FHOD3 deficiency disrupts sarcomere organization and activates caMKII signaling in human stem cell-derived cardiomyocytes

Our study establishes a valuable human-derived model for investigating the molecular mechanisms of FHOD3 deficiency-induced cardiomyopathy. This model allows for extensive investigation into the phenotypes caused by FHOD3 deficiency and identifies CaMKII activation as a crucial factor contributing to the HF phenotype. Additionally, this model serves as an important tool for discovering novel therapeutic agents, and we demonstrate that OM can partially improve myocardial function in FHOD3 KO hESC-CMs.

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Publication Details

Journal
Stem Cell Research & Therapy
Published
2026-01-16
DOI
https://doi.org/10.1186/s13287-026-04902-z
Primary Topic
Congenital heart defects research
Type
article
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article

FHOD3 deficiency disrupts sarcomere organization and activates caMKII signaling in human stem cell-derived cardiomyocytes

Min Zhu, Mingyu Wei, Feng Lan, Ming Cui et al.
Stem Cell Research & Therapy
Congenital heart defects research
article

FHOD3 deficiency disrupts sarcomere organization and activates caMKII signaling in human stem cell-derived cardiomyocytes

Min Zhu, Mingyu Wei, Feng Lan, Ming Cui, Siyao Zhang, X Y Chen, Zhan Shi, X. Y. Hu, Shuwan Xu, Xiaojie Hou, Zhen Gao
article en

Abstract

Our study establishes a valuable human-derived model for investigating the molecular mechanisms of FHOD3 deficiency-induced cardiomyopathy. This model allows for extensive investigation into the phenotypes caused by FHOD3 deficiency and identifies CaMKII activation as a crucial factor contributing to the HF phenotype. Additionally, this model serves as an important tool for discovering novel therapeutic agents, and we demonstrate that OM can partially improve myocardial function in FHOD3 KO hESC-CMs.

Stem Cell Research & TherapyVol. 17(1)
Chinese Academy of Medical Sciences & Peking Union Medical College (CN), Peking University (CN), Sichuan University (CN), West China Hospital of Sichuan University (CN), Beijing Anzhen Hospital (CN), Peking University Third Hospital (CN), First Affiliated Hospital of Zhengzhou University (CN), Fu Wai Hospital (CN)
Good health and well-being
Openalex Percentile: Top 87%
Congenital heart defects research
DOIOpen Access PDFOpenAlex
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FHOD3 deficiency disrupts sarcomere organization and activates caMKII signaling in human stem cell-derived cardiomyocytes — Min Zhu, Mingyu Wei, et al. · Stem Cell Research & Therapy (2026) | TGRS Research Map | TGRS