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180 results on '"Mochida, Yoshiyuki"'

2. Small Leucine-Rich Proteoglycans (SLRPs) and Biomineralization

Author

Mochida, Yoshiyuki, Miguez, Patricia, Yamauchi, Mitsuo, Karamanos, Nikos K., Series Editor, Kletsas, Dimitris, Editorial Board Member, Oh, Eok-Soo, Editorial Board Member, Passi, Alberto, Editorial Board Member, Pihlajaniemi, Taina, Editorial Board Member, Ricard-Blum, Sylvie, Editorial Board Member, Sagi, Irit, Editorial Board Member, Savani, Rashmin, Editorial Board Member, Watanabe, Hideto, Editorial Board Member, Goldberg, Michel, editor, and Den Besten, Pamela, editor

Published
2021
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4. Elevated Fibroblast Growth Factor Signaling Is Critical for the Pathogenesis of the Dwarfism in Evc2/Limbin Mutant Mice.

Author

Zhang, Honghao, Kamiya, Nobuhiro, Tsuji, Takehito, Takeda, Haruko, Scott, Greg, Rajderkar, Sudha, Ray, Manas K, Mochida, Yoshiyuki, Allen, Benjamin, Lefebvre, Veronique, Hung, Irene H, Ornitz, David M, Kunieda, Tetsuo, and Mishina, Yuji

Subjects
Growth Plate, Animals, Humans, Mice, Dwarfism, Ellis-Van Creveld Syndrome, Polydactyly, Tooth Abnormalities, Disease Models, Animal, Fibroblast Growth Factors, Membrane Proteins, Signal Transduction, Mutant Proteins, Developmental Biology, Genetics
Abstract

Ellis-van Creveld (EvC) syndrome is a skeletal dysplasia, characterized by short limbs, postaxial polydactyly, and dental abnormalities. EvC syndrome is also categorized as a ciliopathy because of ciliary localization of proteins encoded by the two causative genes, EVC and EVC2 (aka LIMBIN). While recent studies demonstrated important roles for EVC/EVC2 in Hedgehog signaling, there is still little known about the pathophysiological mechanisms underlying the skeletal dysplasia features of EvC patients, and in particular why limb development is affected, but not other aspects of organogenesis that also require Hedgehog signaling. In this report, we comprehensively analyze limb skeletogenesis in Evc2 mutant mice and in cell and tissue cultures derived from these mice. Both in vivo and in vitro data demonstrate elevated Fibroblast Growth Factor (FGF) signaling in Evc2 mutant growth plates, in addition to compromised but not abrogated Hedgehog-PTHrP feedback loop. Elevation of FGF signaling, mainly due to increased Fgf18 expression upon inactivation of Evc2 in the perichondrium, critically contributes to the pathogenesis of limb dwarfism. The limb dwarfism phenotype is partially rescued by inactivation of one allele of Fgf18 in the Evc2 mutant mice. Taken together, our data uncover a novel pathogenic mechanism to understand limb dwarfism in patients with Ellis-van Creveld syndrome.

Published
2016

11. Lysine hydroxylation of collagen in a fibroblast cell culture system

Author

Uzawa, Katsuhiro, Yeowell, Heather N, Yamamoto, Kazushi, Mochida, Yoshiyuki, Tanzawa, Hideki, and Yamauchi, Mitsuo

Subjects
Life Sciences (General)
Abstract

The lysine (Lys) hydroxylation pattern of type I collagen produced by human fibroblasts in culture was analyzed and compared. Fibroblasts were cultured from normal human skin (NSF), keloid (KDF), fetal skin (FDF), and skin tissues of Ehlers-Danlos syndrome type VIA and VIB patients (EDS-VIA and -VIB). The type I collagen alpha chains with or without non-helical telopeptides were purified from the insoluble matrix and analyzed. In comparison with NSFs, KDF and FDF showed significantly higher Lys hydroxylation, particularly in the telopeptide domains of both alpha chains. Both EDS-VIA and -VIB showed markedly lower Lys hydroxylation in the helical domains of both alpha chains whereas that in the telopeptides was comparable with those of NSFs. A similar profile was observed in the tissue sample of the EDS-VIB patient. These results demonstrate that the Lys hydroxylation pattern is domain-specific within the collagen molecule and that this method is useful to characterize the cell phenotypes in normal/pathological connective tissues.

