Key result
RyR-2 or JP-2 knockout causes early embryonic lethality by disrupting cardiomyocyte calcium homeostasis.
Why the study?
The role of intracellular Ca2+ stores in immature embryonic cardiac myocytes and their contribution to Ca2+ homeostasis was unclear.
RyR-2-mediated Ca2+ release at the junctional membrane complex is essential for cellular Ca2+ homeostasis and survival in immature embryonic cardiac myocytes.
RyR-2 essentiality in mouse embryonic Ca2+ handling warrants no clinical extrapolation; leaves open human developmental or congenital relevance.
In mature cardiac myocytes, Ca2+ influx through the L-type Ca2+ channel activates the ryanodine receptor and triggers Ca2+ release from the sarcoplasmic reticulum (SR). This Ca2+ signal amplification, termed Ca2+-induced Ca2+ release (CICR), occurs within the junctional membrane complex between the plasma membrane and the SR, and is essential for cardiac excitation-contraction (E-C) coupling. On the other hand, Ca2+ available during E-C coupling is predominantly derived from Ca2+ influx in embryonic cardiac myocytes. To examine the role of the intracellular Ca2+ store in immature cardiac myocytes, we have generated knockout mice lacking the cardiac type of the ryanodine receptor (RyR-2), or junctophilin (JP-2) contributing to formation of the junctional membrane complex. Both RyR-2- and JP-2-knockout mice show lethality at early embryonic stages immediately after beginning of heart beating. The loss of RyR-2 produced abnormal SR elements exhibiting vacuolated structures and Ca2+-overloading in embryonic cardiac myocytes. In JP-2-deficient cardiac myocytes, formation of junctional membrane complexes, called pheripheral couplings, was disturbed, and abnormal Ca2+ transients without spatial and temporal synchronization were observed. Therefore, the knockout mice have demonstrated that RyR-2-mediated Ca2+ release at the junctional membrane complex is essential for cellular Ca2+ homeostasis in immature cardiac myocytes.
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Hiroshi Takeshima (2002) studied Embryonic cardiac myocytes. RyR-2 or JP-2 knockout was evaluated on Embryonic lethality and cellular Ca2+ homeostasis. Knockout of RyR-2 or JP-2 in mice caused early embryonic lethality, demonstrating that RyR-2-mediated Ca2+ release is essential for Ca2+ homeostasis in immature cardiac myocytes.
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