The ZPA Regulatory Sequence (ZRS) Hand2 and Twist1 Binding Sites are Not Necessary for ZRS Activity
نویسندگان
چکیده
منابع مشابه
Intrafamilial variability of ZRS-associated syndrome: characterization of a mosaic ZRS mutation by pyrosequencing.
During limb development, the spatio-temporal expression of sonic hedgehog (SHH) is driven by the Zone of polarizing activity Regulatory Sequence (ZRS), located 1 megabase upstream from SHH. Gain-of-function mutations of this enhancer, which cause ectopic expression of SHH, are known to be responsible for congenital limb malformations with variable expressivity, ranging from preaxial polydactyly...
متن کاملIdentification of spontaneous mutations within the long-range limb-specific Sonic hedgehog enhancer (ZRS) that alter Sonic hedgehog expression in the chicken limb mutants oligozeugodactyly and silkie breed.
The evolutionarily conserved, non-coding ~800-base-pair (bp) zone of polarizing activity (ZPA) regulatory sequence (ZRS) controls Shh expression in the posterior limb. We report that the chicken mutant oligozeugodactyly (ozd), which lacks limb Shh expression, has a large deletion within the ZRS. Furthermore, the preaxial polydactylous, Silkie Breed chicken, which develops ectopic anterior limb ...
متن کاملOpposing Functions of the ETS Factor Family Define Shh Spatial Expression in Limb Buds and Underlie Polydactyly
Sonic hedgehog (Shh) expression during limb development is crucial for specifying the identity and number of digits. The spatial pattern of Shh expression is restricted to a region called the zone of polarizing activity (ZPA), and this expression is controlled from a long distance by the cis-regulator ZRS. Here, members of two groups of ETS transcription factors are shown to act directly at the...
متن کامل06-P016 The Shh cis-regulatory element, ZRS, functions to specify spatial information
thesis, absorption or flow of the Cerebral Spinal Fluid (CSF) and is a common developmental disability in humans. Although CH has a significant genetic component in mice and humans, it is poorly understood at the molecular level. The Subcommissural Organ (SCO), a gland residing at the dorsal opening of the Sylvian aqueduct, has been implicated in CSF flow maintenance. We have established BAC tr...
متن کاملShh and ZRS enhancer co-localisation is specific to the zone of polarizing activity
CC-BY-ND 4.0 International license not peer-reviewed) is the author/funder. It is made available under a The copyright holder for this preprint (which was. Abstract Limb-specific Shh expression is regulated by the (~1 Mb distant) ZRS enhancer. In the mouse, limb bud restricted spatiotemporal expression of Shh occurs from ~E10-E11.5 at the distal posterior margin and is essential for correct aut...
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ژورنال
عنوان ژورنال: The FASEB Journal
سال: 2021
ISSN: 0892-6638,1530-6860
DOI: 10.1096/fasebj.2021.35.s1.04726