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      Thyrotropinoma with silent somatotroph and lactotroph adenoma during pregnancy

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          Abstract

          Summary

          Thyrotropinomas are an uncommon cause of hyperthyroidism and are exceedingly rarely identified during pregnancy, with limited evidence to guide management. Most commonly they present as macroadenomas and may cause symptoms of mass effect including headache, visual field defects and hypopituitarism. We present a case of a 35-year-old woman investigated for headaches in whom a 13 mm thyrotropinoma was found. In the lead-up to planned trans-sphenoidal surgery (TSS), she spontaneously conceived and surgery was deferred, as was pharmacotherapy, at her request. The patient was closely monitored through her pregnancy by a multi-disciplinary team and delivered without complication. Pituitary surgery was performed 6 months post-partum. Isolated secondary hypothyroidism was diagnosed postoperatively and replacement thyroxine was commenced. Histopathology showed a double lesion with predominant pituitary transcription factor-1 positive, steroidogenic factor negative plurihormonal adenoma and co-existent mixed thyroid-stimulating hormone, growth hormone, lactotroph and follicle-stimulating hormone staining with a Ki-67 of 1%. This case demonstrates a conservative approach to thyrotropinoma in pregnancy with a successful outcome. This highlights the need to consider the timing of intervention with careful consideration of risks to mother and fetus.

          Learning points
          • Thyrotropinomas are a rare cause of secondary hyperthyroidism. Patients may present with hyperthyroidism or symptoms of mass effect, including headaches or visual disturbance.

          • Thyrotropinoma in pregnancy presents a number of pituitary-related risks including pituitary apoplexy and compression of local structures.

          • Hyperthyroidism in pregnancy raises the risk of complications including spontaneous abortion, preeclampsia, low birthweight and premature labour.

          • Timing of medical and surgical therapies must be carefully considered. A conservative approach requires careful monitoring in case emergent intervention is required.

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          Most cited references13

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          Hyperthyroidism in pregnancy.

          Changes in thyroid hormone concentrations that are characteristic of hyperthyroidism must be distinguished from physiological changes in thyroid hormone economy that occur in pregnancy, especially in the first trimester. Approximately one to two cases of gestational hyperthyroidism occur per 1000 pregnancies. Identification of hyperthyroidism in a pregnant woman is important because adverse outcomes can occur in both the mother and the offspring. Graves' disease, which is autoimmune in nature, is the usual cause; but hyperthyroidism in pregnancy can be caused by any type of hyperthyroidism--eg, toxic multinodular goitre or solitary autonomously functioning nodule. Gestational transient thyrotoxicosis is typically reported in women with hyperemesis gravidarum, and is mediated by high circulating concentrations of human chorionic gonadotropin. Post-partum thyroiditis occurs in 5-10% of women, and many of those affected ultimately develop permanent hypothyroidism. Antithyroid drug treatment of hyperthyroidism in pregnant women is controversial because the usual drugs--methimazole or carbimazole--are occasionally teratogenic; and the alternative--propylthiouracil--can be hepatotoxic. Fetal hyperthyroidism can be life-threatening, and needs to be recognised as soon as possible so that treatment of the fetus with antithyroid drugs via the mother can be initiated. In this Review, we discuss physiological and pathophysiological changes in thyroid hormone economy in pregnancy, the diagnosis and management of hyperthyroidism during pregnancy, severe life-threatening thyrotoxicosis in pregnancy, neonatal thyrotoxicosis, and post-partum hyperthyroidism.
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            2013 European Thyroid Association Guidelines for the Diagnosis and Treatment of Thyrotropin-Secreting Pituitary Tumors

            Hyperthyroidism is mainly due to autoimmune thyroid disorders or toxic goiter, and very rarely to the presence of thyrotropin (TSH)-secreting pituitary adenomas (TSHomas). These tumors are characterized by high levels of circulating free thyroid hormones (FT4 and FT3) in the presence of nonsuppressed serum TSH concentrations. Failure to correctly diagnose TSHomas may result in inappropriate thyroid ablation, which results in a significant increase of pituitary tumor mass. The diagnosis is mainly achieved by measuring TSH after T3 suppression and TRH stimulation tests. These dynamic tests, together with pituitary imaging and genetic testing are useful in distinguishing TSHomas from the syndromes of resistance to thyroid hormone action. The treatment of choice is surgery. In cases of surgical failure, somatostatin analogs have been found to be effective in normalizing TSH secretion in more than 90% of patients.
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              Pituitary tumours: TSH-secreting adenomas.

              Thyrotropin-secreting pituitary adenomas (TSHomas) are a rare cause of hyperthyroidism and account for less than 2% of all pituitary adenomas. In the last years, the diagnosis has been facilitated by the routine use of ultra-sensitive TSH immunometric assays. Failure to recognise the presence of a TSHoma may result in dramatic consequences, such as improper thyroid ablation that may cause the pituitary tumour volume to further expand. The diagnosis mainly rests on dynamic testing, such as T3 suppression tests and TRH, which are useful in differentiating TSHomas from the syndromes of thyroid hormone resistance. The first therapeutical approach to TSHomas is the pituitary neurosurgery. The medical treatment of TSHomas mainly rests on the administration of somatostatin analogues, such as octreotide and lanreotide, which are effective in reducing TSH secretion in more than 90% of patients with consequent normalisation of FT4 and FT3 levels and restoration of the euthyroid state.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                18 August 2022
                2022
                : 2022
                : 21-0194
                Affiliations
                [1 ]Department of Clinical Medicine , Endocrinology
                [2 ]Department of Clinical Medicine , Neurosurgery, Macquarie University, Sydney, New South Wales, Australia
                [3 ]Department of Neurosurgery , Royal Melbourne Hospital, Melbourne, Victoria, Australia
                [4 ]Peter McCallum Cancer Centre , Department of Oncology, Melbourne, Victoria, Australia
                [5 ]School of Medicine , University of Notre Dame, Sydney, New South Wales, Australia
                Author notes
                Correspondence should be addressed to D Lewis; Email: lewis.med@ 123456protonmail.ch
                Author information
                http://orcid.org/0000-0002-1925-9675
                Article
                EDM210194
                10.1530/EDM-21-0194
                9513668
                36137189
                11db973c-dd36-4476-bb4d-dd7e8ecbc31c
                © The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 26 June 2022
                : 18 August 2022
                Categories
                Adult
                Female
                Asian - Filipino
                Australia
                Pituitary
                Pituitary
                Obstetrics
                Unique/Unexpected Symptoms or Presentations of a Disease
                Unique/Unexpected Symptoms or Presentations of a Disease

                adult,female,asian - filipino,australia,pituitary,obstetrics,unique/unexpected symptoms or presentations of a disease,september,2022

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