Czyrek Aleksandra A, Baran Karolina, Hruba Eva, Horackova Aneta, Bosakova Veronika, Chudzian Julia, Fafilek Bohumil, Laskova Veronika, Stepankova Veronika, Bednar David, Karl Kelly, Kasparek Petr, Bosakova Michaela, Killinger Michal, Szotkowska Tereza, Prochazka Jan, Zieba Jennifer T, Rico-Llanos Gustavo, Fric Jan, Hadzic Stefan, Loku Edma, Wujak Magdalena, Svozilova Katerina, Stroblova Michaela, Sedlacek Radislav, Hristova Kalina, Krakow Deborah, Kubovciak Jan, Delattre Mathys, Bartoszewski Rafal, Buchtova Marcela, Krowarsch Daniel, Chaloupkova Radka, Zakrzewska Malgorzata, Krejci Pavel
Department of Biology, Faculty of Medicine, Masaryk University, Brno, 62500, Czech Republic.
International Clinical Research Center, St. Anne's University Hospital, Brno, 65691, Czech Republic.
Cell Mol Life Sci. 2025 Apr 21;82(1):167. doi: 10.1007/s00018-025-05681-1.
Fibroblast growth factors (FGFs) control organ morphogenesis during development as well as tissue homeostasis and repair in the adult organism. Despite their importance, many mechanisms that regulate FGF function are still poorly understood. Interestingly, the thermodynamic stability of 22 mammalian FGFs varies widely, with some FGFs remaining stable at body temperature for more than 24 h, while others lose their activity within minutes. How thermodynamic stability contributes to the function of FGFs during development remains unknown. Here we show that FGF10, an important limb and lung morphogen, exists as an intrinsically unstable protein that is prone to unfolding and is rapidly inactivated at 37 °C. Using rationally driven directed mutagenesis, we have developed several highly stable (STAB) FGF10 variants with a melting temperature of over 19 °C more than that of wildtype FGF10. In cellular assays in vitro, the FGF10-STABs did not differ from wildtype FGF10 in terms of binding to FGF receptors, activation of downstream FGF receptor signaling in cells, and induction of gene expression. In mouse embryonal lung explants, FGF10-STABs, but not wildtype FGF10, suppressed branching, resulting in increased alveolarization and expansion of epithelial tissue. Similarly, FGF10-STAB1, but not FGF10 wildtype, inhibited the growth of mouse embryonic tibias and markedly altered limb morphogenesis when implanted into chicken limb buds, collectively demonstrating that thermal instability should be considered an important regulator of FGF function that prevents ectopic signaling. Furthermore, we show enhanced differentiation of human iPSC-derived lung organoids and improved regeneration in ex vivo lung injury models mediated by FGF10-STABs, suggesting an application in cell therapy.
成纤维细胞生长因子(FGFs)在发育过程中控制器官形态发生,以及成体生物中的组织稳态和修复。尽管它们很重要,但许多调节FGF功能的机制仍知之甚少。有趣的是,22种哺乳动物FGFs的热力学稳定性差异很大,一些FGFs在体温下可保持稳定超过24小时,而另一些则在几分钟内失去活性。热力学稳定性在发育过程中如何促进FGFs的功能仍不清楚。在这里,我们表明FGF10是一种重要的肢体和肺形态发生素,它是一种本质上不稳定的蛋白质,易于展开并在37°C下迅速失活。通过合理驱动的定向诱变,我们开发了几种高度稳定的(STAB)FGF10变体,其解链温度比野生型FGF10高19°C以上。在体外细胞试验中,FGF10-STABs在与FGF受体结合、激活细胞内下游FGF受体信号以及诱导基因表达方面与野生型FGF10没有差异。在小鼠胚胎肺外植体中,FGF10-STABs而非野生型FGF10抑制分支,导致肺泡化增加和上皮组织扩张。同样,FGF10-STAB1而非FGF10野生型抑制小鼠胚胎胫骨的生长,并在植入鸡胚肢芽时显著改变肢体形态发生,共同表明热不稳定性应被视为防止异位信号传导的FGF功能的重要调节因子。此外,我们表明FGF10-STABs介导的人诱导多能干细胞来源的肺类器官的分化增强,以及离体肺损伤模型中的再生改善,提示其在细胞治疗中的应用。