Published
2003
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12. Decorin modulates matrix mineralization in vitro

Author

Mochida, Yoshiyuki, Duarte, Wagner R, Tanzawa, Hideki, Paschalis, Eleftherios P, and Yamauchi, Mitsuo

Subjects
Life Sciences (General)
Abstract

Decorin (DCN), a member of small leucine-rich proteoglycans, is known to modulate collagen fibrillogenesis. In order to investigate the potential roles of DCN in collagen matrix mineralization, several stable osteoblastic cell clones expressing higher (sense-DCN, S-DCN) and lower (antisense-DCN, As-DCN) levels of DCN were generated and the mineralized nodules formed by these clones were characterized. In comparison with control cells, the onset of mineralization by S-DCN clones was significantly delayed; whereas it was markedly accelerated and the number of mineralized nodules was significantly increased in As-DCN clones. The timing of mineralization was inversely correlated with the level of DCN synthesis. In these clones, the patterns of cell proliferation and differentiation appeared unaffected. These results suggest that DCN may act as an inhibitor of collagen matrix mineralization, thus modulating the timing of matrix mineralization.

Published
2003
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14. Ellis van Creveld2 is required for postnatal craniofacial bone development

Author

Badri, Mohammed K., Zhang, Honghao, Ohyama, Yoshio, Venkitapathi, Sundharamani, Kamiya, Nobuhiro, Takeda, Haruko, Ray, Manas, Scott, Greg, Tsuji, Takehito, Kunieda, Tetsuo, Mishina, Yuji, and Mochida, Yoshiyuki

Subjects
Mice, Knockout, Heterozygote, Bone Development, Ellis-Van Creveld Syndrome, Homozygote, Membrane Proteins, Article, Bone and Bones, Facial Bones, Craniofacial Abnormalities, Mice, Inbred C57BL, Mice, Phenotype, Animals, Newborn, Animals, Intercellular Signaling Peptides and Proteins, Female
Abstract

Ellis-van Creveld (EvC) syndrome is a genetic disorder with mutations in either EVC or EVC2 gene. Previous case studies reported that EvC patients underwent orthodontic treatment, suggesting the presence of craniofacial bone phenotypes. To investigate whether a mutation in EVC2 gene causes a craniofacial bone phenotype, Evc2 knockout (KO) mice were generated and cephalometric analysis was performed. The heads of wild type (WT), heterozygous (Het) and homozygous Evc2 KO mice (1-, 3-, and 6-week-old) were prepared and cephalometric analysis based on the selected reference points on lateral X-ray radiographs was performed. The linear and angular bone measurements were then calculated, compared between WT, Het and KO and statistically analyzed at each time point. Our data showed that length of craniofacial bones in KO was significantly lowered by ∼20% to that of WT and Het, the growth of certain bones, including nasal bone, palatal length, and premaxilla was more affected in KO, and the reduction in these bone length was more significantly enhanced at later postnatal time points (3 and 6 weeks) than early time point (1 week). Furthermore, bone-to-bone relationship to cranial base and cranial vault in KO was remarkably changed, i.e. cranial vault and nasal bone were depressed and premaxilla and mandible were developed in a more ventral direction. Our study was the first to show the cause-effect relationship between Evc2 deficiency and craniofacial defects in EvC syndrome, demonstrating that Evc2 is required for craniofacial bone development and its deficiency leads to specific facial bone growth defect. Anat Rec, 299:1110-1120, 2016. © 2016 Wiley Periodicals, Inc.

Published
2016

18. Generation of Evc2/Limbin global and conditional KO mice and its roles during mineralized tissue formation

Author

Zhang, Honghao, Takeda, Haruko, Tsuji, Takehito, Kamiya, Nobuhiro, Rajderkar, Sudha, Louie, Ke’ Ale, Collier, Crystal, Scott, Greg, Ray, Manas, Mochida, Yoshiyuki, Kaartinen, Vesa, Kunieda, Tetsuo, and Mishina, Yuji

Subjects
Article
Abstract

Ellis-van Creveld (EvC) syndrome (OMIM 225500) is an autosomal recessive disease characterized with chondrodysplastic dwarfism in association with abnormalities in oral cavity. Ciliary proteins EVC and EVC2 have been identified as causative genes and they play an important role on Hedgehog signal transduction. We have also identified a causative gene LIMBIN for bovine chondrodysplastic dwarfism (bcd) that is later identified as the bovine ortholog of EVC2. Here, we report generation of conventional and conditional mutant Evc2/Limbin alleles that mimics mutations found in EvC patients and bcd cattle. Resulted homozygous mice showed no ciliary localization of EVC2 and EVC and displayed reduced Hedgehog signaling activity in association with skeletal and oral defects similar to the EvC patients. Cartilage-specific disruption of Evc2/Limbin resulted in similar but milder skeletal defects, whereas osteoblast-specific disruption did not cause overt changes in skeletal system. Neural crest-specific disruption of Evc2/Limbin resulted in defective incisor growth similar to that seen in conventional knockouts; however, differentiation of amelobolasts was relatively normal in the conditional knockouts. These results showcased functions of EVC2/LIMBIN during formation of mineralized tissues. Availability of the conditional allele for this gene should facilitate further detailed analyses of the role of EVC2/LIMBIN in pathogenesis of EvC syndrome. genesis 53:612-626, 2015. © 2015 Wiley Periodicals, Inc.

