Review
Dentistry, Oral Surgery & Medicine
D. A. Cruz Walma, K. M. Yamada
Summary: The extracellular matrix (ECM) plays a crucial role in shaping the development of craniofacial organs by coordinating cell movements and behaviors. Understanding how ECM regulates craniofacial development and contributes to disease and disorders is important for promoting regeneration and preventing degenerative outcomes.
JOURNAL OF DENTAL RESEARCH
(2022)
Review
Cell Biology
Heather E. White, Anjali Goswami, Abigail S. Tucker
Summary: Phenotypic variation in mammals is influenced by their ecological diversification, with the skull playing a crucial role in supporting species survival. Sutures between cranial bones are important in growth and brain development, shaping overall skull morphology and function. The developmental mechanisms of sutures have a significant impact on cranial diversity and evolutionary patterns.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Environmental Sciences
Hannah J. Petty, Jacob E. Barrett, Erin G. Kosmowski, Dandre S. Amos, Sean M. Ryan, Lucas D. Jones, Christopher S. Lassiter
Summary: In this study, the effects of spironolactone on cardiovascular and craniofacial development in zebrafish embryos were investigated. The results suggest that further research on the impact of spironolactone on embryonic development is necessary.
ENVIRONMENTAL TOXICOLOGY AND PHARMACOLOGY
(2022)
Article
Cell Biology
Ching-Fang Chang, Kari M. Brown, Yanfen Yang, Samantha A. Brugmann
Summary: Primary cilia, microtubule-based cellular organelles, play a critical role in cellular function and their dysfunction is associated with various disorders. The C2cd3 protein is essential for ciliogenesis and mutations in this gene are linked to certain ciliopathies. Genetic background was found to influence the phenotypic variation in mice models, suggesting a complex interplay between genetic factors and disease manifestation.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Developmental Biology
D'Juan T. Farmer, Punam Patel, Rachelle Choi, Chih-Yu Liu, J. Gage Crump
Summary: Irx genes play crucial roles in vertebrate skeletal development, with conserved requirements for shoulder blade formation, anterior neural crest precursors, and gill cartilages. Loss of Irx gene clusters leads to joint loss and cartilage fusions through inappropriate chondrogenesis, revealing diverse roles in the formation and segmentation of the facial skeleton.
Article
Cell Biology
Zoe Crane-Smith, Jeffrey Schoenebeck, Katy A. Graham, Paul S. Devenney, Lorraine Rose, Mark Ditzell, Eve Anderson, Joseph I. Thomson, Natasha Klenin, Deborah M. Kurrasch, Laura A. Lettice, Robert E. Hill
Summary: Conserved enhancers play a crucial role in regulating embryonic development, with particular importance in hypothalamic morphogenesis and adjacent cranial bone development. Differential levels of Shh expression have significant effects on neuronal population induction and DV patterning gene expression, impacting various aspects of embryonic development.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2021)
Article
Biology
Fatma F. Mohamed, Chunxi Ge, Shawn A. Hallett, Alec C. Bancroft, Randy T. Cowling, Noriaki Ono, Abdul-Aziz Binrayes, Barry Greenberg, Benjamin Levi, Vesa M. Kaartinen, Renny T. Franceschi
Summary: The development of the craniofacial skeleton requires interactions between progenitor cells and the extracellular matrix that is rich in collagen. Mutations in the DDR2 gene, which codes for a collagen receptor, have been linked to craniofacial abnormalities in humans. This study shows that mice lacking Ddr2 exhibit defects in craniofacial bones, including impaired calvarial growth and frontal suture formation. These defects are associated with abnormal collagen organization, chondrocyte proliferation, and orientation.
Article
Developmental Biology
Kelsey H. Elliott, Sai K. Balchand, Christian Louis Bonatto Paese, Ching-Fang Chang, Yanfen Yang, Kari M. Brown, Daniel T. Rasicci, Hao He, Konrad Thorner, Praneet Chaturvedi, Stephen A. Murray, Jing Chen, Aleksey Porollo, Kevin A. Peterson, Samantha A. Brugmann
Summary: This study provides a searchable transcriptomic resource for the primary ciliome, revealing differentially expressed genes with tissue and temporal specificity. The genes within the differentially expressed ciliome show a lower level of functional constraint across species, indicating organism and cell-specific functional adaptation. The functional validation of ciliary heterogeneity was performed by disrupting ciliary genes using Cas9 gene editing, which exhibited dynamic gene expression profiles during osteogenic differentiation of multipotent neural crest cells. Overall, this novel primary cilia-focused resource allows researchers to explore the correlation between tissue and cell-type specific functions, ciliary heterogeneity, and the range of phenotypes associated with ciliopathies.
