Fgfr1 conditional-knockout in neural crest cells induces heterotopic chondrogenesis and osteogenesis in mouse frontal bones

Kawai, Mariko; Herrmann, David; Fuchs, Alisa; Cheng, Shuofei; Ferrer-Vaquer, Anna; Götz, Rebekka; Driller, Katrin; Neubüser, Annette; Ohura, Kiyoshi

doi:10.1007/s00795-018-0213-z

Cited by 5 publications

(3 citation statements)

References 26 publications

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“…Conditional inactivation of Fgfr1 in the neural crest lineage in mice resulted in severe midfacial clefting including cleft lip and palate and caused heterotopic chondrogenesis and osteogenesis of the anterior frontal bones (Kawai et al, 2019; Wang et al, 2013). Conditional inactivation of Fgfr1 and Fgfr2 in palate mesenchyme, at later stages of development, impaired palatal shelf elevation and resulted in cleft palate.…”

Section: Selected Topics In Genetics Development Regeneration and Dis...mentioning

confidence: 99%

New developments in the biology of fibroblast growth factors

Ornitz

Itoh

2022

WIREs Mechanisms of Disease

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The fibroblast growth factor (FGF) family is composed of 18 secreted signaling proteins consisting of canonical FGFs and endocrine FGFs that activate four receptor tyrosine kinases (FGFRs 1-4) and four intracellular proteins (intracellular FGFs or iFGFs) that primarily function to regulate the activity of voltagegated sodium channels and other molecules. The canonical FGFs, endocrine FGFs, and iFGFs have been reviewed extensively by us and others. In this review, we briefly summarize past reviews and then focus on new developments in the FGF field since our last review in 2015. Some of the highlights in the past 6 years include the use of optogenetic tools, viral vectors, and inducible transgenes to experimentally modulate FGF signaling, the clinical use of small molecule FGFR inhibitors, an expanded understanding of endocrine FGF signaling, functions for FGF signaling in stem cell pluripotency and differentiation, roles for FGF signaling in tissue homeostasis and regeneration, a continuing elaboration of mechanisms of FGF signaling in development, and an expanding appreciation of roles for FGF signaling in neuropsychiatric diseases.

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Section: Selected Topics In Genetics Development Regeneration and Dis...mentioning

confidence: 99%

New developments in the biology of fibroblast growth factors

Ornitz

Itoh

2022

WIREs Mechanisms of Disease

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“…Ras signaling mediates receptor tyrosine kinase (RTK) signaling, including FGF, PDGF and EGF signaling. FGFR1 and FGFR2 knockout mouse models demonstrated the crucial role of FGF signaling for cell proliferation in the growth of palatal shelves ( Hosokawa et al, 2009 ; Kawai et al, 2019 ). PDGFRA mutant mice developed cleft palate due to a defect of the palatal shelf growth associated with the reduced extracellular matrix production.…”

Section: Discussionmentioning

confidence: 99%

Ras signaling and RREB1 are required for the dissociation of medial edge epithelial cells in murine palatogenesis

Inubushi

Fujiwara

Hirose

et al. 2022

Disease Models &Amp; Mechanisms

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Cleft palate is one of the major congenital craniofacial birth defects. The etiology underlying the pathogenesis of cleft palate has largely remained unelucidated. Dissociation of the medial edge epithelium (MEE) at the contacting region of palatal shelves and subsequent migration or apoptosis of MEE cells is required for the proper MEE removal. Ras Responsive Element Binding Protein 1 (RREB1), a RAS transcriptional effector, has recently been shown to play a crucial role in developmental EMT, in which loss of epithelial characteristics is an initial step, during mid-gastrulation of embryonic development. Interestingly, the involvement of RREB1 in cleft palate has been indicated in humans. Here, we demonstrated that pan-Ras inhibitor prevents the dissociation of MEE during palatal fusion. Rreb1 is expressed in the palatal epithelium during palatal fusion, and knockdown of Rreb1 in palatal organ culture resulted in palatal fusion defects by inhibiting the dissociation of MEE cells. Our present findings provide evidence that RREB1-mediated Ras signaling is required during palatal fusion. Aberrant RREB1-mediated Ras signaling might be involved in the pathogenesis of cleft palate.

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“…This process appears to be recapitulated during HO. [ 289,290 ] Expression of osteoblast‐specific transcription factors was identified in cells derived from the neural endoneurium after HO induction. Those transcription factors reappeared at the site of new bone formation after transient expression in the circulation.…”

Section: Crosstalk Between Bone and Intrabony Nerves In Pathophysiolomentioning

confidence: 99%

Crosstalk between Bone and Nerves within Bone

et al. 2021

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For the past two decades, the function of intrabony nerves on bone has been a subject of intense research, while the function of bone on intrabony nerves is still hidden in the corner. In the present review, the possible crosstalk between bone and intrabony peripheral nerves will be comprehensively analyzed. Peripheral nerves participate in bone development and repair via a host of signals generated through the secretion of neurotransmitters, neuropeptides, axon guidance factors and neurotrophins, with additional contribution from nerve‐resident cells. In return, bone contributes to this microenvironmental rendezvous by housing the nerves within its internal milieu to provide mechanical support and a protective shelf. A large ensemble of chemical, mechanical, and electrical cues works in harmony with bone marrow stromal cells in the regulation of intrabony nerves. The crosstalk between bone and nerves is not limited to the physiological state, but also involved in various bone diseases including osteoporosis, osteoarthritis, heterotopic ossification, psychological stress‐related bone abnormalities, and bone related tumors. This crosstalk may be harnessed in the design of tissue engineering scaffolds for repair of bone defects or be targeted for treatment of diseases related to bone and peripheral nerves.

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Fgfr1 conditional-knockout in neural crest cells induces heterotopic chondrogenesis and osteogenesis in mouse frontal bones

Cited by 5 publications

References 26 publications

New developments in the biology of fibroblast growth factors

New developments in the biology of fibroblast growth factors

Ras signaling and RREB1 are required for the dissociation of medial edge epithelial cells in murine palatogenesis

Crosstalk between Bone and Nerves within Bone

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