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Dural cerebral veins (CV) are required for cerebrospinal fluid reabsorption and brain homeostasis, but mechanisms that regulate their growth and remodeling are unknown. We report molecular and cellular processes that regulate dural CV development in mammals and describe venous malformations in humans with craniosynostosis and TWIST1 mutations that are recapitulated in mouse models. Surprisingly, Twist1 is dispensable in endothelial cells but required for specification of osteoprogenitor cells that differentiate into preosteoblasts that produce bone morphogenetic proteins (BMPs). Inactivation of Bmp2 and Bmp4 in preosteoblasts and periosteal dura causes skull and CV malformations, similar to humans harboring TWIST1 mutations. Notably, arterial development appears normal, suggesting that morphogens from the skull and dura establish optimal venous networks independent from arterial influences. Collectively, our work establishes a paradigm whereby CV malformations result from primary or secondary loss of paracrine BMP signaling from preosteoblasts and dura, highlighting unique cellular interactions that influence tissue-specific angiogenesis in mammals.
Pubmed ID: 28844842 RIS Download
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Software that performs quantitative analysis of angiogenesis. It specializes in quantitative assessment of various vessel morphometric and spatial parameters, including vessel length and density, branching index, lacunarity, etc.
View all literature mentionsThis polyclonal secondary targets Mouse IgG (H+L)
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View all literature mentionsThis unknown targets IgG (H+L)
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View all literature mentionsThis unknown targets Rabbit IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis unknown targets IgG (H+L)
View all literature mentionsThis unknown targets IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis polyclonal secondary targets IgG (H+L)
View all literature mentionsThis unknown targets Rat IgG (H+L)
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View all literature mentionsThis polyclonal targets Mouse EphB4
View all literature mentionsThis polyclonal targets Cleaved Caspase-3 (Asp175)
View all literature mentionsThis monoclonal targets Actin alpha-Smooth Muscle - Cy3(TM) antibody produced in mouse
View all literature mentionsThis monoclonal targets 129/Sv mouse-derived endothelioma cell line tEnd.1
View all literature mentionsThis monoclonal targets Genetic locus: TWIST1 (human) mapping to 7p21.1; Twist1 (mouse) mapping to 12 A3. Raised against a recombinant protein corresponding to a region near the C-terminus of twist of human origin.
View all literature mentionsThis monoclonal targets CD102
View all literature mentionsThis polyclonal targets DsRed
View all literature mentionsThis polyclonal targets GFP (FITC)
View all literature mentionsThis monoclonal targets Mouse Endomucin
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View all literature mentionsOpen source Java based image processing software program designed for scientific multidimensional images. ImageJ has been transformed to ImageJ2 application to improve data engine to be sufficient to analyze modern datasets.
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View all literature mentionsMus musculus with name B6.129S4-Myf5tm3(cre)Sor/J from IMSR.
View all literature mentionsMus musculus with name STOCK Tg(Tagln-cre)1Her/J from IMSR.
View all literature mentionsMus musculus with name B6;129S7-Twist1tm2Bhr/Mmnc from MMRRC.
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