{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,28]],"date-time":"2026-06-28T06:45:02Z","timestamp":1782629102359,"version":"3.54.5"},"reference-count":26,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,6,11]],"date-time":"2026-06-11T00:00:00Z","timestamp":1781136000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"name":"Stoffskifteforbundet","award":["NA"],"award-info":[{"award-number":["NA"]}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Thyroid\u00ae"],"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>Resistance to thyroid hormone alpha (RTH\u03b1) is a rare genetic disorder with symptoms of hypothyroidism, but normal or close-to-normal thyroid function tests. Treatment with levothyroxine (L-T4) may be beneficial. This study investigated the efficacy, safety and biochemical changes of high-dose L-T4 and liothyronine (L-T3) treatment.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Four RTH\u03b1 (Ala263Val) patients were treated with L-T4 (1.75 \u00b5g\/kg) in a pilot open-label study and monitored for three years. Quality of life (QoL) was assessed using ThyPro. Analysis of auxiological- and biochemical-parameters, bone mineral density (BMD) and exploratory metabolomics was performed. A partial replacement of L-T4 with L-T3 was attempted.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>Treatment with L-T4 increased FT4, FT3, and rT3 to supraphysiological concentrations (relative to reference intervals) and suppressed thyroid-stimulating hormone with no adverse effects. Heart rate, bone markers, and sex hormone-binding globulin transiently increased. Several classes of lipids were reduced. BMD appeared unaltered. Patients reported improved QoL. One patient presented a short non-sustained ventricular tachycardia on L-T3 + L-T4.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>L-T4 treatment of adult RTH\u03b1 patients was safe and improved QoL in adherent patients.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/10507256261460201","type":"journal-article","created":{"date-parts":[[2026,6,11]],"date-time":"2026-06-11T09:52:44Z","timestamp":1781171564000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Thyroxine Treatment of Adult RTH\u03b1 Patients: Safety, Efficacy, and Metabolomic Changes"],"prefix":"10.1177","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2619-4192","authenticated-orcid":false,"given":"Alexander Bauer","family":"Westbye","sequence":"first","affiliation":[{"name":"Hormone Laboratory, Department of Medical Biochemistry, Oslo University Hospital, Aker, Oslo, Norway."},{"name":"Endocrinology and Metabolism Research Group, Oslo University Hospital, Aker, Oslo, Norway."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louise Koren","family":"Dahll","sequence":"additional","affiliation":[{"name":"Hormone Laboratory, Department of Medical Biochemistry, Oslo University Hospital, Aker, Oslo, Norway."},{"name":"Endocrinology and Metabolism Research Group, Oslo University Hospital, Aker, Oslo, Norway."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"May Kristin","family":"Bredahl","sequence":"additional","affiliation":[{"name":"Hormone Laboratory, Department of Medical Biochemistry, Oslo University Hospital, Aker, Oslo, Norway."},{"name":"Endocrinology and Metabolism Research Group, Oslo University Hospital, Aker, Oslo, Norway."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Per Medb\u00f8e","family":"Thorsby","sequence":"additional","affiliation":[{"name":"Hormone Laboratory, Department of Medical Biochemistry, Oslo University Hospital, Aker, Oslo, Norway."},{"name":"Endocrinology and Metabolism Research Group, Oslo University Hospital, Aker, Oslo, Norway."},{"name":"Institute of Clinical Medicine, Faculty of Medicine, University of Oslo, Oslo, Norway."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara Salehi","family":"Hammerstad","sequence":"additional","affiliation":[{"name":"Institute of Clinical Medicine, Faculty of Medicine, University of Oslo, Oslo, Norway."},{"name":"Department of Endocrinology, Morbid Obesity and Preventive Medicine, Oslo University Hospital, Oslo, Norway."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,6,11]]},"reference":[{"key":"e_1_3_3_2_1","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00030.2013"},{"key":"e_1_3_3_3_1","doi-asserted-by":"publisher","DOI":"10.1210\/er.2009-0007"},{"key":"e_1_3_3_4_1","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2013.3393.nomen"},{"key":"e_1_3_3_5_1","doi-asserted-by":"publisher","DOI":"10.4274\/jcrpe.galenos.2020.2019.0190"},{"key":"e_1_3_3_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.beem.2015.07.007"},{"key":"e_1_3_3_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/bs.ctdb.2017.02.001"},{"key":"e_1_3_3_8_1","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2021.656551"},{"key":"e_1_3_3_9_1","doi-asserted-by":"publisher","DOI":"10.1530\/endoabs.101.PS3-27-08"},{"key":"e_1_3_3_10_1","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2019.0602"},{"key":"e_1_3_3_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/S2213-8587(14)70111-1"},{"key":"e_1_3_3_12_1","doi-asserted-by":"publisher","DOI":"10.1210\/jendso\/bvad089"},{"key":"e_1_3_3_13_1","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2021.785175"},{"key":"e_1_3_3_14_1","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2017.0157"},{"key":"e_1_3_3_15_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1110296"},{"key":"e_1_3_3_16_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2016-1404"},{"key":"e_1_3_3_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40618-023-02043-1"},{"key":"e_1_3_3_18_1","doi-asserted-by":"publisher","DOI":"10.1159\/000539348"},{"key":"e_1_3_3_19_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-2215"},{"key":"e_1_3_3_20_1","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2015.0463"},{"key":"e_1_3_3_21_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-1050"},{"key":"e_1_3_3_22_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-023-38960-1"},{"key":"e_1_3_3_23_1","doi-asserted-by":"publisher","DOI":"10.1097\/HEP.0000000000000551"},{"key":"e_1_3_3_24_1","doi-asserted-by":"publisher","DOI":"10.1530\/ETJ-23-0062"},{"key":"e_1_3_3_25_1","doi-asserted-by":"publisher","DOI":"10.1210\/er.2015-1106"},{"key":"e_1_3_3_26_1","doi-asserted-by":"publisher","DOI":"10.1210\/en.2010-1491"},{"key":"e_1_3_3_27_1","doi-asserted-by":"publisher","DOI":"10.1210\/clinem\/dgad072"}],"container-title":["Thyroid\u00ae"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10507256261460201","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/10507256261460201","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10507256261460201","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,28]],"date-time":"2026-06-28T05:56:01Z","timestamp":1782626161000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/10507256261460201"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,11]]},"references-count":26,"alternative-id":["10.1177\/10507256261460201"],"URL":"https:\/\/doi.org\/10.1177\/10507256261460201","relation":{},"ISSN":["1050-7256","1557-9077"],"issn-type":[{"value":"1050-7256","type":"print"},{"value":"1557-9077","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,11]]},"article-number":"10507256261460201"}}