Evidence map›Paper›PMID 37762919›Full record

ArticleJournal of clinical medicine2023

Clinicopathologic Features, Genetics, Treatment, and Long-Term Outcomes in Japanese Children and Young Adults with Benign Recurrent Intrahepatic Cholestasis: A Multicenter Study.

Ken Kato, Shuichiro Umetsu, Takao Togawa, Koichi Ito, Takayoshi Kawabata, Teruko Arinaga-Hino, Naoya Tsumura, Ryosuke Yasuda, Yutaro Mihara, Hironori Kusano and 6 more

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Journal of clinical medicine, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
1.6field-weighted citation impact, top 14% 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

3 citing papers in PubMed, 7 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
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

16 authors at 5 institutions in 1 country.

Ken KatoDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.
Shuichiro UmetsuDepartment of Pediatric Hepatology and Gastroenterology, Saiseikai Yokohamashi Tobu Hospital, Yokohama 230-0012, Japan.
Takao TogawaDepartment of Pediatrics and Neonatology, Nagoya City University Graduate School of Medical Sciences, Nagoya 467-8601, Japan.
Koichi ItoDepartment of Pediatrics and Neonatology, Nagoya City University Graduate School of Medical Sciences, Nagoya 467-8601, Japan.
Takayoshi KawabataDepartment of Pediatrics, Ishikawa Prefectural Central Hospital, Kanazawa 920-8530, Japan.
Teruko Arinaga-HinoDivision of Gastroenterology, Department of Medicine, Kurume University School of Medicine, Kurume 830-0011, Japan.
Naoya TsumuraDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.
Ryosuke YasudaDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.
Yutaro MiharaDepartment of Pathology, Kurume University School of Medicine, Kurume 830-0011, Japan.ORCID 0000-0002-4119-1486
Hironori KusanoDepartment of Pathology, Kurume University School of Medicine, Kurume 830-0011, Japan.ORCID 0000-0002-9911-2007
Shogo ItoDepartment of Pediatrics and Neonatology, Nagoya City University Graduate School of Medical Sciences, Nagoya 467-8601, Japan.
Kazuo ImagawaDepartment of Child Health, Institute of Medicine, University of Tsukuba, Tsukuba 305-8546, Japan.
Hisamitsu HayashiLaboratory of Molecular Pharmacokinetics, Graduate School of Pharmaceutical Science, The University of Tokyo, Tokyo 113-0033, Japan.ORCID 0000-0002-5247-895X
Ayano InuiDepartment of Pediatric Hepatology and Gastroenterology, Saiseikai Yokohamashi Tobu Hospital, Yokohama 230-0012, Japan.
Yushiro YamashitaDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.
Tatsuki MizuochiDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume 830-0011, Japan.ORCID 0000-0002-2943-7068
Kurume University · JPNagoya City University · JPIshikawa Prefectural Central Hospital · JPThe University of Tokyo · JPUniversity of Tsukuba · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundFew reports of benign recurrent intrahepatic cholestasis (BRIC) have focused on East Asian patients. We describe the clinicopathologic features, genetics, treatment, and outcomes in Japanese BRIC patients.

methodsWe recruited patients with BRIC type 1 (BRIC-1) or 2 (BRIC-2) treated at four pediatric centers and one adult center between April 2007 and March 2022. Demographics, clinical course, laboratory results, molecular genetic findings concerning

resultsSeven Japanese patients with BRIC were enrolled (four male, three female; four BRIC-1 and three BRIC-2). The median age at onset for BRIC-1 was 12 years; for BRIC-2, it was 1 month. Intermittent cholestatic attacks numbered from one to eight during the 11 years of median follow-up. Six patients received a mainstream education; only one patient attended special education. None developed cirrhosis. Three with BRIC-1 showed compound heterozygosity for a variant

conclusionsTo our knowledge, this is the first East Asian multicenter study of BRIC patients. Onset age and number of cholestatic attacks varied. Rifampicin and cholestyramine were effective against attacks. No patient developed cirrhosis; most had normal growth and development. The long-term outcomes were satisfactory.

Indexed as

ABCB11ATP8B1childrencholestasischolestyraminerifampicin

Identifiers

PMID37762919
PMCPMC10532077
OpenAlexW4386778969

What OpenQuestion holds

Textmetadata
LicenceCC BY
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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.