Evidence map›Paper›PMID 41448627›Full record

ArticleIntestinal research2026

Orofacial granulomatosis in pediatric Crohn's disease: clinical outcomes and genetic background in the era of biologics: a retrospective study in Japan.

Ryusuke Nambu, Takeo Naito, Mei Haruyama, Junichi Hosokawa, Hirotaka Shimizu, Ichiro Takeuchi, Shin-Ichiro Hagiwara, Tatsuki Mizuochi, Yugo Takaki, Takashi Ishige and 9 more

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In one paragraph

Article in Intestinal research, 2026. 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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0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

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

19 authors.

Ryusuke Nambu *Center for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology and Hepatology, Saitama Children's Medical Center, Saitama, Japan. nambee1231@gmail.com.
Takeo Naito *Division of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan.
Mei HaruyamaCenter for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology and Hepatology, Saitama Children's Medical Center, Saitama, Japan.
Junichi HosokawaKazusa DNA Research Institute, Kisarazu, Japan.
Hirotaka ShimizuCenter for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology, National Center for Child Health and Development, Tokyo, Japan.
Ichiro TakeuchiCenter for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology, National Center for Child Health and Development, Tokyo, Japan.
Shin-Ichiro HagiwaraDepartment of Gastroenterology, Nutrition and Endocrinology, Osaka Women's and Children's Hospital, Izumi, Japan.
Tatsuki MizuochiDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Japan.
Yugo TakakiDepartment of Pediatric Gastroenterology and Hepatology, Japanese Red Cross Kumamoto Hospital, Kumamoto, Japan.
Takashi IshigeDepartment of Pediatrics, Gunma University Graduate School of Medicine, Maebashi, Japan.
Takuya NishizawaDepartment of Pediatrics, Gunma University Graduate School of Medicine, Maebashi, Japan.
Takahiro KudoDepartment of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan.
Natsuki ItoDepartment of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan.
Yosuke KawaiDivision of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan.
Yoichi KakutaDivision of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan.
Masao NagasakiDivision of Biomedical Information Analysis, Medical Research Center for High Depth Omics, Medical Institute of Bioregulation, Kyushu University, Fukuoka, Japan.
Toshiaki ShimizuDepartment of Pediatrics, Juntendo University Faculty of Medicine, Tokyo, Japan.
Itaru IwamaCenter for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology and Hepatology, Saitama Children's Medical Center, Saitama, Japan.
Katsuhiro AraiCenter for Pediatric Inflammatory Bowel Disease, Division of Gastroenterology, National Center for Child Health and Development, Tokyo, Japan.

Funding

CURE:JPMXP1323015486 for MIBKyushu UniversityMedical Research Center Initiative for High Depth OmicsMEXT Cooperative Research Project Program
6 · The paper itself

Abstract

BACKGROUND/

aimsOccasionally, pediatric Crohn's disease (CD) may develop after diagnosis of orofacial granulomatosis (OFG), which is characterized by chronic granulomatous lesions of the oral mucosa, lips, and perioral area. This study aimed to clarify clinical characteristics, treatment responses, and potential genetic contributors in pediatric patients with CD complicating OFG.

methodsWe studied pediatric patients with CD complicating OFG who were treated from 2013 to 2022 at 7 Japanese institutions specializing in pediatric inflammatory bowel disease. Their clinical courses were analyzed retrospectively, and analyses of 71 genes associated with monogenic inflammatory bowel disease were performed.

resultsAmong 13 patients, 8 were girls. Median ages at diagnosis of OFG and CD were 9.2 (3.8-15.3) and 10.3 (6.4-15.3) years old, respectively. Upper gastrointestinal lesions were frequent in 8 cases (62%), while perianal lesions were present in 7 (54%). OFG failed to improve or relapsed despite remission of intestinal lesions in about half of the patients (n = 7, 54%). During follow-up, OFG went into remission in 7 patients, including 6 of the 9 who were treated with biologics (66%) and 1 of the 4 who were not (25%). In 8 patients, the NCF1 p.Arg90His allele was detected by genetic analysis; 7 were heterozygous and 1 homozygous, a higher prevalence than in the general Japanese population.

conclusionsClinical features of OFG associated with pediatric CD are diverse, and biologic agents were beneficial for OFG patients. NCF1 p.Arg90His mutation may contribute to the pathogenesis of pediatric CD complicating OFG.

Indexed as

GeneticsInflammatory bowel diseasesNCF1Pediatrics

Identifiers

PMID41448627
PMCPMC13434525

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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.