Tooth eruption: altered gene expression in the dental follicle of patients with cleidocranial dysplasia

Dorotheou, Domna; Gkantidis, Nikolaos; Karamolegkou, Marina; Kalyvas, Demos; Kiliaridis, Stavros; Kitraki, Efthimia

doi:10.1111/ocr.12000

Cited by 30 publications

(25 citation statements)

References 25 publications

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“…These results indicate that DFCs RUNX2+/m can impair osteoclastogenesis in the local region during tooth eruption. Other studies had also reported that RUNX2 mutation could markedly restrict the ability of DFCs and PDLCs to induce osteoclasts differentiation, which was consistent with the results in the current study (Dorotheou et al., ; Ge et al., ; Lossdorfer et al., ).…”

Section: Discussionsupporting

confidence: 93%

Abnormal bone remodelling activity of dental follicle cells from a cleidocranial dysplasia patient

et al. 2018

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Section: Discussionsupporting

confidence: 93%

Abnormal bone remodelling activity of dental follicle cells from a cleidocranial dysplasia patient

et al. 2018

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“…More recent studies of dental follicle cells (DFC) from patients with CCD have reported disturbed osteoclast‐inductive signalling in DFCs, which the authors suggest could be responsible for delayed tooth eruption in patients with CCD . Sun et al .…”

Section: Cleidocranial Dysplasiamentioning

confidence: 99%

“…LOSS-D € ORFER et al (66) carried out an in vitro study on human periodontal ligament cells from two patients with CCD and found that these cells showed a reduced capacity to induce the differentiation of active osteoclasts. More recent studies of dental follicle cells (DFC) from patients with CCD have reported disturbed osteoclast-inductive signalling in DFCs, which the authors suggest could be responsible for delayed tooth eruption in patients with CCD (67)(68)(69). SUN et al (70) isolated and cultured DFCs and periodontal ligament cells (PDLCs) from an 11-yr-old individual with CCD and concluded that the RUNX2 mutation disturbs the modulatory effects of both DFCs and PDLCs on the differentiation of osteoclasts and osteoblasts, thereby interfering with bone remodelling.…”

Section: Animal Models and Molecular Studiesmentioning

confidence: 99%

Tooth formation and eruption – lessons learnt from cleidocranial dysplasia

Kreiborg

Jensen

2018

European J Oral Sciences

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The principles of formation, renewal, and eruption of teeth are discussed. Numerous genetic aberrations may affect the formation and eruption of teeth. Cleidocranial dysplasia (CCD), caused by mutations in the runt-related transcription factor 2 (RUNX2) gene, is such a condition. The dental phenotype includes problems in both tooth formation (multiple supernumerary permanent teeth) and tooth eruption (lack of shedding of primary teeth and delayed or arrested eruption of permanent teeth). Clinical studies, animal models, and molecular biology studies have documented that RUNX2 is of paramount importance for osteoblast differentiation, for regression of the dental lamina, and for osteoclastogenesis in the dental follicle and the periodontal ligament. Jensen & Kreiborg, 25 yr ago, proposed a treatment strategy to be applied to patients with CCD, focussing on the importance of early treatment to promote spontaneous eruption of permanent teeth through extraction of primary teeth, surgical removal of supernumerary teeth, and removal of bone covering the first formed permanent teeth at the time when root formation of the permanent teeth has reached half or two-thirds of their final length. This strategy still seems valid and seems to lead to reduction in the burden of care for patients compared with the treatment protocols otherwise recommended.

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“…Cleidocranial dysplasia (CCD), which is caused by mutation of Runx2, is a hereditary congenital disorder characterized by the abnormalities of bone development such as clavicle dysplasia, unclosed fontanels, and retained primary teeth due to no root resorption [11][12][13][14][15][16]. Previous studies reported that Runx2, which is the master osteogenic transgenic factor regulating osteogenesis metabolism, also induces differentiation of osteoclast through regulation of RANKL and OPG [17][18][19][20][21].…”

mentioning

confidence: 99%

Periodontal Ligament Stem Cells Modulate Root Resorption of Human Primary Teeth via Runx2 Regulating RANKL/OPG System

Wang

Dong

et al. 2014

Stem Cells and Development

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Physiological primary teeth exfoliation is a normal phenomenon during teeth development. However, retained primary teeth can often be observed in the patients with cleidocranial dysplasia (CCD) caused by mutation of Runx2. The potential regulative mechanism is still unknown. In the present study, periodontal ligament stem cells (PDLSCs) were derived from different resorbed stages of primary teeth and permanent teeth from normal patients and primary teeth from CCD patient. The proliferative, osteogenic and osteoclast-inductive capacities of PDLSCs from each group were detected. We demonstrated here that the proliferative ability of PDLSCs was reduced while the osteogenic and the osteoclast-inductive capacity of PDLSCs were enhanced during root resorption. The results also showed that PDLSCs from permanent teeth and CCD patient expressed low level of Runx2 and RANKL while high level of OPG. However, expression of Runx2 and RANKL were increased while expression of OPG was decreased in PDLSCs derived from resorbed teeth. Furthermore, Runx2 regulating the expression of RANKL and OPG and the osteoclast-inductive capacity of PDLSCs were confirmed by gain or loss of function assay. These data suggest that PDLSCs promote osteoclast differentiation via Runx2 upregulating RANKL and downregulating OPG, leading to enhanced root resorption that results in physiological exfoliation of primary teeth.

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Tooth eruption: altered gene expression in the dental follicle of patients with cleidocranial dysplasia

Abstract: The observed alterations in the expression and ratios of the aforementioned factors in the dental follicle of CCD individuals suggest a disturbed paracrine signaling for bone remodeling that could be responsible for the impaired tooth eruption seen in these patients.

Cited by 30 publications

References 25 publications

Abnormal bone remodelling activity of dental follicle cells from a cleidocranial dysplasia patient

Abnormal bone remodelling activity of dental follicle cells from a cleidocranial dysplasia patient

Tooth formation and eruption – lessons learnt from cleidocranial dysplasia

Periodontal Ligament Stem Cells Modulate Root Resorption of Human Primary Teeth via Runx2 Regulating RANKL/OPG System

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