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Mol Endocrinol. 2002 Jan;16(1):24-32.

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

Author information

1
Laboratoire de Biologie Moléculaire et Cellulaire de l'Ecole Normale Supérieure de Lyon, Unité Mixte de Recherche Centre National de la Recherche Scientifique 5665 LA INRA913, 69364 Lyon Cedex 07, France. Frederic.Flamant@ens-lyon.fr

Abstract

Mice devoid of all TRs are viable, whereas Pax8(-/-) mice, which lack the follicular cells producing T4 and T3 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.

PMID:
11773436
DOI:
10.1210/mend.16.1.0766
[Indexed for MEDLINE]

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