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Respiratory defects in the CrtapKO mouse model of Osteogenesis Imperfecta

Am J Physiol Lung Cell Mol Physiol. 2020; 
Dimori M, Heard-Lipsmeyer ME, Byrum SD, Mackintosh SG, Kurten RC, Carroll JL, Morello R.
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Recombinant Proteins … antibody 181 diluted in TBS-T overnight at 4 °C, followed by washing in TBS-T Primary antibodies were 182 Crtap (1:1000) [5] and β-actin (cat# A00702, GenScript, Piscataway, NJ, USA) Secondary 183 antibodies were goat … Get A Quote

摘要

Respiratory disease is a leading cause of mortality in patients with Osteogenesis imperfecta (OI), a connective tissue disease that causes severely reduced bone mass and is most commonly caused by dominant mutations in type I collagen genes. Previous studies proposed that impaired respiratory function in OI patients was secondary to skeletal deformities however recent evidence suggests the existence of a primary lung defect. Here, we analyzed the lung phenotype of CrtapKO mice, a mouse model of recessive OI. While we confirm changes in the lung parenchyma that are reminiscent of emphysema, we show that CrtapKO lung fibroblasts synthesize type I collagen with altered post-translation modifications consistent wit... More

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