Fig. 7
- ID
- ZDB-FIG-170320-8
- Publication
- Ramspacher et al., 2015 - Developmental Alterations in Heart Biomechanics and Skeletal Muscle Function in Desmin Mutants Suggest an Early Pathological Root for Desminopathies
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Pharmacological Targeting of Desma Aggregates Rescue ct122aGt Phenotype (A) Confocal and electron microscopy images showing the impact of Doxycycline (Doxy) treatment on ct122aGt muscle. Doxy-treated embryos showed more condensed and smaller aggregates compared to the controls. Scale bar, 20 μm (SHG). Scale bar, 100 nm (TEM) (B) Quantification of aggregate size and number in Doxy-treated embryos compared to the corresponding vehicle-treated embryos. (C) Evaluation of the phenotypes of Doxy and vehicle-treated embryos using SHG and the scale defined in (C) show that only weak and normal phenotypes were observed after Doxy treatment, while strong phenotypes are present in the controls. Note that the vehicle contains ethanol, which is leading to a stronger phenotype than in the non-vehicle-treated ct122aGt line. (D) Touch-evoke response assays showing that Doxy treatment improves swimming distance but not speed in ct122aGt. (E) Doxy-treated ct122aGt showed increased fractional shortening (FS) in the atrium but could re-establish FS in the ventricle. (F) Doxy treatment increases desma solubility (index of solubility) in dose-dependent manner. Actin is the loading control. ct122aRGt indicates desmact122aRGt homozygous embryos. The error bars correspond to the SEM. |
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| Stage Range: | Long-pec to Day 4 |