oaidate |
2023-07-26 |