Key result
Targeted ablation of the alpha-tropomyosin gene is lethal in homozygous mice, whereas heterozygous mice maintain normal cardiac function and structure despite reduced mRNA levels.
Total alpha-tropomyosin deficiency is incompatible with life, whereas haploinsufficiency is compensated by regulatory mechanisms maintaining normal cardiac structure and function.
Alpha-tropomyosin is essential for embryogenesis; leaves open its specific roles in adult cardiac function.
We created a mouse that lacks a functional alpha-tropomyosin gene using gene targeting in embryonic stem cells and blastocyst-mediated transgenesis. Homozygous alpha-tropomyosin "knockout" mice die between embryonic day 9.5 and 13.5 and lack alpha-tropomyosin mRNA. Heterozygous alpha-tropomyosin knockout mice have approximately 50% as much cardiac alpha-tropomyosin mRNA as wild-type littermates but similar alpha-tropomyosin protein levels. Cardiac gross morphology, histology, and function (assessed by working heart preparations) of heterozygous alpha-tropomyosin knockout and wild-type mice were indistinguishable. Mechanical performance of skinned papillary muscle strips derived from mutant and wild-type hearts also revealed no differences. We conclude that haploinsufficiency of the alpha-tropomyosin gene produces little or no change in cardiac function or structure, whereas total alpha-tropomyosin deficiency is incompatible with life. These findings imply that in heterozygotes there is a regulatory mechanism that maintains the level of myofibrillar tropomyosin despite the reduction in alpha-tropomyosin mRNA.
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Blanchard et al. (1997) studied alpha-tropomyosin deficiency. Targeted ablation of the alpha-tropomyosin gene vs. Wild-type littermates was evaluated on Survival, cardiac morphology, histology, and function. Targeted ablation of the alpha-tropomyosin gene is lethal in homozygous mice, whereas heterozygous mice maintain normal cardiac function and structure despite reduced mRNA levels.
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