{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T10:09:18Z","timestamp":1790762958844,"version":"4.1.0"},"reference-count":65,"publisher":"SAGE Publications","issue":"11","license":[{"start":{"date-parts":[[2019,11,1]],"date-time":"2019-11-01T00:00:00Z","timestamp":1572566400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Thyroid\u00ae"],"published-print":{"date-parts":[[2019,11]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>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).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>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\u2032 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.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>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.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>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.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1089\/thy.2019.0068","type":"journal-article","created":{"date-parts":[[2019,7,30]],"date-time":"2019-07-30T08:06:15Z","timestamp":1564473975000},"page":"1669-1682","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":47,"title":["Adult Mice Lacking Mct8 and Dio2 Proteins Present Alterations in Peripheral Thyroid Hormone Levels and Severe Brain and Motor Skill Impairments"],"prefix":"10.1177","volume":"29","author":[{"given":"Soledad","family":"B\u00e1rez-L\u00f3pez","sequence":"first","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."},{"name":"Unit 708, Center for Biomedical Research On Rare Diseases (Ciberer), Instituto de Salud Carlos III, Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carmen","family":"Grijota-Mart\u00ednez","sequence":"additional","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."},{"name":"Unit 708, Center for Biomedical Research On Rare Diseases (Ciberer), Instituto de Salud Carlos III, Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eva","family":"Aus\u00f3","sequence":"additional","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."},{"name":"Departamento de \u00d3ptica, Farmacolog\u00eda y Anatom\u00eda, Universidad de Alicante, Alicante, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mario","family":"Fern\u00e1ndez-de Frutos","sequence":"additional","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ana","family":"Montero-Pedrazuela","sequence":"additional","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ana","family":"Guada\u00f1o-Ferraz","sequence":"additional","affiliation":[{"name":"Instituto de Investigaciones Biom\u00e9dicas Alberto Sols, Consejo Superior de Investigaciones Cient\u00edficas (CSIC)-Universidad Aut\u00f3noma de Madrid (UAM), Madrid, Spain."},{"name":"Unit 708, Center for Biomedical Research On Rare Diseases (Ciberer), Instituto de Salud Carlos III, Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2019,11,14]]},"reference":[{"key":"e_1_3_4_2_2","first-page":"325","article-title":"Some examples of the inheritance of mental deficiency: apparently sex-linked idiocy and microencephaly","volume":"48","author":"Allan W","year":"1944","unstructured":"Allan W, , Herndon C, , Dudley F. 1944. 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