Evidence map›Paper›PMID 40091034›Full record

ReviewBMC pediatrics2025

Congenital long QT syndrome caused by a KCNH2 pathogenic variant exhibiting "motor seizures": a case report and literature review.

Mei Jin, Fan Yang, Yakun Du, Libo Zhao, Xueran Zhao, Jing Liu, Jing Zhang, Suzhen Sun

Abstract readCase ReportsReview
In one paragraph

Review in BMC pediatrics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0citing papers in PubMed
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1 · What the graph read from it

What it found

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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

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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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

8 authors.

Mei JinDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Fan YangDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Yakun DuDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Libo ZhaoDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Xueran ZhaoDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Jing LiuDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Jing ZhangDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China.
Suzhen SunDepartment of Pediatric Neurology, The Children Hospital of Hebei Province, Shijiazhuang, Hebei, 050000, China. sunsuzhen2004@126.com.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A retrospective analysis was conducted to evaluate the clinical characteristics, diagnostic challenges, and management strategies in a child with congenital long QT syndrome (cLQTS) caused by a KCNH2 gene pathogenic variant presenting as "motor seizures". The case involved a 10-year-old boy with a two-year history of recurrent loss of consciousness, which had worsened during the preceding week. Clinical manifestations included sudden episodes of unconsciousness, rightward strabismus of both eyes, cyanosis of the lips, guttural vocalizations, rigidity and shaking of the upper limbs, and urinary incontinence. These events typically lasted approximately two minutes, initially occurring semiannually but escalating to daily episodes over the past week, affecting both awake and sleep states. Video electroencephalography (VEEG) showed generalized slow waves and low voltage activity, while electrocardiography (ECG) demonstrated QTc prolongation, paired, and multi-source ventricular ectopy preceding torsades de pointes. Genetic testing identified a pathogenic c.1697G > A mutation in the KCNH2 gene corroborating the clinical diagnosis of cLQTS. Following confirmation, the patient was initiated on long-term oral therapy with propranolol and nicorandil. Under this regimen, the patient was seizure-free for 7-month. For patients with seizures or seizure-like episodes, such as extremity movement or rigidity, it is necessary to perform an ECG examination. Additionally, dynamic ECG and electrolyte assessments should be conducted when necessary to minimize the risk of misdiagnosis and inappropriate treatment. When VEEG shows a "slow-flat-slow" pattern, differentiation from A-S syndrome caused by malignant arrhythmias is critical. Once cLQTS is diagnosed, it is imperative to initiate prompt and aggressive treatment to mitigate the risks of syncope and sudden cardiac death.

Indexed as

ERG1 Potassium ChannelLong QT SyndromeSeizuresChildElectrocardiographyElectroencephalographyHumansMaleMutationERG1 Potassium ChannelKCNH2 protein, humanChildCongenital long QT syndromeKCNH2 geneSeizuresTorsades de pointes

Identifiers

PMID40091034
PMCPMC11912787

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