Genomic deletion of a long-range bone enhancer misregulates sclerostin in Van Buchem disease

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Figure 1.
Figure 1.

Generation and characterization of Van Buchem transgenic mouse models. (A) A 158-kb human BAC (SOSTwt) spanning SOST and MEOX1 was engineered using in vitro BAC recombination in Escherichia coli (Lee et al. 2001) by deleting the 52-kb noncoding region missing in VB patients (SOSTvbΔ). Human SOST expression was analyzed by rtPCR in adult tissues (B), embryos (C), and measured by quantitative rtPCR in E10.5 embryos (D) from two independent lines of each SOSTwt and SOSTvbΔ transgene.

This Article

  1. Published in Advance June 17, 2005, doi: 10.1101/gr.3437105 Genome Res. 15: 928-935

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