Zebrafish sp7 mutants show tooth cycling independent of attachment, eruption and poor differentiation of teeth

Kague, Érika; Witten, P. Eckhard; Soenens, Mieke; Campos, Camila Lovaglio; Lubiana, Tiago; Fisher, Shannon; Hammond, Chrissy L.; Brown, Kate Robson; Passos‐Bueno, Maria Rita; Huysseune, Ann

doi:10.1016/j.ydbio.2018.01.021

Cited by 26 publications

(22 citation statements)

References 36 publications

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“…As a bone-specific transcription factor 45, SP7 is required for the differentiation of mesenchymal stem cells and osteoblasts as well as bone formation 46. Diseases associated with SP7, which include osteogenesis imperfecta and zebrafish sp7 mutants, cause poor differentiation of teeth 47. Both the intensity of alizarin red staining in craniofacial bones and teeth as well as the expression of col10a1 and sp7 were reduced in clcn7 morphants, indicating that ClC-7 deficiency impaired the calcification of craniofacial bones and teeth.…”

Section: Discussionmentioning

confidence: 99%

ClC-7 Regulates the Pattern and Early Development of Craniofacial Bone and Tooth

Ji¹,

Li²,

Yang³

et al. 2019

Theranostics

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Human CLCN7 encodes voltage-gated chloride channel 7 (ClC-7); mutations of CLCN7 lead to osteopetrosis which is characterized by increased bone mass and impaired osteoclast function. In our previous clinical practice, we noticed that osteopetrosis patients with CLCN7 mutations had some special deformities in craniofacial morphology and tooth dysplasia. It is unclear whether these phenotypes are the typical features of CLCN7 involved osteopetrosis and whether ClC-7 could regulate the development of craniofacial bone and tooth in some signaling pathways. Methods : First, we collected 80 osteopetrosis cases from the literature and compared their craniofacial and dental phenotypes. Second, four osteopetrosis pedigrees with CLCN7 mutations were recruited from our clinic for gene testing and clinical analysis of their craniofacial and dental phenotypes. Third, we used a zebrafish model with clcn7 morpholino treatment to detect the effects of ClC-7 deficiency on the development of craniofacial and dental phenotypes. General observation, whole mount alcian blue and alizarin red staining, whole mount in situ hybridization, scanning electron microscope observation, lysoSensor staining, Q-PCR and western blotting were performed to observe the in vivo characteristics of craniofacial bone and tooth changes. Fourth, mouse marrow stromal cells were further primarily cultured to detect ClC-7 related mRNA and protein changes using siRNA, Q-PCR and western blotting. Results: Over 84% of osteopetrosis patients in the literature had some typical craniofacial and tooth phenotypes, including macrocephaly, frontal bossing, and changes in shape and proportions of facial skeleton, and these unique features are more severe and frequent in autosomal recessive osteopetrosis than in autosomal dominant osteopetrosis patients. Our four pedigrees with CLCN7 mutations confirmed the aforementioned clinical features. clcn7 knockdown in zebrafish reproduced the craniofacial cartilage defects and various dental malformations combined the decreased levels of col10a1 , sp7 , dlx2b , eve1 , and cx43 . Loss of clcn7 function resulted in lysosomal storage in the brain and jaw as well as downregulated cathepsin K (CTSK). The craniofacial phenotype severity also presented a dose-dependent relationship with the levels of ClC-7 and CTSK. ClC-7/CTSK further altered the balance of TGF-β/BMP signaling pathway, causing elevated TGF-β-like Smad2 signals and reduced BMP-like Smad1/5/8 signals in clcn7 morphants. SB431542 inhibitor of TGF-β pathway partially rescued the aforementioned craniofacial bone and tooth defects of ...

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Section: Discussionmentioning

confidence: 99%

ClC-7 Regulates the Pattern and Early Development of Craniofacial Bone and Tooth

Ji¹,

Li²,

Yang³

et al. 2019

Theranostics

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“…The SDL was recently shown to be a key tissue in tooth regeneration: mouse molars, which do not naturally undergo regeneration, can be prompted to do so by stabilizing Wnt signaling in the rudimentary mouse SDL [32]. In most cases, a replacement tooth germ eventually prompts osteoclast activity to enzymatically dissociate the base of the predecessor tooth and remodel the underlying bone of attachment (if present) either before or during early eruption [33][34][35][36][37]. This osteoclast activity facilitates the shedding of the predecessor and makes space for the replacement tooth to finish mineralization and eruption, thus completing the regenerative process.…”

