Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review

Abstract Background Thyrotropin-secreting pituitary neuroendocrine tumors (PitNETs) are rare pituitary adenomas that are occasionally accompanied by hypersecretion of other anterior pituitary hormones, such as growth hormone (GH) and prolactin (PRL). The clinical, biochemical, and pathological chara...

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Main Authors: Guiliang Peng, Chuanhong Guo, Yangfan Lv, Dandan Li, Ling Zhou, Rufei Shen, Yong Chen, Xin Zheng, Zheng Sun, Hongting Zheng, Min Long
Format: Article
Language:English
Published: BMC 2022-12-01
Series:BMC Endocrine Disorders
Subjects:
Online Access:https://doi.org/10.1186/s12902-022-01220-2
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author Guiliang Peng
Chuanhong Guo
Yangfan Lv
Dandan Li
Ling Zhou
Rufei Shen
Yong Chen
Xin Zheng
Zheng Sun
Hongting Zheng
Min Long
author_facet Guiliang Peng
Chuanhong Guo
Yangfan Lv
Dandan Li
Ling Zhou
Rufei Shen
Yong Chen
Xin Zheng
Zheng Sun
Hongting Zheng
Min Long
author_sort Guiliang Peng
collection DOAJ
description Abstract Background Thyrotropin-secreting pituitary neuroendocrine tumors (PitNETs) are rare pituitary adenomas that are occasionally accompanied by hypersecretion of other anterior pituitary hormones, such as growth hormone (GH) and prolactin (PRL). The clinical, biochemical, and pathological characteristics may represent diverse circumstances. Case presentation In this report, a 33-year-old female diagnosed with a TSH PitNET co-secreting GH presented no obvious clinical symptoms. The main characteristics were elevated thyroid-stimulating hormone (TSH), free tri-iodothyronine (FT3), and free thyroxine (FT4) levels accompanied by slightly elevated GH and insulin-like growth factor-1 (IGF-1) levels. Magnetic resonance imaging (MRI) detected a pituitary macroadenoma (18 × 16 × 16 mm) with cavernous sinus and suprasellar invasion. Immunohistochemistry revealed diffuse positivity for TSH, strong immunoreactivity for GH, and sporadic positivity for PRL. The electron microscope and double immunofluorescence staining confirmed a plurimorphous plurihormonal adenoma producing TSH, GH, and PRL. After preoperative somatostatin receptor ligand (SRL) treatment and transsphenoidal surgery, the patient achieved temporary clinical and biochemical remission. However, 3 months after surgery, the patient was suspected of having Hashimoto’s thyroiditis due to higher thyroglobulin antibody (TGAb), thyroid peroxidase antibody (TPOAb), and thyroid receptor antibody (TRAb) and an enlarged thyroid nodule. During follow-up, thyroid function and TSH slowly transformed from transient hyperthyroidism to hypothyroidism. They were maintained in the normal range by L-T4. Conclusion In the TSH PitNET, the positive immunohistochemistry for TSH, GH, and PRL translated into hormonal overproduction with TSH and GH.
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spelling doaj.art-1ecbad1b8d7641ce9b6ef23a066105df2022-12-25T12:19:11ZengBMCBMC Endocrine Disorders1472-68232022-12-012211810.1186/s12902-022-01220-2Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature reviewGuiliang Peng0Chuanhong Guo1Yangfan Lv2Dandan Li3Ling Zhou4Rufei Shen5Yong Chen6Xin Zheng7Zheng Sun8Hongting Zheng9Min Long10Department of Endocrinology, translational Research Key Laboratory for Diabetes, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Pathology, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Pathology, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityNational Drug Clinical Trial Institute, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Endocrinology, translational Research Key Laboratory for Diabetes, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Endocrinology, translational Research Key Laboratory for Diabetes, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Neurosurgery, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Neurosurgery, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Medicine, Division of Diabetes, Endocrinology and Metabolism, Baylor College of MedicineDepartment of Endocrinology, translational Research Key Laboratory for Diabetes, The Second Affiliated Hospital (Xinqiao Hospital) of Army Medical UniversityDepartment of Endocrinology, The First Affiliation Hospital (Southwest Hospital) of Army Medical UniversityAbstract Background Thyrotropin-secreting pituitary neuroendocrine tumors (PitNETs) are rare pituitary adenomas that are occasionally accompanied by hypersecretion of other anterior pituitary hormones, such as growth hormone (GH) and prolactin (PRL). The clinical, biochemical, and pathological characteristics may represent diverse circumstances. Case presentation In this report, a 33-year-old female diagnosed with a TSH PitNET co-secreting GH presented no obvious clinical symptoms. The main characteristics were elevated thyroid-stimulating hormone (TSH), free tri-iodothyronine (FT3), and free thyroxine (FT4) levels accompanied by slightly elevated GH and insulin-like growth factor-1 (IGF-1) levels. Magnetic resonance imaging (MRI) detected a pituitary macroadenoma (18 × 16 × 16 mm) with cavernous sinus and suprasellar invasion. Immunohistochemistry revealed diffuse positivity for TSH, strong immunoreactivity for GH, and sporadic positivity for PRL. The electron microscope and double immunofluorescence staining confirmed a plurimorphous plurihormonal adenoma producing TSH, GH, and PRL. After preoperative somatostatin receptor ligand (SRL) treatment and transsphenoidal surgery, the patient achieved temporary clinical and biochemical remission. However, 3 months after surgery, the patient was suspected of having Hashimoto’s thyroiditis due to higher thyroglobulin antibody (TGAb), thyroid peroxidase antibody (TPOAb), and thyroid receptor antibody (TRAb) and an enlarged thyroid nodule. During follow-up, thyroid function and TSH slowly transformed from transient hyperthyroidism to hypothyroidism. They were maintained in the normal range by L-T4. Conclusion In the TSH PitNET, the positive immunohistochemistry for TSH, GH, and PRL translated into hormonal overproduction with TSH and GH.https://doi.org/10.1186/s12902-022-01220-2Growth hormone (GH)ImmunohistochemistryPlurihormonal pit-1-positive adenomaPituitary neuroendocrine tumor (PitNET)Thyroid-stimulating hormone (TSH)
spellingShingle Guiliang Peng
Chuanhong Guo
Yangfan Lv
Dandan Li
Ling Zhou
Rufei Shen
Yong Chen
Xin Zheng
Zheng Sun
Hongting Zheng
Min Long
Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
BMC Endocrine Disorders
Growth hormone (GH)
Immunohistochemistry
Plurihormonal pit-1-positive adenoma
Pituitary neuroendocrine tumor (PitNET)
Thyroid-stimulating hormone (TSH)
title Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
title_full Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
title_fullStr Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
title_full_unstemmed Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
title_short Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review
title_sort pleiomorphism plurihormonal pit 1 positive macroadenoma with central hyperthyroidism a rare case report and literature review
topic Growth hormone (GH)
Immunohistochemistry
Plurihormonal pit-1-positive adenoma
Pituitary neuroendocrine tumor (PitNET)
Thyroid-stimulating hormone (TSH)
url https://doi.org/10.1186/s12902-022-01220-2
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