Minerva Foundation Institute for Medical Research, Helsinki, Finland.
Department of Anatomy, University of Helsinki, Helsinki, Finland.
J Cell Mol Med. 2024 Apr;28(7):e18243. doi: 10.1111/jcmm.18243.
Humans lacking heme oxygenase 1 (HMOX1) display growth retardation, haemolytic anaemia, and vulnerability to stress; however, cardiac function remains unclear. We aimed to explore the cardiac function of zebrafish lacking hmox1a at baseline and in response to stress. We generated zebrafish hmox1a mutants using CRISPR/Cas9 genome editing technology. Deletion of hmox1a increases cardiac output and further induces hypertrophy in adults. Adults lacking hmox1a develop myocardial interstitial fibrosis, restrain cardiomyocyte proliferation and downregulate renal haemoglobin and cardiac antioxidative genes. Larvae lacking hmox1a fail to respond to hypoxia, whereas adults are insensitive to isoproterenol stimulation in the heart, suggesting that hmox1a is necessary for cardiac response to stress. Haplodeficiency of hmox1a stimulates non-mitochondrial respiration and cardiac cell proliferation, increases cardiac output in larvae in response to hypoxia, and deteriorates cardiac function and structure in adults upon isoproterenol treatment. Intriguingly, haplodeficiency of hmox1a upregulates cardiac hmox1a and hmox1b in response to isoproterenol. Collectively, deletion of hmox1a results in cardiac remodelling and abrogates cardiac response to hypoxia and isoproterenol. Haplodeficiency of hmox1a aggravates cardiac response to the stress, which could be associated with the upregulation of hmox1a and hmox1b. Our data suggests that HMOX1 homeostasis is essential for maintaining cardiac function and promoting cardioprotective effects.
缺乏血红素加氧酶 1(HMOX1)的人类表现出生长迟缓、溶血性贫血和对压力敏感;然而,心脏功能仍不清楚。我们旨在探索缺乏 hmox1a 的斑马鱼的心脏功能在基线和应激反应中的情况。我们使用 CRISPR/Cas9 基因组编辑技术生成了缺乏 hmox1a 的斑马鱼突变体。hmox1a 的缺失增加了心输出量,并进一步导致成年鱼发生肥大。缺乏 hmox1a 的成年鱼发生心肌间质纤维化,抑制心肌细胞增殖,并下调肾脏血红蛋白和心脏抗氧化基因。缺乏 hmox1a 的幼虫不能对缺氧做出反应,而成年鱼对心脏中的异丙肾上腺素刺激不敏感,这表明 hmox1a 对于心脏对压力的反应是必要的。hmox1a 的单倍体缺陷刺激非线粒体呼吸和心脏细胞增殖,增加幼虫对缺氧的心脏输出量,并在异丙肾上腺素处理时恶化成年鱼的心脏功能和结构。有趣的是,hmox1a 的单倍体缺陷在对异丙肾上腺素的反应中上调心脏 hmox1a 和 hmox1b。总之,hmox1a 的缺失导致心脏重塑,并取消了心脏对缺氧和异丙肾上腺素的反应。hmox1a 的单倍体缺陷加重了心脏对压力的反应,这可能与 hmox1a 和 hmox1b 的上调有关。我们的数据表明,HMOX1 动态平衡对于维持心脏功能和促进心脏保护作用至关重要。