断裂 creb3l1 基因会导致斑马鱼 Danio rerio 尾鳍再生和模式化缺陷。

IF 2 3区 生物学 Q2 ANATOMY & MORPHOLOGY Developmental Dynamics Pub Date : 2024-07-14 DOI:10.1002/dvdy.726
Peyton E. VanWinkle, Eunjoo Lee, Bridge Wynn, Tomasz J. Nawara, Holly Thomas, John Parant, Cecilia Alvarez, Rosa Serra, Elizabeth Sztul
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引用次数: 0

摘要

背景:cAMP反应元件结合蛋白3-like-1(CREB3L1)基因与小鼠的骨骼发育有关,CREB3L1基因敲除的小鼠骨骼脆弱,而在人类,CREB3L1基因突变与成骨不全症有关。然而,Creb3l1调控骨骼发育的机制尚不完全清楚:为了探究Creb3l1在生物生理学中的作用,我们利用CRISPR-Cas9基因组编辑技术生成了Creb3l1缺乏的斑马鱼模型。与哺乳动物的表型不同,Creb3l1缺失的斑马鱼除了尾鳍分叉模式减少外,并没有表现出骨生成异常。突变鱼的骨骼形态和整体骨密度均正常。但是,突变体后尾鳍的再生能力明显受到影响,整体再生和矿化骨面积减少。此外,突变体鱼在再生过程中表现出严重的模式化缺陷,鳍条的分叉复杂性显著降低,分叉部位远端化。对骨骼发育相关基因的分析表明,在Creb3l1缺陷鱼体内,shha和ptch2的模式化出现异常,这将Creb3l1与鳍再生过程中的Sonic Hedgehog信号传导联系起来:我们的研究结果揭示了Creb3l1在再生过程中调节组织生长和形态的新作用。
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Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio

Background

The gene cAMP-Responsive Element Binding protein 3-like-1 (CREB3L1) has been implicated in bone development in mice, with CREB3L1 knock-out mice exhibiting fragile bones, and in humans, with CREB3L1 mutations linked to osteogenesis imperfecta. However, the mechanism through which Creb3l1 regulates bone development is not fully understood.

Results

To probe the role of Creb3l1 in organismal physiology, we used CRISPR-Cas9 genome editing to generate a Danio rerio (zebrafish) model of Creb3l1 deficiency. In contrast to mammalian phenotypes, the Creb3l1 deficient fish do not display abnormalities in osteogenesis, except for a decrease in the bifurcation pattern of caudal fin. Both, skeletal morphology and overall bone density appear normal in the mutant fish. However, the regeneration of caudal fin postamputation is significantly affected, with decreased overall regenerate and mineralized bone area. Moreover, the mutant fish exhibit a severe patterning defect during regeneration, with a significant decrease in bifurcation complexity of the fin rays and distalization of the bifurcation sites. Analysis of genes implicated in bone development showed aberrant patterning of shha and ptch2 in Creb3l1 deficient fish, linking Creb3l1 with Sonic Hedgehog signaling during fin regeneration.

Conclusions

Our results uncover a novel role for Creb3l1 in regulating tissue growth and patterning during regeneration.

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来源期刊
Developmental Dynamics
Developmental Dynamics 生物-发育生物学
CiteScore
5.10
自引率
8.00%
发文量
116
审稿时长
3-8 weeks
期刊介绍: Developmental Dynamics, is an official publication of the American Association for Anatomy. This peer reviewed journal provides an international forum for publishing novel discoveries, using any model system, that advances our understanding of development, morphology, form and function, evolution, disease, stem cells, repair and regeneration.
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