Project information
Propojení funkce Sprouty s FGF a primárními ciliemi ve vývoji
- Project Identification
- GA21-04178S
- Project Period
- 1/2021 - 12/2023
- Investor / Pogramme / Project type
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Czech Science Foundation
- Standard Projects
- MU Faculty or unit
- Faculty of Science
- Cooperating Organization
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Institute of Animal Physiology and Genetics of thr ASCR, v. v. i.
Charles University
- Responsible person prof. Abigail Tucker
Sprouty proteins provide a control mechanism for appropriate receptor tyrosine kinase signaling, including FGF signaling pathway. Deficiencies in mouse Sprouties cause developmental abnormalities leading to hearing impairment, craniofacial defects, retarded postnatal growth or esophageal achalasia and intestinal pseudo-obstruction. In human, Sprouty2 has been identified to play a role in nephropathy, thanatophoric dysplasia and different types of cancer. Interestingly, fibroblast growth factor (FGF) signaling hyperactivation
syndromes and syndromes associated with primary cilia dysfunction share a range of common phenotypic manifestations, beside others, craniofacial and achondroplasia malformations. The project aims to characterize Sprouty proteins role during process of skeletogenesis. Further, we will analyze mouse model with deletion of Sprouty2 and Sprouty 4 to determine tissues and
organs developmental defects implicated in ciliopathies and evaluate primary cilia associated signaling during morphogenesis in Sprouty deficient mice.
Publications
Total number of publications: 5
2023
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Molecular profiling of the vestibular lamina highlights a key role for Hedgehog signalling
Development, year: 2023, volume: 150, edition: 7, DOI
2022
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The Development of Dentin Microstructure Is Controlled by the Type of Adjacent Epithelium
Journal of Bone and Mineral Research, year: 2022, volume: 37, edition: 2, DOI
2021
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Loss of Sprouty produces a ciliopathic skeletal phenotype in mice through elongation of primary cilia and upregulation of Hedgehog signaling
Year: 2021, type: Conference abstract
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Loss of Sprouty Produces a Ciliopathic Skeletal Phenotype in Mice Through Upregulation of Hedgehog Signaling
Journal of Bone and Mineral Research, year: 2021, volume: 36, edition: 11, DOI
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Ztráta Sprouty2/4 u myší vede k ciliopatickému fenotypu
Year: 2021, type: Conference abstract