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    1. Silencing Of The Drosophila Ortholog Of SOX5 In Heart Leads To Cardiac Dysfunction As Detected By Optical Coherence Tomography

      Silencing Of The Drosophila Ortholog Of SOX5 In Heart Leads To Cardiac Dysfunction As Detected By Optical Coherence Tomography

      The SRY-related HMG-box 5 (SOX5) gene encodes a member of the SOX family of transcription factors. Recently, genome-wide association studies have implicated SOX5 as a candidate gene for susceptibility to four cardiac-related endophenotypes: higher resting heart rate (RHR), the electrocardiographic PR interval, atrial fibrillation (AF) and left ventricular mass (LVM). We have determined that human SOX5 has a highly conserved Drosophila ortholog, Sox102F, and have employed transgenic Drosophila models to quantitatively measure cardiac function in adult flies. For this purpose, we have developed a high-speed and ultrahigh-resolution optical coherence tomography (OCT) imaging system, which enables rapid cross-sectional imaging of the ...

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    2. SMAD signaling drives heart and muscle dysfunction in a Drosophila model of muscular dystrophy

      SMAD signaling drives heart and muscle dysfunction in a Drosophila model of muscular dystrophy
      Loss-of-function mutations in the genes encoding dystrophin and the associated membrane proteins, the sarcoglycans, produce muscular dystrophy and cardiomyopathy. The dystrophin complex provides stability to the plasma membrane of striated muscle during muscle contraction. Increased SMAD signaling due to activation of the transforming growth factor-β (TGFβ) pathway has been described in muscular dystrophy; however, it is not known whether this canonical TGFβ signaling is pathogenic in the muscle itself. Drosophila deleted for the γ/δ-sarcoglycan gene (Sgcd) develop progressive muscle and heart dysfunction and serve as a model for the human disorder. We used dad-lacZ flies to demonstrate the signature ...
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