Article
Environmental Sciences
Melanie K. Stewart, Luca Hoehne, Stefanie Dudczig, Deidre M. Mattiske, Andrew J. Pask, Patricia R. Jusuf
Summary: Endocrine disrupting chemicals (EDCs) can interfere and disrupt hormone signalling necessary for various developmental pathways by interacting with native hormone receptors. These chemicals are pervasive in our environment, particularly in our waterways, making aquatic wildlife highly vulnerable to their effects. Many EDCs can bind to and activate oestrogen receptors, leading to aberrant oestrogen signalling.
ECOTOXICOLOGY AND ENVIRONMENTAL SAFETY
(2023)
Article
Multidisciplinary Sciences
Nora Alhazmi, Shannon H. Carroll, Kenta Kawasaki, Katherine C. Woronowicz, Shawn A. Hallett, Claudio Macias Trevino, Edward B. Li, Roland Baron, Francesca Gori, Pamela C. Yelick, Matthew P. Harris, Eric C. Liao
Summary: The study highlights the differential functions of Rspo2 and Rspo3 in dentocranial morphogenesis in zebrafish and in mouse, showcasing their synergistic roles in cartilage morphogenesis, fin development, and pharyngeal tooth development. Adult rspo3(-/-) zebrafish mutants exhibit a dysmorphic cranial skeleton and decreased average tooth number, demonstrating the importance of Rspo3 in craniofacial development.
SCIENTIFIC REPORTS
(2021)
Article
Medicine, Research & Experimental
Shuyu Guo, Li Meng, Haojie Liu, Lichan Yuan, Na Zhao, Jieli Ni, Yang Zhang, Jingling Ben, Yi-Ping Li, Jinqing Ma
Summary: The Trio gene interacts with Myh9 to play a key role in neural crest cell migration and differentiation during craniofacial development, affecting craniofacial growth. Injecting specific genes into zebrafish with Trio deficiency partially rescues craniofacial abnormalities, providing potential model systems to study the pathogenic mechanisms of Trio mutations.
Article
Biochemistry & Molecular Biology
Ranjeet D. Kar, Johann K. Eberhart
Summary: Most human birth defects have variable phenotypes, which are thought to be caused by complex gene-environment interactions. Mutations in the Gata3 gene can lead to birth defects, and zebrafish gata3 mutants mimic the variability seen in humans. In this study, an unbiased bioinformatic approach was used to identify environmental modifiers of gata3 mutant craniofacial phenotypes.
INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES
(2023)
Article
Cell Biology
Zhaohui Wei, Qiang Hong, Zijiao Ding, Jingwen Liu
Summary: The study generated mutant lines for cxcl12a and cxcr4b using the CRISPR/Cas9 system. Results showed that Cxcl12a is crucial for pharyngeal cartilage formation by promoting the proliferation of craniofacial neural crest cells. Additionally, Cxcl12a and Cxcl12b were found to have a synergistic influence on pharyngeal arch and pouch formation.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2023)
Article
Cell Biology
Kristyna Olbertova, Dusan Hrckulak, Vitezslav Kriz, Wojciech Jesionek, Jan Kubovciak, Milan Esner, Vladimir Korinek, Marcela Buchtova
Summary: In this study, the distribution of LGR5-positive cells during craniofacial development was analyzed using Lgr5-EGFP-CreERT2 mice. LGR5 expressing cells were primarily located in the mesenchyme adjacent to the craniofacial epithelial structures undergoing folding. Knockout of the Lgr5 gene led to distinct defects in LGR5-positive areas and abnormal development of craniofacial structures. These findings suggest a novel role of LGR5 in the regulation of morphogenetic processes during the formation of complex epithelial structures in the craniofacial areas.
FRONTIERS IN CELL AND DEVELOPMENTAL BIOLOGY
(2022)
Review
Pediatrics
Luana Nosetti, Marco Zaffanello, Francesca De Bernardi di Valserra, Daniela Simoncini, Giulio Beretta, Pietro Guacci, Giorgio Piacentini, Massimo Agosti
Summary: Adenotonsillar hypertrophy is the main cause of sleep-disordered breathing in children, and adenotonsillectomy is the go-to treatment. The importance of this research lies in revealing the close connection between adenotonsillar hypertrophy and craniofacial development, providing new insights for the treatment and prevention of related complications.
