干旱区生态环境知识资源中心(Arid): Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio

Arid

DOI	10.1002/dvdy.726
	Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
	VanWinkle, Peyton E.; Lee, Eunjoo; Wynn, Bridge; Nawara, Tomasz J.; Thomas, Holly; Parant, John; Alvarez, Cecilia; Serra, Rosa; Sztul, Elizabeth
通讯作者	VanWinkle, PE
来源期刊	DEVELOPMENTAL DYNAMICS
ISSN	1058-8388
EISSN	1097-0177
出版年	2024
英文摘要	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.
英文关键词	proximo-distal axis ptch2 runx2a shha zebrafish creb3l1 fin bifurcation fin regeneration
类型	Article ; Early Access
语种	英语
收录类别	SCI-E
WOS记录号	WOS:001267281400001
WOS关键词	TRANSCRIPTION FACTOR OASIS ; STRESS TRANSDUCER OASIS ; SONIC-HEDGEHOG ; CREB/ATF-FAMILY ; EXPRESSION ; ROLES ; CELLS ; PHOSPHORYLATION ; DIFFERENTIATION ; OSTEOBLASTS
WOS类目	Anatomy & Morphology ; Developmental Biology
WOS研究方向	Anatomy & Morphology ; Developmental Biology
资源类型	期刊论文
条目标识符	http://119.78.100.177/qdio/handle/2XILL650/403296
推荐引用方式 GB/T 7714	VanWinkle, Peyton E.,Lee, Eunjoo,Wynn, Bridge,et al. Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio[J],2024.
APA	VanWinkle, Peyton E..,Lee, Eunjoo.,Wynn, Bridge.,Nawara, Tomasz J..,Thomas, Holly.,...&Sztul, Elizabeth.(2024).Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio.DEVELOPMENTAL DYNAMICS.
MLA	VanWinkle, Peyton E.,et al."Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio".DEVELOPMENTAL DYNAMICS (2024).