ArticleProceedings of the National Academy of Sciences of the United States of America1995
Analysis of the mouse Splotch-delayed mutation indicates that the Pax-3 paired domain can influence homeodomain DNA-binding activity.
Article in Proceedings of the National Academy of Sciences of the United States of America, 1995. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.
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Who cites it
12 citing papers in PubMed.
- Altered sacral neural crest development in Pax3 spina bifida mutants underlies deficits of bladder innervation and function.Developmental biology · 2021Article
- Recurrent PAX3-MAML3 fusion in biphenotypic sinonasal sarcoma.Nature genetics · 2014Article
- Leucophores are similar to xanthophores in their specification and differentiation processes in medaka.Proceedings of the National Academy of Sciences of the United States of America · 2014Article
- Functional interaction between Foxd3 and Pax3 in cardiac neural crest development.Genesis (New York, N.Y. : 2000) · 2011Article
- Mutational analysis of the eyeless gene and phenotypic rescue reveal that an intact Eyeless protein is necessary for normal eye and brain development in Drosophila.Developmental biology · 2009Article
- Cell-type-specific regulation of distinct sets of gene targets by Pax3 and Pax3/FKHR.Oncogene · 2005Article
- Review
- Crystal structure of the human Pax6 paired domain-DNA complex reveals specific roles for the linker region and carboxy-terminal subdomain in DNA binding.Genes & development · 1999Article
- The highly conserved beta-hairpin of the paired DNA-binding domain is required for assembly of Pax-Ets ternary complexes.Molecular and cellular biology · 1999Article
- Waardenburg syndrome.Journal of medical genetics · 1997Review
- The incidence of deafness is non-randomly distributed among families segregating for Waardenburg syndrome type 1 (WS1).Journal of medical genetics · 1997Review
- An alternative splicing event in the Pax-3 paired domain identifies the linker region as a key determinant of paired domain DNA-binding activity.Molecular and cellular biology · 1996Article
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3 authors.
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Abstract
The murine Pax-3 protein contains two DNA-binding domains, a paired domain and a homeodomain, and alterations in the Pax-3 gene are responsible for the neural tube defects observed in the Splotch (Sp) mouse mutant. Of five Sp alleles, Splotch-delayed (Spd) is the only one that encodes a full-length Pax-3 protein, containing a single glycine-to-arginine substitution within the paired domain. To better understand the consequence of this mutation on Pax-3 function, we have analyzed the DNA-binding properties of wild-type and Spd Pax-3, using oligonucleotides that bind primarily to the paired domain (e5) or exclusively to the homeodomain (P2). Wild-type Pax-3 was found to bind e5 in a specific manner. In contrast, the Spd mutation reduced binding of Pax-3 to e5 17-fold, revealing a defect in DNA binding by the paired domain. Surprisingly, the Spd mutation also drastically reduced the homeodomain-specific binding to P2 by 21-fold when compared with the wild-type protein. Interestingly, a deletion which removes the Spd mutation was found to restore P2-binding activity, suggesting that within the full-length Pax-3 protein, the paired domain and homeodomain may interact. We conclude, therefore, that the Spd mutation is phenotyically expressed in vitro by a defect in the DNA-binding properties of Pax-3. Furthermore, it is apparent that the paired domain and homeodomain of Pax-3 do not function as independent domains, since a mutation in the former impairs the DNA-binding activity of the latter.
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