Evidence map›Paper›PMID 38632277›Full record

ArticleScientific reports2024

HOIL-1L deficiency induces cell cycle alteration which causes immaturity of skeletal muscle and cardiomyocytes.

Kentaro Akagi, Shiro Baba, Hiroaki Fujita, Yasuhiro Fuseya, Daisuke Yoshinaga, Hirohito Kubota, Eitaro Kume, Fumiaki Fukumura, Koichi Matsuda, Takayuki Tanaka and 4 more

Erratum issuedAbstract 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. An erratum has been issued. Not yet cited in PubMed.

0numbers the graph read from it
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

5 · Who and what money

Authors and funding

14 authors.

Kentaro AkagiDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Shiro BabaDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan. shibaba@kuhp.kyoto-u.ac.jp.
Hiroaki FujitaDepartment of Molecular and Cellular Physiology, Graduate School of Medicine, Kyoto University, Yoshidakonoe-cho, Sakyo-ku, Kyoto City, Kyoto, 606-8501, Japan.
Yasuhiro FuseyaDepartment of Molecular and Cellular Physiology, Graduate School of Medicine, Kyoto University, Yoshidakonoe-cho, Sakyo-ku, Kyoto City, Kyoto, 606-8501, Japan.
Daisuke YoshinagaDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Hirohito KubotaDepartment of Molecular and Cellular Physiology, Graduate School of Medicine, Kyoto University, Yoshidakonoe-cho, Sakyo-ku, Kyoto City, Kyoto, 606-8501, Japan.
Eitaro KumeDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Fumiaki FukumuraDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Koichi MatsudaDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Takayuki TanakaDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Takuya HirataDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Megumu K SaitoDepartment of Clinical Application, Center for iPS Cell Research and Application (CiRA), Kyoto University, 53 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.
Kazuhiro IwaiDepartment of Molecular and Cellular Physiology, Graduate School of Medicine, Kyoto University, Yoshidakonoe-cho, Sakyo-ku, Kyoto City, Kyoto, 606-8501, Japan.
Junko TakitaDepartment of Pediatrics, Graduate School of Medicine, Kyoto University, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto City, Kyoto, 606-8507, Japan.

Funding

Grant-in-Aid for Scientific Research 20K08421the Core Center for Regenerative Medicine and Cell and Gene Therapy JP23bm1323001
6 · The paper itself

Abstract

HOIL-1L deficiency was recently reported to be one of the causes of myopathy and dilated cardiomyopathy (DCM). However, the mechanisms by which myopathy and DCM develop have not been clearly elucidated. Here, we sought to elucidate these mechanisms using the murine myoblast cell line C2C12 and disease-specific human induced pluripotent stem cells (hiPSCs). Myotubes differentiated from HOIL-1L-KO C2C12 cells exhibited deteriorated differentiation and mitotic cell accumulation. CMs differentiated from patient-derived hiPSCs had an abnormal morphology with a larger size and were excessively multinucleated compared with CMs differentiated from control hiPSCs. Further analysis of hiPSC-derived CMs showed that HOIL-1L deficiency caused cell cycle alteration and mitotic cell accumulation. These results demonstrate that abnormal cell maturation possibly contribute to the development of myopathy and DCM. In conclusion, HOIL-1L is an important intrinsic regulator of cell cycle-related myotube and CM maturation and cell proliferation.

Indexed as

Cell CycleInduced Pluripotent Stem CellsMuscular DiseasesUbiquitin-Protein LigasesAnimalsCell DifferentiationCell LineHumansMiceMuscle, SkeletalMyocytes, CardiacTranscription FactorsRBCK1 protein, humanRbck1 protein, mouseTranscription FactorsUbiquitin-Protein Ligases

Identifiers

PMID38632277
PMCPMC11024103

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.