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      TSH adenoma and syndrome of resistance to thyroid hormones—Two cases report of syndrome of inappropriate secretion of thyrotropin

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          SITSH (syndrome of inappropriate secretion of thyrotropin) is a rare clinical state defined as uninhibited serum thyroid stimulating hormone in the presence of elevated thyroid hormone. This state is complicated and mainly caused by the abnormal feedback of hypothalamus–pituitary thyroid axis. The TSH adenoma (TSH‐oma) and resistance to thyroid hormones (RTH) are the main etiologies of SITSH. As is well known that the treatment strategies of RTH and TSH‐oma are apparently different, thus identifying the difference between RTH and TSH‐oma is of great significance for the diagnosis and treatment of SITSH.

          Case description

          A 62‐year‐old man with a state of elevated thyroid hormones and inappropriate elevated serum TSH level was hospitalized in 2016. Results of the pituitary enhanced magnetic resonance imaging and the somatostatin test respectively demonstrated a space‐occupying lesion of pituitary and an elevated serum sex hormone binding globulin (SHBG) and inhibited TSH secretion, which indicated the occurrence of TSH‐oma. In 2019, a 23‐year‐old girl with a state of elevated thyroid hormones and inappropriate normal serum TSH was hospitalized. Interestingly, whole exome sequencing detection suggested a pathogenic mutation in thyroid hormone receptor β ( THRB) gene, which has been shown to be associated with RTH.


          The difference between TSH‐oma and RTH ought to be clarified for their accurate diagnose and treatment. The clinical experiences of the two cases reported here suggest that more detail information such as family medical history, serum SHBG level, and THRB gene test is helpful for the diagnose and treatment of TSH‐oma and RTH. Additionally, we also summarized the identification points, diagnosis process, and treatment strategies for these two rare diseases.


          Due to the complicated manifestation of TSH‐oma and RTH, the diagnosis and treatment often delayed. The clinical experiences of the TSH‐oma and RTH suggest that more information should be identified. In this article, by comparing TSH‐oma and RTH, we summarized the identification points, diagnosis process and treatment strategies of these two rare diseases.

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          Most cited references 22

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          Familial syndrome combining deaf-mutism, stuppled epiphyses, goiter and abnormally high PBI: possible target organ refractoriness to thyroid hormone.

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            Multiple mechanisms for regulation of the transcriptional activity of thyroid hormone receptors.

             Robert Cheng (1999)
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              Thyrotropin-secreting pituitary adenomas: epidemiology, diagnosis, and management.

              Inappropriate secretion of TSH was first described in 1960 in a patient with evidence of hyperthyroidism and expanded sella on imaging. It was later found that a type of pituitary adenoma that secretes TSH (thyrotropinoma) was the underlying cause. The objective of the present review article is to summarize data on the epidemiology, pathogenesis, diagnosis, and management of thyrotropinomas. The prevalence of thyrotropinomas is lower than that of other pituitary adenomas. Early diagnosis is now possible thanks to the availability of magnetic resonance imaging and sensitive laboratory assays. As a corollary, many patients now present earlier in the course of their disease and have smaller tumors at the time of diagnosis. Treatment also has evolved over time. Transsphenoidal surgery is still considered definitive therapy. Meanwhile, radiation therapy, including radiosurgery, is effective in achieving tumor control in the majority of patients. In the past, radiation therapy was used as second line treatment in patients with residual or recurrent tumor after surgery. However, the availability of somatostatin analogs, which can lead to normalization of thyroid function as well as shrink these tumors, has led to an increase in the role of medical therapy in patients who are not in remission after pituitary surgery. In addition, dopamine agonists have shown some efficacy in the management of these tumors. Better understanding of the molecular pathogenesis of thyrotropinomas may lead to rationally designed therapies for patients with thyrotropinomas.

                Author and article information

                Brain Behav
                Brain Behav
                Brain and Behavior
                John Wiley and Sons Inc. (Hoboken )
                10 March 2021
                May 2021
                : 11
                : 5 ( doiID: 10.1002/brb3.v11.5 )
                [ 1 ] Department of Endocrinology Southwest Hospital Third Military Medical University (Army Medical University) Chongqing China
                [ 2 ] Breast and Thyroid Surgical Department Chongqing General Hospital University of Chinese Academy of Sciences Chongqing China
                Author notes
                [* ] Correspondence

                Jiong‐yu Hu, Department of endocrinology, Southwest Hospital, Third Military Medical University (Army Medical University), Chongqing, China.

                Email: jiongyuhu@ 123456163.com

                Fan Zhang, Breast and thyroid surgical department, Chongqing General Hospital, University of Chinese Academy of Sciences, Chongqing, China.

                Email: zhangfan316@ 123456163.com

                © 2021 The Authors. Brain and Behavior published by Wiley Periodicals LLC

                This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

                Page count
                Figures: 6, Tables: 2, Pages: 7, Words: 4077
                Funded by: Medical Technology Innovation Fund of Chongqing General Hospital
                Award ID: Y2020ZDXM06
                Original Article
                Original Research
                Custom metadata
                May 2021
                Converter:WILEY_ML3GV2_TO_JATSPMC version:6.0.2 mode:remove_FC converted:14.05.2021


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