Evidence map›Paper›PMID 39681577›Full record

ArticleScientific reports2024

Sarcomere gene variants did not improve cardiac function in pediatric patients with dilated cardiomyopathy from Japanese cohorts.

Keiichi Hirono, Yukiko Hata, Shojiro Ichimata, Naoki Nishida, Teruhiko Imamura, Yoshihiro Asano, Yuki Kuramoto, Kaori Tsuboi, Shinya Takarada, Mako Okabe and 47 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Scientific reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
–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

2 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Geographical and Ethnic Heterogeneity in Genetic Dilated Cardiomyopathies.International journal of molecular sciences · 2026
    Pooled it
  2. 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

57 authors.

Keiichi HironoDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan. khirono@med.u-toyama.com.
Yukiko HataLegal Medicine, University of Toyama, Toyama, Japan.
Shojiro IchimataLegal Medicine, University of Toyama, Toyama, Japan.
Naoki NishidaLegal Medicine, University of Toyama, Toyama, Japan.
Teruhiko Imamura2Nd Department of Internal Medicine, Faculty of Medicine, University of Toyama, Toyama, Japan.
Yoshihiro AsanoNational Cerebral and Cardiovascular Center, Osaka, Japan.
Yuki KuramotoDepartment of Cardiovascular Medicine, Osaka University Graduate School of Medicine, Osaka, Japan.
Kaori TsuboiDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Shinya TakaradaDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Mako OkabeDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Hideyuki NakaokaDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Keijiro IbukiDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Sayaka OzawaDepartment of Pediatrics, Faculty of Medicine, University of Toyama, Toyama City, 2630 Sugitani, Toyama, 930-0194, Japan.
Jun MuneuchiDepartment of Pediatrics, Kyushu Hospital, Japan Community Healthcare Organization, Fukuoka, Japan.
Kazushi YasudaDepartment of Pediatric Cardiology, Aichi Children's Health and Medical Center, Aichi, Japan.
Kotaro UrayamaDepartment of Pediatrics, Tsuchiya General Hospital, Hiroshima, Japan.
Hideharu OkaDepartment of Pediatrics, Asahikawa Medical University Hospital, Hokkaido, Japan.
Tomoyuki MiyamotoDepartment of Pediatrics, Yokosuka General Hospital Uwamachi, Kanagawa, Japan.
Kenji BabaDepartment of Pediatrics, Okayama University Graduate School of Medicine, Okayama, Japan.
Akio KatoDepartment of Pediatric Cardiology, Okinawa Prefectural Nanbu Medical Center and Children's Medical Center, Okinawa, Japan.
Hirofumi SaikiDepartment of Pediatrics and Pediatric Cardiology, Iwate Medical University School of Medicine, Iwate, Japan.
Naoki KuwabaraDepartment of Pediatric Cardiology, Gifu Prefectural General Medical Center, Gifu, Japan.
Masako HaradaDivision of Pediatrics, Developmental and Urological-Reproductive Medicine, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan.
Shiro BabaDepartment of Pediatrics, Kyoto University Hospital, Kyoto, Japan.
Mari MorikawaDepartment of Pediatrics, Kanazawa Medical University, Ishikawa, Japan.
Hidenori IwasakiDepartment of Pediatrics, Kanazawa University Hospital, Ishikawa, Japan.
Yuichiro HirataDepartment of Pediatrics, Kyushu University Hospital, Fukuoka, Japan.
Yuki ItoDepartment of Pediatric Cardiology, National Cerebral and Cardiovascular Center, Osaka, Japan.
Heima SakaguchiDepartment of Pediatric Cardiology, National Cerebral and Cardiovascular Center, Osaka, Japan.
Susumu UrataDivision of Cardiology, National Center for Child Health and Development, Tokyo, Japan.
Koichi TodaDepartment of Pediatric Cardiology, Saitama Medical University International Medical Center, Saitama, Japan.
Emi KittakaDepartment of Cardiology, Saitama Children's Medical Center, Saitama, Japan.
Seigo OkadaDepartment of Pediatrics, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan.
Yohei HasebeDepartment of Pediatrics, Faculty of Medicine, University of Yamanashi, Yamanashi, Japan.
