Clinical Characteristics, Diagnosis, and Treatment of Thyroid Stimulating Hormone-Secreting Pituitary Neuroendocrine Tumor (TSH PitNET): A Single-Center Experience

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

doi:10.3803/EnM.2023.1877

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Clinical Characteristics, Diagnosis, and Treatment of Thyroid Stimulating Hormone-Secreting Pituitary Neuroendocrine Tumor (TSH PitNET): A Single-Center Experience

Authors: 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

Citation: Endocrinology and Metabolism (대한내분비학회지), Vol.39(2) : epub-396, 2024-04

Journal Title: Endocrinology and Metabolism(대한내분비학회지)

ISSN: 2093-596X

Issue Date: 2024-04

MeSH: Adult ; Aged ; Female ; Follow-Up Studies ; Humans ; Male ; Middle Aged ; Neuroendocrine Tumors* / blood ; Neuroendocrine Tumors* / diagnosis ; Neuroendocrine Tumors* / metabolism ; Neuroendocrine Tumors* / therapy ; Pituitary Neoplasms* / blood ; Pituitary Neoplasms* / diagnosis ; Pituitary Neoplasms* / metabolism ; Retrospective Studies ; Thyrotropin* / blood ; Thyrotropin* / metabolism ; Treatment Outcome ; Young Adult

Keywords: Central hyperthyroidism ; Mixed gangliocytoma pituitary adenoma ; Pituitary neuroendocrine tumor ; Thyrotropin-secreting pituitary neuroendocrine tumor ; alpha-Subunit

Abstract: Backgruound: 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.