Rescue of the En-1 Mutant Phenotype by Replacement of En-1 with En-2
M Hanks, W Wurst, L Anson-Cartwright, AB Auerbach… - Science, 1995 - science.org
M Hanks, W Wurst, L Anson-Cartwright, AB Auerbach, AL Joyner
Science, 1995•science.orgThe related mouse Engrailed genes En-1 and En-2 are expressed from the one-and
approximately five-somite stages, respectively, in a similar presumptive mid-hindbrain
domain. However, mutations in En-1 and En-2 produce different phenotypes. En-1 mutant
mice die at birth with a large mid-hindbrain deletion, whereas En-2 mutants are viable, with
cerebellar defects. To determine whether these contrasting phenotypes reflect differences in
temporal expression or biochemical activity of the En proteins, En-1 coding sequences were …
approximately five-somite stages, respectively, in a similar presumptive mid-hindbrain
domain. However, mutations in En-1 and En-2 produce different phenotypes. En-1 mutant
mice die at birth with a large mid-hindbrain deletion, whereas En-2 mutants are viable, with
cerebellar defects. To determine whether these contrasting phenotypes reflect differences in
temporal expression or biochemical activity of the En proteins, En-1 coding sequences were …
The related mouse Engrailed genes En-1 and En-2 are expressed from the one- and approximately five-somite stages, respectively, in a similar presumptive mid-hindbrain domain. However, mutations in En-1 and En-2 produce different phenotypes. En-1 mutant mice die at birth with a large mid-hindbrain deletion, whereas En-2 mutants are viable, with cerebellar defects. To determine whether these contrasting phenotypes reflect differences in temporal expression or biochemical activity of the En proteins, En-1 coding sequences were replaced with En-2 sequences by gene targeting. This rescued all En-1 mutant defects, demonstrating that the difference between En-1 and En-2 stems from their divergent expression patterns.
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