Shinsuke HoshinoDepartment of Pediatrics, Shiga University of Medical Science, Shiga, Japan.
Takanari FujiiPediatric Heart Disease and Adult Congenital Heart Disease Center, Showa University Hospital, Tokyo, Japan.
Norie MitsushitaDepartment of Cardiology, Shizuoka Children's Hospital, Shizuoka, Japan.
Masaki NiiDepartment of Cardiology, Shizuoka Children's Hospital, Shizuoka, Japan.
Kayo OginoDepartment of Pediatrics, Kurashiki Central Hospital, Okayama, Japan.
Mitsuhiro FujinoDepartment of Pediatric Cardiology, Osaka City General Hospital, Osaka, Japan.
Yoko YoshidaDecision of Pediatric Electrophysiology, Osaka City General Hospital, Osaka, Japan.
Yutaka FukudaDepartment of Pediatrics, Takeda General Hospital, Fukushinma, Japan.
Satoru IwashimaDepartment of Pediatrics, Chutoen General Medical Center, Shizuoka, Japan.
Kiyohiro TakigikuDepartment of Pediatric Cardiology, Nagano Children's Hospital, Nagano, Japan.
Yasushi SakataDivision of Pediatrics and Perinatology, Faculty of Medicine, Tottori University, Tottori, Japan.
Ryo InuzukaDepartment of Pediatrics, Tokyo University Hospital, Tokyo, Japan.
Jun MaedaDivision of Cardiology, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan.
Yasunobu HayabuchiDepartment of Pediatrics, Tokushima University Hospital, Tokushima, Japan.
Tao FujiokaDepartment of Pediatrics, Japanese Red Cross Medical Center, Tokyo, Japan.
Hidemasa NamikiDepartment of Pediatrics and Child Health, Nihon University School of Medicine, Tokyo, Japan.
Shuhei FujitaDepartment of Pediatrics, Toyama Prefectural Central Hospital, Toyama, Japan.
Koichi NishidaDepartment of Pediatrics, Fukui Cardiovascular Center, Fukui, Japan.
Ayako KuraokaDepartment of Cardiology, Fukuoka Children's Hospital, Fukuoka, Japan.
Nobuhiko KanDepartment of Fetal and Neonatal Cardiology, Fukuoka Children's Hospital, Fukuoka, Japan.
Sachiko KidoDepartment of Cardiology, Hyogo Prefectural Kobe Children's Hospital, Kobe, Japan.
Ken WatanabeDepartment of Pediatrics, Kitano Hospital Tazuke Kofukai Medical Research Institute, Osaka, Japan.
Fukiko IchidaDepartment of Pediatrics, International University of Health and Welfare, Tokyo, Japan.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Dilated cardiomyopathy (DCM) is a progressive myocardial disorder characterized by impaired cardiac contraction and ventricular dilation. However, some patients with DCM improve when experiencing left ventricular reverse remodeling (LVRR). Currently, the detailed association between genotypes and clinical outcomes, including LVRR, particularly among children, remains uncertain. Pediatric patients with DCM from multiple Japanese institutions recorded between 2014 and 2023 were enrolled. We identified their DCM-related genes and explored the association between gene variants and clinical outcomes, including LVRR. We included 123 pediatric patients (62 males; median age: 8 [1-51] months) and found 50 pathogenic variants in 45 (35.0%) of them. The most identified gene was MYH7 (14.0%), followed by RYR2 (12.0%) and TPM1 (8.0%). LVRR was achieved in 47.5% of these patients. The left ventricular ejection fraction remained unchanged (31.4% to 39.8%, P = 0.1913) in patients with sarcomere gene variants and in those with non-sarcomere gene variants (33.4% to 47.8%, P = 0.0522) but significantly increased in those without gene variants (33.6% to 54.1%, P < 0.0001). LVRR was not uniform across functional gene groups. Hence, an individualized gene-guided prediction approach may be adopted for children with DCM.

Indexed as

Cardiomyopathy, DilatedSarcomeresCardiac MyosinsChildChild, PreschoolCohort StudiesEast Asian PeopleFemaleGenetic VariationHumansInfantJapanMaleMyosin Heavy ChainsVentricular Function, LeftVentricular RemodelingCardiac MyosinsMYH7 protein, humanMyosin Heavy ChainsDilated cardiomyopathyGeneticsHeart failureLeft ventricular reverse remodelingSarcomere gene

Identifiers

PMID39681577
PMCPMC11649763

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