Cellular and molecular mechanisms in the development of a cleft lip and/or cleft palate; insights from zebrafish (<scp><i>Danio rerio</i></scp>)

Atukorallaya, Devi; Ratnayake, Ravindra Kumar

doi:10.1002/ar.24547

Cited by 6 publications

(6 citation statements)

References 85 publications

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“…During palate development, the frontonasal mesenchyme process and the mesenchyme of the maxillary processes are derived from migratory neural crest cells. Previous research has suggested that the human primary palate is homologous to the medial ethmoid region and the secondary palate is homologous to the lateral ethmoid region of zebrafish [41,42]. The V-shaped fusion seam of the frontonasal process and the bilateral maxillary processes of the human palate are analogous to the V-shaped junction found in the zebrafish ethmoid cartilage.…”

Section: Introductionmentioning

confidence: 85%

Effect of Embryonic Alcohol Exposure on Craniofacial and Skin Melanocyte Development: Insights from Zebrafish (Danio rerio)

Zavareh

Silva

Gimhani

et al. 2022

Toxics

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Alcohol is a common addictive substance and prenatal alcohol exposure could cause fetal alcohol spectrum disorder (FASD) and can lead to various birth defects. The small teleost zebrafish (Danio rerio) has been identified as a fine animal model in developmental biology and toxicological research. Zebrafish models are widely used to study the harmful effects of alcohol and limited studies are available on the craniofacial and skin malformations associated with FASD. The present study attempts to investigate the effect of alcohol on early zebrafish embryonic development. The effects of prenatal alcohol exposure on neural crest cell-derived organ formation, including pharyngeal dentition, palatal bones and skin melanocytes were analysed. Whole-mount cartilage and bone staining and imaging techniques were applied to determine the effects of alcohol on the above-mentioned structures. The tooth size and shape were affected by alcohol exposure, but the number of teeth in the pharyngeal dentition was not affected. Only first-generation teeth showed size differences. The alcohol-exposed ethmoid bone, which is homologous to the human hard palate, was smaller and less dense in cell arrangement compared with the control medial ethmoid bone. The skin pigmentation defects included reduced melanocyte density, melanin contraction, smaller melanocyte surface area and aberrations in melanosome dispersion, revealing that alcohol significantly influenced and downregulated each and every step of the melanocyte developmental process. This descriptive study summarises the effects of alcohol on the development of neural crest cell-derived structures and highlights the importance of zebrafish in studying the phenotypic characteristics of fetal alcohol spectrum disorder.

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Section: Introductionmentioning

confidence: 85%

Effect of Embryonic Alcohol Exposure on Craniofacial and Skin Melanocyte Development: Insights from Zebrafish (Danio rerio)

Zavareh

Silva

Gimhani

et al. 2022

Toxics

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“…In this case, the microvilli of RPE cells interact with photoreceptor outer segments of the neural retina ( 20 ). Another example is the fusion of three prominences of the facial epidermis during the formation of the lips, mouth, and palate, in which a disturbed process results in a cleft of the lip or the palate ( 21 ) ( 22 ) ( 23 ). However, there are currently no reports of similar structures observed in these tissues.…”

Section: Discussionmentioning

confidence: 99%

Mechanical transapical coupling of endometrial epithelial cells during implantation

Sakurai,

Kinoshita,

Otani

et al. 2023

Preprint

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Epithelial cells lining the lumen of organs have an apical-basal polarity, and mechanical cell contacts are located in the lateral and basal regions. During early pregnancy in rodents, the luminal space of the uterus is closed, and opposing epithelial cells attach to each other at apical–apical surfaces. Here, we show that cells are mechanically coupled at the apical cell–cell contacts. The apical plasma membrane was intricately intertwined between opposing cells. Extracellular matrix and tight junction molecules were localized at apical contacts. Claudin inhibition resulted in impaired luminal closure, suggesting a functional requirement of claudins for the establishment of transapical coupling. The present results demonstrate a mechanical cell–cell interaction that occurs between apical–apical surfaces in addition to lateral and basal cell junctions.One Sentence SummariesTransapical coupling of epithelial cells mediated by extracellular matrix and tight junction molecules

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“…Impaired formation of the ethmoid plate in zebrafish is considered to be homologous to cleft palate in mammals. 6,[37][38][39][40][41] However, ethmoid plate formation in zebrafish occurs by convergence of frontonasal and maxillary neural crest cells, which is analogous to the mammalian hard palate. 41 As a result, the soft palate that forms approximately one-third of the roof of the mammalian mouth 42 is absent in zebrafish.…”

Section: Discussionmentioning

confidence: 99%

Involvement of homeobox transcription factor Mohawk in palatogenesis

Adachi

Higuchi

Wakai

et al. 2021

Congenital Anomalies

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Palatogenesis is affected by many factors, including gene polymorphisms and exposure to toxic chemicals during sensitive developmental periods. Cleft palate is one of the most common congenital anomalies, and ongoing efforts to elucidate the molecular mechanisms underlying palatogenesis are providing useful insights to reduce the risk of this disorder. To identify novel potential regulators of palatogenesis, we analyzed public transcriptome datasets from a mouse model of cleft palate caused by selective deletion of transforming growth factor-β (TGFβ) receptor type 2 in cranial neural crest cells. We identified the homeobox transcription factor Mohawk (Mkx) as a gene downregulated in the maxilla of TGFβ knockout mice compared with wild-type mice. To examine the role of mkx in palatogenesis, we used CRISPR/Cas9 editing to generate zebrafish with impaired expression of mkxa and mkxb, the zebrafish homologs of Mkx. We found that mkx crispants expressed reduced levels of gli1, a critical transcription factor in the Sonic hedgehog (SHH) signaling pathway that plays an important role in the regulation of palatogenesis. Furthermore, we found that mkxa À/À zebrafish were more susceptible than mkxa +/+ zebrafish to the deleterious effects of cyclopamine, an inhibitor of SHH signaling, on upper jaw development. These results suggest that Mkx may be involved in palatogenesis regulated by TGFβ and SHH signaling, and that impairment in Mkx function may be related to the etiology of cleft palate.

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Cellular and molecular mechanisms in the development of a cleft lip and/or cleft palate; insights from zebrafish (Danio rerio)

Cited by 6 publications

References 85 publications

Effect of Embryonic Alcohol Exposure on Craniofacial and Skin Melanocyte Development: Insights from Zebrafish (Danio rerio)

Effect of Embryonic Alcohol Exposure on Craniofacial and Skin Melanocyte Development: Insights from Zebrafish (Danio rerio)

Mechanical transapical coupling of endometrial epithelial cells during implantation

Involvement of homeobox transcription factor Mohawk in palatogenesis

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