- Hypothalamus and pituitary gland
- Clinical Characteristics, Diagnosis, and Treatment of Thyroid Stimulating Hormone-Secreting Pituitary Neuroendocrine Tumor (TSH PitNET): A Single-Center Experience
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Jung Heo, Yeon-Lim Suh, Se Hoon Kim, Doo-Sik Kong, Do-Hyun Nam, Won-Jae Lee, Sung Tae Kim, Sang Duk Hong, Sujin Ryu, You-Bin Lee, Gyuri Kim, Sang-Man Jin, Jae Hyeon Kim, Kyu Yeon Hur
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Endocrinol Metab. 2024;39(2):387-396. Published online February 5, 2024
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DOI: https://doi.org/10.3803/EnM.2023.1877
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 Abstract
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- Background
Thyroid-stimulating hormone (TSH)-secreting pituitary neuroendocrine tumor (TSH PitNET) is a rare subtype of PitNET. We investigated the comprehensive characteristics and outcomes of TSH PitNET cases from a single medical center. Also, we compared diagnostic methods to determine which showed superior sensitivity.
Methods
A total of 17 patients diagnosed with TSH PitNET after surgery between 2002 and 2022 in Samsung Medical Center was retrospectively reviewed. Data on comprehensive characteristics and treatment outcomes were collected. The sensitivities of diagnostic methods were compared.
Results
Seven were male (41%), and the median age at diagnosis was 42 years (range, 21 to 65); the median follow-up duration was 37.4 months. The most common (59%) initial presentation was hyperthyroidism-related symptoms. Hormonal co-secretion was present in four (23%) patients. Elevated serum alpha-subunit (α-SU) showed the greatest diagnostic sensitivity (91%), followed by blunted response at thyrotropin-releasing hormone (TRH) stimulation (80%) and elevated sex hormone binding globulin (63%). Fourteen (82%) patients had macroadenoma, and a specimen of one patient with heavy calcification was negative for TSH. Among 15 patients who were followed up for more than 6 months, 10 (67%) achieved hormonal and structural remission within 6 months postoperatively. A case of growth hormone (GH)/TSH/prolactin (PRL) co-secreting mixed gangliocytoma-pituitary adenoma (MGPA) was discovered.
Conclusion
The majority of the TSH PitNET cases was macroadenoma, and 23% showed hormone co-secretion. A rare case of GH/TSH/PRL co-secreting MGPA was discovered. Serum α-SU and TRH stimulation tests showed great diagnostic sensitivity. Careful consideration is needed in diagnosing TSH PitNET. Achieving remission requires complete tumor resection. In case of nonremission, radiotherapy or medical therapy can improve the long-term remission rate.
- Thyroid
- Steroid Responsive Xanthomatous Hypophysitis Associated with Autoimmune Thyroiditis: A Case Report
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Ji Young Joung, Hyemin Jeong, Yoon Young Cho, Kyoungmin Huh, Yeon-Lim Suh, Kwang-Won Kim, Ji Cheol Bae
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Endocrinol Metab. 2013;28(1):65-69. Published online March 25, 2013
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DOI: https://doi.org/10.3803/EnM.2013.28.1.65
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Abstract
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We report the case of a 36-year-old woman who presented with headache, fever, and amenorrhea. Laboratory analysis revealed hypopituitarism and autoimmune thyroiditis, while a cerebrospinal fluid study suggested concurrent aseptic meningitis. A magnetic resonance image (MRI) scan revealed a 1.0×0.9 cm cystic mass enlarging the sella turcica. Surgical resection via an endoscopic transsphenoidal route was performed. The histological finding of the excised tissue revealed foamy histiocytes with vacuolated cytoplasm, supporting the diagnosis of xanthomatous hypophysitis. Although a residual soft lesion was observed on the MRI image postoperatively, the patient's headache and fever improved. Ten months after surgery, the patient complained of visual impairment and headache, and the residual mass had enlarged into the suprasellar area. High dose (500 mg intravenous) methylprednisolone was administered for 3 days. During the methylprednisolone pulse therapy, the patient's visual acuity and headache improved. A follow-up MRI taken after methylprednisolone therapy showed a marked mass reduction. Our case supports an autoimmune pathophysiology for xanthomatous hypophysitis and suggests that high dose glucocorticoid therapy as a treatment option. -
Citations
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AACE Clinical Case Reports.2021; 7(3): 220. CrossRef - Xanthomatous Hypophysitis: A Case Report and Comprehensive Literature Review
Jianyu Zhu, Zhicheng Wang, Wenze Wang, Jinghua Fan, Yi Zhang, Xiaoxu Li, Jie Liu, Shenzhong Jiang, Kan Deng, Lian Duan, Yong Yao, Huijuan Zhu
Frontiers in Endocrinology.2021;[Epub] CrossRef - Recurring Primary Xanthomatous Hypophysitis Behaving Like Pituitary Adenoma: Additional Case and Literature Review
Mansour Mathkour, Tyler Zeoli, Cassidy Werner, Tyler Scullen, Juanita Garces, Joseph Keen, Marcus Ware
World Neurosurgery.2020; 138: 27. CrossRef - Imaging findings in hypophysitis: a review
Ferdinando Caranci, Giuseppe Leone, Andrea Ponsiglione, Massimo Muto, Fabio Tortora, Mario Muto, Sossio Cirillo, Luca Brunese, Alfonso Cerase
La radiologia medica.2020; 125(3): 319. CrossRef - Hypophysitis in the era of immune checkpoint inhibitors and immunoglobulin G4-related disease
Leen Wehbeh, Sama Alreddawi, Roberto Salvatori
Expert Review of Endocrinology & Metabolism.2019; 14(3): 167. CrossRef - Xanthomatous Hypophysitis Is Associated with Ruptured Rathke’s Cleft Cyst
Kai Duan, Sylvia L. Asa, Daniel Winer, Zadeh Gelareh, Fred Gentili, Ozgur Mete
Endocrine Pathology.2017; 28(1): 83. CrossRef - Xanthomatous Hypophysitis Presenting with Diabetes Insipidus Completely Cured Through Transsphenoidal Surgery: Case Report and Literature Review
Wei Lin, Lu Gao, Xiaopeng Guo, Wenze Wang, Bing Xing
World Neurosurgery.2017; 104: 1051.e7. CrossRef - Review of xanthomatous lesions of the sella
B.K. Kleinschmidt‐DeMasters, Kevin O. Lillehei, Todd C. Hankinson
Brain Pathology.2017; 27(3): 377. CrossRef - Xanthomatous hypophysitis
Bishoy Hanna, Yan M. Li, Timothy Beutler, Parul Goyal, Walter A. Hall
Journal of Clinical Neuroscience.2015; 22(7): 1091. CrossRef - Clinical Characteristics, Management, and Outcome of 22 Cases of Primary Hypophysitis
Sun Mi Park, Ji Cheol Bae, Ji Young Joung, Yoon Young Cho, Tae Hun Kim, Sang-Man Jin, Sunghwan Suh, Kyu Yeon Hur, Kwang-Won Kim
Endocrinology and Metabolism.2014; 29(4): 470. CrossRef - Brief Review of Articles in 'Endocrinology and Metabolism' in 2013
Won-Young Lee
Endocrinology and Metabolism.2014; 29(3): 251. CrossRef
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