Published
2015

19. Expression of Evc2 in craniofacial tissues and craniofacial bone defects in Evc2 knockout mouse

Author

Badri, Mohammed K., primary, Zhang, Honghao, additional, Ohyama, Yoshio, additional, Venkitapathi, Sundharamani, additional, Alamoudi, Ahmed, additional, Kamiya, Nobuhiro, additional, Takeda, Haruko, additional, Ray, Manas, additional, Scott, Greg, additional, Tsuji, Takehito, additional, Kunieda, Tetsuo, additional, Mishina, Yuji, additional, and Mochida, Yoshiyuki, additional

Published
2016
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21. FAM20A binds to and regulates FAM20C localization

Author

Ohyama, Yoshio, primary, Lin, Ju-Hsien, additional, Govitvattana, Nattanan, additional, Lin, I-Ping, additional, Venkitapathi, Sundharamani, additional, Alamoudi, Ahmed, additional, Husein, Dina, additional, An, Chunying, additional, Hotta, Hak, additional, Kaku, Masaru, additional, and Mochida, Yoshiyuki, additional

Published
2016
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22. Ellis Van Creveld2 is Required for Postnatal Craniofacial Bone Development

Author

Badri, Mohammed K., primary, Zhang, Honghao, additional, Ohyama, Yoshio, additional, Venkitapathi, Sundharamani, additional, Kamiya, Nobuhiro, additional, Takeda, Haruko, additional, Ray, Manas, additional, Scott, Greg, additional, Tsuji, Takehito, additional, Kunieda, Tetsuo, additional, Mishina, Yuji, additional, and Mochida, Yoshiyuki, additional

Published
2016
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24. Identification and characterization of neural crest-derived cells in adult periodontal ligament of mice

Author

Mishina, Yuji, Mochida, Yoshiyuki, Komatsu, Yoshihiro, Yamauchi, Mitsuo, Kaku, Masaru, and Ko, Ching-Chang

Subjects
animal structures, stomatognathic system, embryonic structures
Abstract

Cells derived from the neural crest (NC) contribute to the development of several adult tissues, including tooth and periodontal tissue. Here, two transgenic lines, Wnt1-Cre/ZEG and P0-Cre/ZEGwere analyzed to determine the fate and distribution of neural crest cells (NCCs) in adult mouse periodontal ligament (PDL).

Published
2012
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25. Generation ofEvc2/Limbinglobal and conditional KO mice and its roles during mineralized tissue formation

Author

Zhang, Honghao, primary, Takeda, Haruko, additional, Tsuji, Takehito, additional, Kamiya, Nobuhiro, additional, Rajderkar, Sudha, additional, Louie, Ke'Ale, additional, Collier, Crystal, additional, Scott, Greg, additional, Ray, Manas, additional, Mochida, Yoshiyuki, additional, Kaartinen, Vesa, additional, Kunieda, Tetsuo, additional, and Mishina, Yuji, additional

Published
2015
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26. Lysyl Oxidase Binds Transforming Growth Factor-β and Regulates Its Signaling via Amine Oxidase Activity

Author

Katafuchi, Michitsuna, Yamauchi, Mitsuo, Csiszar, Katalin, Mochida, Yoshiyuki, Fong, Keith S.K., Atsawasuwan, Phimon, and Kaku, Masaru

Subjects
enzymes and coenzymes (carbohydrates), integumentary system
Abstract

Lysyl oxidase (LOX), an amine oxidase critical for the initiation of collagen and elastin cross-linking, has recently been shown to regulate cellular activities possibly by modulating the functions of growth factors. In this study, we investigated the interaction between LOX and transforming growth factor-β1 (TGF-β1), a potent growth factor abundant in bone, the effect of LOX on TGF-β1 signaling, and its potential mechanism. The specific binding between mature LOX and mature TGF-β1 was demonstrated by immunoprecipitation and glutathione S-transferase pulldown assay in vitro. Both proteins were colocalized in the extracellular matrix in an osteoblastic cell culture system, and the binding complex was identified in the mineral-associated fraction of bone matrix. Furthermore, LOX suppressed TGF-β1-induced Smad3 phosphorylation likely through its amine oxidase activity. The data indicate that LOX binds to mature TGF-β1 and enzymatically regulates its signaling in bone and thus may play an important role in bone maintenance and remodeling.

Published
2008
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