Evidence map›Paper›PMID 42644218›Full record

ArticleAnnals of clinical and translational neurology2026

Nationwide Survey of Association Between Fever and Epileptic Seizure in CDKL5 Deficiency Disorder Revealed Therapeutic Implications.

Siyi Wang, Jialin Du, Jinghui Liu, Dongmei Hu, Hongxin Wang, Xiaoxiao Man, Lehong Gao, Shimin Hu, Xianghong Meng, Hongyang Zhao and 4 more

Registry-linked trialAbstract read
In one paragraph

Article in Annals of clinical and translational neurology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06663163 (The Association Between Fever and CDKL5 Deficiency Disorder), which is not on this map. Not yet cited 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

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.

NCT06663163 completednot on this map

The Association Between Fever and CDKL5 Deficiency Disorder: a Nationwide Survey

TypeobservationalSponsorXuanwu Hospital, BeijingRan2024 to 2024Enrolled131ConditionsCDKL5 Deficiency DisorderArmsObservation
3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

14 authors.

Siyi WangDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Jialin DuDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Jinghui LiuDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Dongmei HuDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.ORCID https://orcid.org/0009-0007-6798-9254
Hongxin WangJinan Hospital of Xuanwu Hospital, Shandong First Medical University, Jinan, China.
Xiaoxiao ManJinan Hospital of Xuanwu Hospital, Shandong First Medical University, Jinan, China.
Lehong GaoDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Shimin HuDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Xianghong MengDepartment of Neurosurgery, Shenzhen University General Hospital, Shenzhen, China.
Hongyang ZhaoDepartment of Pediatrics, Central Hospital Affiliated to Shandong First Medical University, Jinan, China.
Minjing HuDepartment of Neurology, Affiliated Hospital of Nantong University, Nantong, China.
Yingxue YangDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.
Zhiqi XiongState Key Laboratory of Neuroscience, Key Laboratory of Primate Neurobiology, Institute of Neuroscience, Chinese Academy of Sciences Center for Excellence in Brain Science and Intelligence Technology, Shanghai, China.ORCID https://orcid.org/0000-0001-7727-8412
Liankun RenDepartment of Neurology, Xuanwu Hospital, Capital Medical University, Beijing, China.ORCID https://orcid.org/0000-0001-5147-3068

Funding

Beijing Municipal Administration of Hospitals ZLRK202321National Key Research and Development Program of China 2022YFC2503805National Key Research and Development Program of China 2024YFA1306903National Natural Science Foundation of China 82371457
6 · The paper itself

Abstract

objectiveCDKL5 deficiency disorder (CDD) is a rare, severe developmental and epileptic encephalopathy. There is a pressing need to develop effective and sustainable therapeutic strategies. We aimed to investigate the causal association between febrile episodes and epileptic seizures for therapeutic implications in CDD patients.

methodsThe study was a nationwide, cross-sectional survey on CDD patients in China (ClinicalTrials.gov, NCT06663163). Detailed phenotypic and genotypic data were collected through an online questionnaire with uploaded original medical records, genetic testing results, and peri-fever seizure diaries. The primary outcome was changes in epileptic seizure frequency during and post-fever phases compared to a 1-month pre-fever phase based on seizure diaries.

resultsBetween October 2024 and December 2024, we received 131 questionnaires. Forty-seven questionnaires were removed after excluding duplicates and missing data. Ultimately, 84 eligible participants with complete uploads were included, from 26 of 34 (76.5%) province-level regions in China. Among these, 47 (56.0%) patients had significant decreased seizure frequency only during febrile episodes, and 27 (32.1%) patients with daily seizures achieved at least a seizure-free day. Notably, 20 patients (23.8%) exhibited post-fever seizure reduction: 12 (25.5%) for 3 days to 1 week, and 6 (12.8%) for more than 2 weeks (maximum > 40 days). The effect was independent of patients' clinical and genetic characteristics.

interpretationOur findings, for the first time, revealed that fever-related seizure reduction is a distinctive and prevalent genotype-phenotype for CDD, thereby providing evidence for further fundamental research into the underlying mechanisms and offering potential clinical implications for seizure control in CDD.

Indexed as

CDKL5 deficiency disorderepilepsyfevergenotype–phenotype

Identifiers

PMID42644218
PMCPMC13507912

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