Evidence map›Paper›PMID 33407677›Full record

ArticleOrphanet journal of rare diseases2021

Acute encephalopathy in children with tuberous sclerosis complex.

Shingo Numoto, Hirokazu Kurahashi, Atsushi Sato, Masaya Kubota, Takashi Shiihara, Tohru Okanishi, Ryuta Tanaka, Ichiro Kuki, Tetsuhiro Fukuyama, Mitsuru Kashiwagi and 5 more

Open access · goldAbstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2021. 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
0.4field-weighted citation impact, top 36% 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, 5 citations in OpenAlex.

  1. Article
  2. Review
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

15 authors at 13 institutions in 1 country.

Shingo NumotoDepartment of Pediatrics, Aichi Medical University, 1-1 Yazako Karimata, Nagakute, Aichi, 480-1195, Japan. numoto.shingo.068@mail.aichi-med-u.ac.jp.ORCID 0000-0001-8135-772X
Hirokazu KurahashiDepartment of Pediatrics, Aichi Medical University, 1-1 Yazako Karimata, Nagakute, Aichi, 480-1195, Japan.
Atsushi SatoDepartment of Pediatrics, The University of Tokyo Hospital, Tokyo, Japan.
Masaya KubotaDivision of Neurology, National Center for Child Health and Development, Tokyo, Japan.
Takashi ShiiharaDepartment of Neurology, Gunma Children's Medical Center, Shibukawa, Gunma, Japan.
Tohru OkanishiDepartment of Child Neurology, Seirei Hamamatsu General Hospital, Hamamatsu, Japan.
Ryuta TanakaDepartment of Child Health, Ibaraki Pediatric Education and Training Station, University of Tsukuba, Mito, Japan.
Ichiro KukiDepartment of Pediatric Neurology, Osaka City General Hospital, Osaka, Japan.
Tetsuhiro FukuyamaDivision of Neurology, Nagano Children's Hospital, Nagano, Japan.
Mitsuru KashiwagiDepartment of Pediatrics, Hirakata City Hospital, Osaka, Japan.
Mitsuru IkenoDepartment of Pediatrics, Faculty of Medicine, Juntendo University, Tokyo, Japan.
Kazuo KubotaDepartment of Pediatrics, Gifu University Graduate School of Medicine, Gifu, Japan.
Manami AkasakaDepartment of Pediatrics, School of Medicine, Iwate Medical University, Morioka, Japan.
Masakazu MimakiDepartment of Pediatrics, Teikyo University School of Medicine, Tokyo, Japan.
Akihisa OkumuraDepartment of Pediatrics, Aichi Medical University, 1-1 Yazako Karimata, Nagakute, Aichi, 480-1195, Japan.ORCID 0000-0003-2447-2857
Aichi Medical University · JPGifu University · JPGunma Children's Medical Center · JPIwate Medical University · JPJuntendo University · JPMunicipal Hirakata City Hospital · JPNagano Children's Hospital · JPNational Center For Child Health and Development · JPOsaka City General Hospital · JPSeirei Hamamatsu General Hospital · JPTeikyo University · JPUniversity of Tokyo Hospital · JPUniversity of Tsukuba · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveWe examined the clinical manifestations of acute encephalopathy (AE) and identify risk factors for AE in children with tuberous sclerosis complex (TSC).

methodsThe clinical data of 11 children with clinically diagnosed TSC associated with AE and 109 children with clinically diagnosed TSC alone aged 4 years or older were collected from 13 hospitals.

resultsOf the 11 children with AE, 5 had histories of febrile seizures (FS), and all had histories of febrile status epilepticus (FSE). AE developed within 24 h after fever onset in all children with seizures lasting 30 min or longer. All children developed coma after seizure cessation. Head magnetic resonance imaging (MRI) revealed widespread abnormalities in the cerebral cortex, subcortical white matter, corpus callosum, basal ganglia, and thalamus. One child died; seven had severe neurological sequelae; and the other three, mild sequelae. Logistic regression analysis revealed that a history of FSE was correlated with the development of AE. SIGNIFICANCE: AE in children with TSC was characterized by sudden onset after fever, followed by coma, widespread brain edema evident on MRI, and poor outcomes. A history of FSE was a risk factor for the development of AE.

Indexed as

Brain DiseasesSeizures, FebrileStatus EpilepticusTuberous SclerosisChildHumansInfantMagnetic Resonance ImagingSeizuresClinical neurology historyInfantile spasmsMRIPrognosisStatus epilepticus

Identifiers

PMID33407677
PMCPMC7789140
OpenAlexW3098359599

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Registered trials

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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.