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Título: | Adult Mice Lacking Mct8 and Dio2 Proteins Present Alterations in Peripheral Thyroid Hormone Levels and Severe Brain and Motor Skill Impairments |
Autor: | Bárez-López, Soledad CSIC ORCID; Grijota Martínez, María del Carmen CSIC ORCID; Ausó Monreal, Eva CSIC; Fernández-de Frutos, Mario; Montero-Pedrazuela, Ana CSIC ORCID ; Guadaño-Ferraz, Ana CSIC ORCID | Palabras clave: | Mct8 deficiency Deiodinase type 2 Mouse model Motor skills impairments Histological brain impairments |
Fecha de publicación: | 14-nov-2019 | Editor: | Mary Ann Liebert | Citación: | Thyroid : official journal of the American Thyroid Association 29(11): 1669-1682 (2019) | Resumen: | [Background]: Mutations in the thyroid hormone (TH) transporter monocarboxylate transporter 8 (MCT8) lead to peripheral hyperthyroidism and profound psychomotor alterations in humans. Mice lacking Mct8 present peripheral hyperthyroidism but no gross neurological abnormalities due to brain compensatory mechanisms involving the enzyme deiodinase type 2 (Dio2). [Methods]: Here we have analyzed the endocrine and neurologic phenotype of mice lacking both Mct8 and Dio2 at three and six months of age. Thyroxine (T4) and 3,5,3′ triiodothyronine (T3) levels/content were measured by specific radioimmunoassays; motor skill performance was evaluated by the footprint, rotarod, four limb hanging wire, and balance beam tests; and brain histological analysis was performed by immunostaining for neurofilament and parvalbumin. [Results]: We have found that this mouse model presents peripheral hyperthyroidism and brain hypothyroidism. Interestingly, the severity of the brain hypothyroidism seems permanent and varies across regions, with the striatum being a particularly affected area. We have also found brain alterations at the histological level compatible with TH deficiency and impaired motor skills. [Conclusions]: These findings indicate the potential of Mct8/Dio2-deficient mice to represent a model for human MCT8 deficiency, to understand the mechanisms underlying its pathophysiology, and ultimately design therapeutic interventions for human patients. |
Versión del editor: | http://dx.doi.org/10.1089/thy.2019.0068 | URI: | http://hdl.handle.net/10261/206250 | DOI: | 10.1089/thy.2019.0068 | ISSN: | 1050-7256 | E-ISSN: | 1557-9077 |
Aparece en las colecciones: | (IIBM) Artículos |
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