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- W1970422746 abstract "The double mutant utr-/-/mdx mouse has been postulated to be a better model of Duchenne Muscular Dystrophy than mdx mouse because it displays a progression of pathological features comparable to that in humans. We previously demonstrated that FDB fibers from mdx mice show limitations in action potential (AP) elicited Ca2+ release. Here we investigated the properties of the APs and the ability to release Ca2+ (30 mM [EGTA], 20°C) in response to single and trains of APs (20 pulses, at 33Hz and 100Hz) in FDB fibers isolated from control, mdx and utr-/-/mdx mice. Single APs of normal amplitude but longer duration were recorded in utr-/-/mdx fibers, whereas the amplitude of the Ca2+ release was ∼37% smaller than in normal fibers, but comparable with that found in mdx fibers. Fibers from the three strains sustained trains of Ca release at 33 Hz in which the amplitude of individual Ca2+ release transients decayed exponentially towards a sustained release amplitude with two time constants (τ1=10ms, τ2=200ms). In response to 100Hz trains, the amplitude of Ca2+ release in normal fibers decayed still with a double exponential (τ1=3.5ms, τ2=41ms) in which the amplitude of the 2nd and 20th transient along the train were ∼50% and ∼35% that of the first one, respectively. In contrast, fibers isolated from both mdx and utr-/-/mdx mice could be divided in two groups according to their tetanic response to 100Hz trains: approximately 75% of mdx and 33% of utr-/-/mdx fibers showed a behavior similar to that observed in normal fibers; in the remaining 25% of mdx and 67% of utr-/-/mdx fibers, respectively, the amplitude of the 2nd Ca2+ release transient was ∼20% of the first one, and this amplitude was sustained throughout the train." @default.
- W1970422746 created "2016-06-24" @default.
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- W1970422746 date "2009-02-01" @default.
- W1970422746 modified "2023-09-28" @default.
- W1970422746 title "The Absence of Utrophin Does Not Further the Impairment of Ca2+ Release Displayed by mdx Muscle" @default.
- W1970422746 doi "https://doi.org/10.1016/j.bpj.2008.12.1159" @default.
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