The molecular complex of ciliary and golgin protein is critical for skull development

Yamaguchi, Hiroyuki; Meyer, Matthew D; He, Li; Senavirathna, Lakmini; Pan, Sheng; Komatsu, Yoshihiro

doi:10.1242/dev.199559

Cited by 12 publications

(21 citation statements)

References 55 publications

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“…We have recently reported that proliferation and differentiation activities in CNC-derived osteoblasts are normal in Trip11 cKO mice. 11 This suggests that the initial process of osteogenesis may not be affected by deletion of Trip11. However, it remains unclear whether disruption of Trip11 affects cell survival and thus causes craniofacial skeletal defects.…”

Section: Loss Of Trip11 Increases Apoptosis In Skull Tissuesmentioning

confidence: 98%

“…As we have recently reported, GMAP210 is critical for regulating collagen trafficking in the area of the osteogenic front (OF). 11 In addition to this unique function, GMAP210 is also required for osteoblast cell survival via orchestrating both ER and Golgi stress. Therefore, loss of Trip11 in CNC-derived osteoblasts may result in both less collagen secretion and increased cell death, leading to skull defects in mice (Figure 8).…”

Section: Loss Of Trip11 Is Associated With Aberrant Golgi Stress Duri...mentioning

confidence: 99%

“…Staining of craniofacial tissues with Alizarin red and Alcian blue was carried out as previously described. 11,32,33 Cephalometric analysis was performed using the lateral and dorsoventral pictures after skeletal staining. Definitions of cephalometric landmarks and measurements are described in Tables 1, 2.…”

Section: Skeletal Preparations Histological Analysis and Fluorescence...mentioning

confidence: 99%

“…Immunocytochemistry was performed as previously described. 11,36 The primary antibody GM130 (1:500, BD Biosciences, 610 822) was used. Plot profile was performed 30 to measure the size of cis-Golgi using ImageJ software.…”

Section: Isolation Of Primary Osteoblast and Immunocytochemistrymentioning

confidence: 99%

“…11 We found that the molecular complex of ciliary protein intraflagellar transport 20 (IFT20) and GMAP210 regulates skull development. 11 Importantly, the molecular complex of IFT20-GMAP210 plays a pivotal role in trafficking of type I collagen. Thus, this cellular mechanism is required for smooth collagen trafficking and secreting adequate amounts of collagen matrix essential for skull maturation.…”

Section: Introductionmentioning

confidence: 99%

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Disruption of Trip11 in cranial neural crest cells is associated with increased ER and Golgi stress contributing to skull defects in mice

Yamaguchi

Meyer

et al. 2022

Developmental Dynamics

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Background Absence of Golgi microtubule‐associated protein 210 (GMAP210), encoded by the TRIP11 gene, results in achondrogenesis. Although TRIP11 is thought to be specifically required for chondrogenesis, human fetuses with the mutation of TRIP11 also display bony skull defects where chondrocytes are usually not present. This raises an important question of how TRIP11 functions in bony skull development. Results We disrupted Trip11 in neural crest‐derived cell populations, which are critical for developing skull in mice. In Trip11 mutant skulls, expression levels of ER stress markers were increased compared to controls. Morphological analysis of electron microscopy data revealed swollen ER in Trip11 mutant skulls. Unexpectedly, we also found that Golgi stress increased in Trip11 mutant skulls, suggesting that both ER and Golgi stress‐induced cell death may lead to osteopenia‐like phenotypes in Trip11 mutant skulls. These data suggest that Trip11 plays pivotal roles in the regulation of ER and Golgi stress, which are critical for osteogenic cell survival. Conclusion We have recently reported that the molecular complex of ciliary protein and GMAP210 is required for collagen trafficking. In this paper, we further characterized the important role of Trip11 being possibly involved in the regulation of ER and Golgi stress during skull development.

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Section: Loss Of Trip11 Increases Apoptosis In Skull Tissuesmentioning

confidence: 98%

Section: Loss Of Trip11 Is Associated With Aberrant Golgi Stress Duri...mentioning

confidence: 99%