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| Status |
Public on Mar 24, 2023 |
| Title |
ZNF469 plays a critical role in regulating synthesis of ECM |
| Organism |
Danio rerio |
| Experiment type |
Expression profiling by high throughput sequencing
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| Summary |
Purpose: In recent years, the mutation of ZNF469 is considered to be one of the pathogenesis of brittle cornea syndrome（BCS）. We establish a znf469 mutant zebrafish line to explore its phenotype and the possible molecular mechanism.
Methods: We generated the znf469 mutant zebrafish by using the CRISPR/Cas9 system. TEM is applied to examine the phenotype of the cornea in different development stage. RNA sequencing and quantitative RT-PCR are used to reveal the molecular mechanism.
Results: Macroscopically, the homozygous znf469-4del zebrafish larva exhibited a curved body from 72 hpf similar to kyphoscoliosis and a noninflated swimbladder at 7 dpf. TEM reveals the extreme reduction of corneal stroma in homozygous znf469-4del zebrafish in both central and peripheral cornea since the early development stage. RNA-seq analysis demonstrates that the znf469 mutation leads to the decreased synthesis of various ECM component like collagens and proteoglycans but increased synthesis of 26S proteasome families.
Conclusion: Our work indicates that znf469 is a critical gene that encoding a transcription factor, regulating the synthesis and degradation of a large number of ECM components which is also the pathologic basis of the ocular and extraocular phenotypes in the znf469 mutant zebrafish model.
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| Overall design |
Comparative gene expression profiling analysis of RNA-seq data for WT and znf469-4del zebrafish larvae at 7dpf.
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| Web link |
https://0-www-ncbi-nlm-nih-gov.brum.beds.ac.uk/pmc/articles/PMC10233312/
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| |
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| Contributor(s) |
Bao J, Yu X, Ping X, Shentu X, Zou J |
| Citation(s) |
37256609 |
| Submission date |
Feb 16, 2023 |
| Last update date |
Jun 23, 2023 |
| Contact name |
Jing Bao |
| E-mail(s) |
22018486@zju.edu.cn
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| Organization name |
The Second Affiliated Hospital of Zhejiang University
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| Street address |
88 Jiefang Road
|
| City |
Hangzhou |
| State/province |
Zhejiang |
| ZIP/Postal code |
310009 |
| Country |
China |
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| Platforms (1) |
| GPL24995 |
Illumina NovaSeq 6000 (Danio rerio) |
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| Samples (6)
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| Relations |
| BioProject |
PRJNA935945 |
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