Runx2-deficient mice lack mandibular condylar cartilage and have deformed Meckel?s cartilage

Shibata, Shunichi; Suda, Naoto; Yoda, S.; Fukuoka, Hiroki; Ohyama, Kimie; Yamashita, Yasuo; Komori, Toshihisa

doi:10.1007/s00429-004-0393-2

Cited by 52 publications

(55 citation statements)

References 26 publications

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“…Furthermore, same genes may exert different function in the development of primary and secondary cartilage. For example, in Runx2-deficient mice, primary cartilage forms, but the condylar cartilage is completely eliminated (Ducy et al, 1997;Komori et al, 1997;Otto et al, 1997;Shibata et al, 2004).…”

Section: Discussionmentioning

confidence: 97%

Shox2-deficiency leads to dysplasia and ankylosis of the temporomandibular joint in mice

Wei

et al. 2008

Mechanisms of Development

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The temporomandibular joint (TMJ) is a unique synovial joint whose development differs from the formation of other synovial joints. Mutations have been associated with the developmental defects of the TMJ only in a few genes. In this study, we report the expression of the homeobox gene Shox2 in the cranial neural crest derived mesenchymal cells of the maxilla-mandibular junction and later in the progenitor cells and undifferentiated chondrocytes of the condyle as well as the glenoid fossa of the developing TMJ. A conditional inactivation of Shox2 in the cranial neural crest-derived cells causes developmental abnormalities in the TMJ, including dysplasia of the condyle and glenoid fossa. The articulating disc forms but fuses with the fibrous layers of the condyle and glenoid fossa, clinically known as TMJ ankylosis. Histological examination indicates a delay in development in the mutant TMJ, accompanied by a significantly reduced rate of cell proliferation. In situ hybridization further demonstrates an altered expression of several key osteogenic genes and a delayed expression of the osteogenic differentiation markers. Shox2 appears to regulate the expression of osteogenic genes and is essential for the development and function of the TMJ. The Shox2 conditional mutant thus provides a unique animal model of TMJ ankylosis.

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Section: Discussionmentioning

confidence: 97%

Shox2-deficiency leads to dysplasia and ankylosis of the temporomandibular joint in mice

Wei

et al. 2008

Mechanisms of Development

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“…The RUNX transcription factors modulate the maturation of cartilage and muscle cells (59,69). Of note, increased airway wall size is found in children exposed to prenatal maternal smoking (17).…”

Section: Discussionmentioning

confidence: 99%

RUNX transcription factors: association with pediatric asthma and modulated by maternal smoking

Haley

Lasky‐Su²,

Manoli

et al. 2011

American Journal of Physiology-Lung Cellular and Molecular Phys

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Intrauterine smoke exposure (IUS) is a strong risk factor for development of airways responsiveness and asthma in childhood. Runt-related transcription factors (RUNX1-3) have critical roles in immune system development and function. We hypothesized that genetic variations in RUNX1 would be associated with airway responsiveness in asthmatic children and that this association would be modified by IUS. Family-based association testing analysis in the Childhood Asthma Management Program genome-wide genotype data showed that 17 of 100 RUNX1 single-nucleotide polymorphisms (SNPs) were significantly (P < 0.03-0.04) associated with methacholine responsiveness. The association between methacholine responsiveness and one of the SNPs was significantly modified by a history of IUS exposure. Quantitative PCR analysis of immature human lung tissue with and without IUS suggested that IUS increased RUNX1 expression at the pseudoglandular stage of lung development. We examined these associations by subjecting murine neonatal lung tissue with and without IUS to quantitative PCR (N = 4-14 per group). Our murine model showed that IUS decreased RUNX expression at postnatal days (P)3 and P5 (P < 0.05). We conclude that 1) SNPs in RUNX1 are associated with airway responsiveness in asthmatic children and these associations are modified by IUS exposure, 2) IUS tended to increase the expression of RUNX1 in early human development, and 3) a murine IUS model showed that the effects of developmental cigarette smoke exposure persisted for at least 2 wk after birth. We speculate that IUS exposure-altered expression of RUNX transcription factors increases the risk of asthma in children with IUS exposure.

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“…Runx2 knockout mice do not form the condylar cartilage, which suggests a role for RUNX2 protein in its differentiation (Shibata et al, 2004(Shibata et al, , 2006. This cartilage may be derived either from the periosteum of the dentary or from a sesamoid that is independent from the rest of the developing jaw (reviewed in Hall, 2005, p 155-159).…”

Section: The Evolution Of Cartilage Differentiationmentioning

confidence: 99%

Runx2 is essential for larval hyobranchial cartilage formation in Xenopus laevis

Kerney

Gross

Howard

2007

Developmental Dynamics

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The vertebrate transcription factor protein Runx2 is regarded as a "master regulator" of bone formation due to the dramatic loss of the osseous skeleton in the mouse homozygous knockout. However, Runx2 mRNA also is expressed in the pre-hypertrophic cartilaginous skeleton of the mouse and chicken, where its developmental function is largely unknown. Several tiers of Runx2 regulation exist in the mouse, any of which may account for its seeming biological inactivity during early stages of skeletogenesis. Unlike mouse and chicken, zebrafish require Runx2 function in early cartilage differentiation. The present study reveals that the earlier functional role of Runx2 in cartilage differentiation is shared between zebrafish and Xenopus. A combination of morpholino oligonucleotide injections and neural crest transplants indicate that Runx2 is involved in differentiation of the cartilaginous hyobranchial skeleton in the frog, Xenopus laevis. Additionally, in situ hybridizations show runx2 mRNA expression in mesenchymal precursors of the cartilaginous skull, which reveals the earliest pre-patterning of these cartilages described to date. The early distribution of runx2 resolves the homology of the larval suprarostral plate, which is one of the oldest controversies of anuran skull development. Together these data reveal a shift in Runx2 protein function during vertebrate evolution towards its exclusive roles in cartilage hypertrophy and bone differentiation within the amniote lineage. Developmental Dynamics 236:1650 -1662, 2007.

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Runx2-deficient mice lack mandibular condylar cartilage and have deformed Meckel?s cartilage

Cited by 52 publications

References 26 publications

Shox2-deficiency leads to dysplasia and ankylosis of the temporomandibular joint in mice

Shox2-deficiency leads to dysplasia and ankylosis of the temporomandibular joint in mice

RUNX transcription factors: association with pediatric asthma and modulated by maternal smoking

Runx2 is essential for larval hyobranchial cartilage formation in Xenopus laevis

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