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Publication : Congenital hypothyroid Pax8(-/-) mutant mice can be rescued by inactivating the TRalpha gene.

First Author  Flamant F Year  2002
Journal  Mol Endocrinol Volume  16
Issue  1 Pages  24-32
PubMed ID  11773436 Mgi Jnum  J:73626
Mgi Id  MGI:2156109 Doi  10.1210/mend.16.1.0766
Citation  Flamant F, et al. (2002) Congenital Hypothyroid Pax8(-/-) Mutant Mice Can Be Rescued by Inactivating the TRalpha Gene. Mol Endocrinol 16(1):24-32
abstractText  Mice devoid of all TRs are viable, whereas Pax8(-/-) mice, which lack the follicular cells producing T(4) and T(3) in the thyroid gland, die during the first weeks of postnatal life. A precise comparison between the two types of mutants reveals that their phenotypes are similar, but the defects in spleen, bone, and small intestine are more pronounced in Pax8(-/-) mice. This is interpreted as the result of a negative effect of the unliganded TR on thyroid hormone target genes expression in the Pax8(-/-) mutants. Pax8/TRalpha compound mutants can survive to adulthood, and the expression of target genes is partially restored. This demonstrates the importance of TRalpha aporeceptor activity in several aspects of postnatal development.
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