Rescue of the En-1 mutant phenotype by replacement of En-1 with En-2
- PMID: 7624797
- DOI: 10.1126/science.7624797
Rescue of the En-1 mutant phenotype by replacement of En-1 with En-2
Abstract
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.
Comment in
-
Knocking genes in instead of out.Science. 1995 Aug 4;269(5224):636. doi: 10.1126/science.7624790. Science. 1995. PMID: 7624790 No abstract available.
Similar articles
-
Two Pax2/5/8-binding sites in Engrailed2 are required for proper initiation of endogenous mid-hindbrain expression.Mech Dev. 2000 Feb;90(2):155-65. doi: 10.1016/s0925-4773(99)00242-7. Mech Dev. 2000. PMID: 10640701
-
Drosophila engrailed can substitute for mouse Engrailed1 function in mid-hindbrain, but not limb development.Development. 1998 Nov;125(22):4521-30. doi: 10.1242/dev.125.22.4521. Development. 1998. PMID: 9778510
-
Multiple developmental defects in Engrailed-1 mutant mice: an early mid-hindbrain deletion and patterning defects in forelimbs and sternum.Development. 1994 Jul;120(7):2065-75. doi: 10.1242/dev.120.7.2065. Development. 1994. PMID: 7925010
-
Antisense inhibition of engrailed genes in mouse embryos reveals roles for these genes in craniofacial and neural tube development.Teratology. 1995 May;51(5):300-10. doi: 10.1002/tera.1420510506. Teratology. 1995. PMID: 7482351
-
Genetic control of segmentation in the vertebrate hindbrain.Perspect Dev Neurobiol. 1995;3(1):29-38. Perspect Dev Neurobiol. 1995. PMID: 8542253 Review.
Cited by
-
Cannabinoid type-1 receptor signaling in dopaminergic Engrailed-1 expressing neurons modulates motivation and depressive-like behavior.Front Mol Neurosci. 2024 Apr 4;17:1379889. doi: 10.3389/fnmol.2024.1379889. eCollection 2024. Front Mol Neurosci. 2024. PMID: 38660383 Free PMC article.
-
Nigral transcriptomic profiles in Engrailed-1 hemizygous mouse models of Parkinson's disease reveal upregulation of oxidative phosphorylation-related genes associated with delayed dopaminergic neurodegeneration.Front Aging Neurosci. 2024 Feb 5;16:1337365. doi: 10.3389/fnagi.2024.1337365. eCollection 2024. Front Aging Neurosci. 2024. PMID: 38374883 Free PMC article.
-
Interspecies chimerism with human embryonic stem cells generates functional human dopamine neurons at low efficiency.Stem Cell Reports. 2024 Jan 9;19(1):54-67. doi: 10.1016/j.stemcr.2023.11.009. Epub 2023 Dec 21. Stem Cell Reports. 2024. PMID: 38134925 Free PMC article.
-
Imaging and multi-omics datasets converge to define different neural progenitor origins for ATRT-SHH subgroups.Nat Commun. 2023 Oct 20;14(1):6669. doi: 10.1038/s41467-023-42371-7. Nat Commun. 2023. PMID: 37863903 Free PMC article.
-
Transcriptomic Analyses of Brains of RBM8A Conditional Knockout Mice at Different Developmental Stages Reveal Conserved Signaling Pathways Contributing to Neurodevelopmental Diseases.Int J Mol Sci. 2023 Feb 27;24(5):4600. doi: 10.3390/ijms24054600. Int J Mol Sci. 2023. PMID: 36902031 Free PMC article.
Publication types
MeSH terms
Substances
LinkOut - more resources
Full Text Sources
Other Literature Sources
Molecular Biology Databases
Research Materials