Congenital Hypothyroid Pax8−/− Mutant Mice Can Be Rescued by Inactivating the TRα Gene
Open accessAbstract
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.
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Loss of TRs allows the survival of congenitally hypothyroid mice.
“Loss of TRs similarly allows the survival of congenitally hypothyroid mice”
From Thyroid hormone regulates distinct paths to maturation in pigment cell lineages (2019) · cites this paper as Flamant et al., 2002
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Role of Thyroid Hormones in Skeletal Development and Bone Maintenance
Bassett, J. H. D., & Williams, G. R. (2016) · Endocrine Reviews · doi:10.1210/er.2015-1106 · Cited by 540 · see citing works
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The syndromes of reduced sensitivity to thyroid hormone
Dumitrescu, A. M., & Refetoff, S. (2013) · Biochimica et Biophysica Acta (BBA) - General Subjects · doi:10.1016/j.bbagen.2012.08.005 · Cited by 253 · see citing works
Thyroid hormone regulates distinct paths to maturation in pigment cell lineages
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- Authors
- Flamant, F., Poguet, A.-L., Plateroti, M., Chassande, O., Gauthier, K., Streichenberger, N., Mansouri, A., & Samarut, J.
- Affiliations
- Laboratoire de Biologie Moléculaire et Cellulaire de l’Ecole Normale Supérieure de Lyon (F.F., A.-L.P., M.P., O.C., K.G., J.S.), Unité Mixte de Recherche Centre National de la Recherche Scientifique 5665 LA INRA913, 69364 Lyon Cedex 07, France
- École Normale Supérieure de Lyon (FR)
- Lyon 1 Université (FR)
- Max Planck Institute for Biophysical Chemistry (DE)
- Venue
- Molecular Endocrinology
- Publisher
- The Endocrine Society
- Published
- 2002-01-01
- Volume
- 16
- Issue
- 1
- Pages
- 24-32
- ISSN
- 0888-8809 (ISSN-L)
- 1944-9917
- OpenAlex
- W2143237288
- Cited by
- 184
- References
- 1
- Citations by year
- 2025: 1 · 2024: 3 · 2023: 6 · 2022: 5 · 2021: 8 · 2020: 1 · 2019: 6 · 2018: 7 · 2017: 11 · 2016: 8 · 2015: 6 · 2014: 9 · 2013: 9 · 2012: 11
- FWCI
- 5.59
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Molecular Endocrinology (publishedVersion)
- PubMed (publishedVersion)
- HAL (submittedVersion)
- Max Planck Institute for Plasma Physics (submittedVersion)
- MPG.PuRe (Max Planck Society) (submittedVersion)
- Funders
- Ligue Contre le Cancer
- Max-Planck-Gesellschaft
- Centre National de la Recherche Scientifique
- Institut National de la Recherche Agronomique
- Topics
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Thyroid Disorders and Treatments
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Estrogen and related hormone effects
- Keywords
- congenital hypothyroidism, PAX8, thyroid receptor alpha, thyroid follicular cells, postnatal development, spleen, bone, small intestine, thyroid hormone signaling
- MeSH terms
- PAX8 Transcription Factor, Animals, Congenital Hypothyroidism, DNA-Binding Proteins, Gene Expression Regulation, Homozygote, Hypothyroidism, Mice, Mutant Strains, Nuclear Proteins, Receptors, Thyroid Hormone, Spleen, Thyroid Gland, Thyrotropin, Triiodothyronine, Trans-Activators, Survival Rate, Receptors, Cytoplasmic and Nuclear, Gene Silencing, Mice, Paired Box Transcription Factors
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