Evidence map›Paper›PMID 30244407›Full record

ArticleJournal of applied genetics2018

Identification novel LQT syndrome-associated variants in Polish population and genotype-phenotype correlations in eight families.

Małgorzata Szperl, Urszula Kozicka, Agnieszka Kosiec, Piotr Kukla, Marta Roszczynko, Elżbieta Katarzyna Biernacka

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In one paragraph

Article in Journal of applied genetics, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact, top 80% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 3 citations in OpenAlex.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors at 1 institution in 1 country.

Małgorzata SzperlDepartment of Epidemiology, Cardiovascular Disease Prevention and Health Promotion, Institute of Cardiology, Ul. Niemodlińska 33, 40-635, Warsaw, Poland. mszperl@yahoo.com.
Urszula KozickaDepartment of Congenital Cardiac Defects, Institute of Cardiology, Warsaw, Poland.
Agnieszka KosiecDepartment of Molecular Biology, Institute of Cardiology, Warsaw, Poland.
Piotr KuklaDepartment of Cardiology and Internal Medicine, Specialistic Hospital, Gorlice, Poland.
Marta RoszczynkoDepartment of Molecular Biology, Institute of Cardiology, Warsaw, Poland.
Elżbieta Katarzyna BiernackaDepartment of Congenital Cardiac Defects, Institute of Cardiology, Warsaw, Poland.
Institute of Cardiology · PL

Funding

Institute of Cardiology, Warsaw, Poland funds no. 2.62/II/12
6 · The paper itself

Abstract

Congenital long QT syndrome (LQTS) is a primary cardiac channelopathy. Genetic testing has not only diagnostic but also prognostic and therapeutic implications. At present, 15 genes have been associated with the disease, with most mutations located in 3 major LQTS-susceptibility genes. During a routine genetic screening for KCNQ1, KCNH2 and SCN5A genes in index cases with LQTS, seven novel variants in KCNH2 and SCN5A genes were found. Genotype-phenotype correlations were analysed in these patients and their families. An open reading frame and splice site analysis of the exons was conducted using next-generation sequencing. In novel variants, phenotypes of carriers and their affected relatives were analysed. In 39 unrelated patients, 40 pathogenic/putative pathogenic mutations were found. Thirty-three of them, predominantly missense, were reported previously: 11 were in the KCNQ, 17 in the KCNH2 and 5 in the SCN5A gene. Seven novel missense variants were found in eight families. Among them, four variants were in typical for LQTS location. Two variants in the KCNH2 gene (p.D803Y and p.D46F) and one in the SCN5A gene (G1391R) were in amino acid (AA) position which up to present has not been reported in LQTS. Phenotype analysis showed the life-threatening course of the disease in index cases with a history of sudden cardiac death in six families. Mutation carriers presented with ECG abnormalities and some of them received beta-blocker therapy. We report three novel variants (KCNQ1 p.46, KCNH2 p.D803Y, SCN5A p.G1391R) which have never been reported for this AA location in LQTS; the phenotype-genotype correlation suggests their pathogenicity.

Indexed as

Genetic Association StudiesAdultDNA Mutational AnalysisERG1 Potassium ChannelFemaleGenetic TestingHeterozygoteHumansKCNQ1 Potassium ChannelLong QT SyndromeMaleMutation, MissenseNAV1.5 Voltage-Gated Sodium ChannelPedigreePhenotypePolandERG1 Potassium ChannelKCNH2 protein, humanKCNQ1 Potassium ChannelKCNQ1 protein, humanNAV1.5 Voltage-Gated Sodium ChannelSCN5A protein, humanGenetic screeningLong QT syndromeNovel variants

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.