Section: Introductionmentioning

confidence: 99%

Distinct tooth regeneration systems deploy a conserved battery of genes

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Sundaram

Mackey

et al. 2021

EvoDevo

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Background Vertebrate teeth exhibit a wide range of regenerative systems. Many species, including most mammals, reptiles, and amphibians, form replacement teeth at a histologically distinct location called the successional dental lamina, while other species do not employ such a system. Notably, a ‘lamina-less’ tooth replacement condition is found in a paraphyletic array of ray-finned fishes, such as stickleback, trout, cod, medaka, and bichir. Furthermore, the position, renewal potential, and latency times appear to vary drastically across different vertebrate tooth regeneration systems. The progenitor cells underlying tooth regeneration thus present highly divergent arrangements and potentials. Given the spectrum of regeneration systems present in vertebrates, it is unclear if morphologically divergent tooth regeneration systems deploy an overlapping battery of genes in their naïve dental tissues. Results In the present work, we aimed to determine whether or not tooth progenitor epithelia could be composed of a conserved cell type between vertebrate dentitions with divergent regeneration systems. To address this question, we compared the pharyngeal tooth regeneration processes in two ray-finned fishes: zebrafish (Danio rerio) and threespine stickleback (Gasterosteus aculeatus). These two teleost species diverged approximately 250 million years ago and demonstrate some stark differences in dental morphology and regeneration. Here, we find that the naïve successional dental lamina in zebrafish expresses a battery of nine genes (bmpr1aa, bmp6, cd34, gli1, igfbp5a, lgr4, lgr6, nfatc1, and pitx2), while active Wnt signaling and Lef1 expression occur during early morphogenesis stages of tooth development. We also find that, despite the absence of a histologically distinct successional dental lamina in stickleback tooth fields, the same battery of nine genes (Bmpr1a, Bmp6, CD34, Gli1, Igfbp5a, Lgr4, Lgr6, Nfatc1, and Pitx2) are expressed in the basalmost endodermal cell layer, which is the region most closely associated with replacement tooth germs. Like zebrafish, stickleback replacement tooth germs additionally express Lef1 and exhibit active Wnt signaling. Thus, two fish systems that either have an organized successional dental lamina (zebrafish) or lack a morphologically distinct successional dental lamina (sticklebacks) deploy similar genetic programs during tooth regeneration. Conclusions We propose that the expression domains described here delineate a highly conserved “successional dental epithelium” (SDE). Furthermore, a set of orthologous genes is known to mark hair follicle epithelial stem cells in mice, suggesting that regenerative systems in other epithelial appendages may utilize a related epithelial progenitor cell type, despite the highly derived nature of the resulting functional organs.

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“…In zebrafish, cells expressing TRAP are small in size and mononuclear, at least in early development, and therefore not easily revealed on conventionally stained sections. We have successfully employed GMA embedding and TRAP staining on sections of both larval and adult zebrafish (Witten et al, 2001; Kague et al, 2018). The reaction product can be visualized both in bright field (Fig.…”

Section: Resultsmentioning

confidence: 99%

Beyond the whole-mount phenotype: high-resolution imaging in fluorescence-based applications on zebrafish

et al. 2019

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Zebrafish is now widely used in biomedical research as a model for human diseases, but the relevance of the model depends on a rigorous analysis of the phenotypes obtained. Many zebrafish disease models, experimental techniques and manipulations take advantage of fluorescent reporter molecules. However, phenotypic analysis often does not go beyond establishing overall distribution patterns of the fluorophore in whole-mount embryos or using vibratome or paraffin sections with poor preservation of tissue architecture and limited resolution. Obtaining high-resolution data of fluorescent signals at the cellular level from internal structures mostly depends on the availability of expensive imaging technology. Here, we propose a new and easily applicable protocol for embedding and sectioning of zebrafish embryos using in-house prepared glycol methacrylate (GMA) plastic that is suited for preservation of fluorescent signals (including photoactivatable fluorophores) without the need for antibodies. Four main approaches are described, all involving imaging fluorescent signals on semithin (3 µm or less) sections. These include sectioning transgenic animals, whole-mount immunostained embryos, cell tracking, as well as on-section enzyme histochemistry.

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Zebrafish sp7 mutants show tooth cycling independent of attachment, eruption and poor differentiation of teeth

Cited by 26 publications

References 36 publications

ClC-7 Regulates the Pattern and Early Development of Craniofacial Bone and Tooth

ClC-7 Regulates the Pattern and Early Development of Craniofacial Bone and Tooth

Distinct tooth regeneration systems deploy a conserved battery of genes

Beyond the whole-mount phenotype: high-resolution imaging in fluorescence-based applications on zebrafish

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