A Patient with Thyrotropinoma Cosecreting Growth Hormone and Follicle-Stimulating Hormone with Low α-Glycoprotein: A New Subentity?

Elhadd, Tarik; Ghosh, Sujoy; Teoh, Wei Leng; Trevethick, Katy Ann; Hanzély, Zoltán; Dunn, Lee; Malik, Iqbal; Collier, Andrew

doi:10.1089/thy.2008.0384

Cited by 9 publications

(10 citation statements)

References 16 publications

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“…Long acting somatostatin analogue drugs reduce TSH secretion and normalize FT4 and FT3 levels in 90% of patients suffering with pituitary TSH secreting tumors (37). In 25 percent of thyrotropinoma cases there is autonomous secretion of a second pituitary hormone (38). One of our 9 patients with recurrent Cushing's syndrome also had the symptoms of central hyperthyroidism with nal diagnosis of plurihormonal pituitary adenoma with expression of ACTH, FSH and TSH (case 7, Table 2).…”

Section: Discussionmentioning

confidence: 85%

The Value of Ga68-DOTATATE PET/CT in Diagnosis and Management of Suspected Pituitary Tumors

Novruzov

Aliyev

Wan

et al. 2021

Preprint

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Backroud Gallium 68-tetraazacyclododecane-tetraacetic acid-octreotate (Ga-68‑DOTATATE) is a selective somatostatin analogue ligand, which shows increased affinity for somatostatin receptor subtype (SSTR) 2 and has been used routinely for imaging neuroendocrine tumors with PET/CT. We investigated the utility of Ga-68‑DOTATATE positron emission tomography/computed tomography (PET/CT) in patients with suspected pituitary pathology. We reviewed imaging for twenty consecutive patients (8 men, 12 women, mean age of 48.2, range: 14-78) with suspected pituitary pathology who were referred for Ga-68-DOTATATE PET-CT. Results Nine patients presented with recurrent Cushing's syndrome following surgical resection of pituitary adenomas due to recurrent Cushing's disease (seven patients) and ectopic ACTH secreting tumor (2 patients). All seven patients with recurrent Cushing's disease showed positive pituitary Ga-68-DOTATATE uptake while both cases of ectopic hormonal secretion had absent pituitary uptake. In 1 of these 2 patients Ga-68-DOTATATE was able to localize the source of ectopic ACTH tumor. Six patients presented de novo with Cushing's due to ectopic ACTH secretion; Ga-68‑DOTATATE PET/CT was able to localize ectopic tumors in six of eight patients (3 lungs, 2 pancreases, 1 mid-gut) There was high uptake Ga-68-DOTATATE in 3 cases of recurrent central hyperthyroidism (SUVmax 6.6-14.3) and 2 cases of prolactinoma (SUVmax 5.5 and 11.3).Conclusion Absent Ga-68-DOTATATE activity in the pituitary fossa is useful in excluding pituitary disease in recurrent Cushing’s. Recurrent pituitary thyrotropinomas and prolactinomas showed moderate to high pituitary activity. In addition, in Cushing’s syndrome Ga-68-DOTATATE is useful for detection of ectopic sources of ACTH production, especially where anatomic imaging is negative.

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Section: Discussionmentioning

confidence: 85%

The Value of Ga68-DOTATATE PET/CT in Diagnosis and Management of Suspected Pituitary Tumors

Novruzov

Aliyev

Wan

et al. 2021

Preprint

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“…Plurihormonal TSHomas with hypersecretion of GH revealed acromegaly usually masks secondary hyperthyroidism (17,18). A unique study revealed a plurihormonal TSHoma hypersecretion of TSH, GH, and FSH (14). The majority of plurihormonal pituitary tumors may only present with mass tumor effect (headache and visual impairment), without any other symptoms of a functioning pituitary tumor.…”

Section: Discussionmentioning

confidence: 99%

“…The clinical symptoms were variable. Some only presented with hyperthyroidism (12,9,14). Simultaneously, some patients showed acromegaly with hyperprolactinemia (11) or hyperthyroidism (16,17).…”

Section: Discussionmentioning

confidence: 99%

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

Peng

Guo

et al. 2022

Preprint

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Background Thyrotropin (TSH)-secreting pituitary neuroendocrine tumors (PitNETs) are pretty rare pituitary adenomas, occasionally accompanied by hypersecretion of other anterior pituitary hormones: 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 woman diagnosed with TSH PitNET co-secreting GH presented no obvious clinical symptoms. The main characteristics were elevated TSH, fT3, and fT4 levels accompanied by slightly elevated GH and IGF-1levels. The magnetic resonance imaging (MRI) detected a pituitary macroadenoma (18×16×16mm) with cavernous sinus and suprasellar invasion. The immunohistochemistry revealed diffuse positivity for TSH, strong immunoreactivity for GH, and sporadic positivity for PRL. The electron microscope and double immunofluorescence staining confirmed that a plurimorphous plurihormonal adenoma produced TSH, GH, and PRL. After preoperative somatostatin receptor ligands (SRLs) treatment and transsphenoidal surgery, the patient achieved clinical and biochemical remission during follow-up. 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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“…Long acting somatostatin analog drugs reduce TSH secretion and normalize FT4 and FT3 levels in 90% of patients suffering with pituitary TSH secreting tumors (Ben-Shlomo and Melmed 2010 ). In 25% of thyrotropinoma cases, there is autonomous secretion of a second pituitary hormone (Elhadd et al 2009 ). One of our 9 patients with recurrent Cushing’s syndrome also had the symptoms of central hyperthyroidism with final diagnosis of plurihormonal pituitary adenoma with expression of ACTH, FSH, and TSH (case 7, Table 2 ).…”

Section: Discussionmentioning

confidence: 99%

The value of [68Ga]Ga-DOTA-TATE PET/CT in diagnosis and management of suspected pituitary tumors

Novruzov

Aliyev²,

Wan

et al. 2021

European J Hybrid Imaging

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Background Gallium 68-tetraazacyclododecane-tetraacetic acid-octreotate ([68Ga]Ga-DOTA-TATE) is a selective somatostatin analogue ligand, which shows increased affinity for somatostatin receptor subtype (SSTR) 2 and has been used routinely for imaging neuroendocrine tumors with PET/CT. We investigated the utility of [68Ga]Ga-DOTA-TATE positron emission tomography/computed tomography (PET/CT) in patients with suspected pituitary pathology. We reviewed imaging for twenty consecutive patients (8 men, 12 women, mean age of 48.2, range 14–78) with suspected pituitary pathology who were referred for [68Ga]Ga-DOTA-TATE PET/CT. Results Nine patients presented with recurrent Cushing’s syndrome following surgical resection of pituitary adenomas due to recurrent Cushing’s disease (seven patients) and ectopic ACTH secreting tumor (2 patients). All seven patients with recurrent Cushing’s disease showed positive pituitary [68Ga]Ga-DOTA-TATE uptake while both cases of ectopic hormonal secretion had absented pituitary uptake. In 1 of these 2 patients, [68Ga]Ga-DOTA-TATE was able to localize the source of ectopic ACTH tumor. Six patients presented de novo with Cushing’s due to ectopic ACTH secretion; [68Ga]Ga-DOTA-TATE PET/CT was able to localize ectopic tumors in six of eight patients (3 lungs, 2 pancreases, 1 mid-gut) There was high uptake [68Ga]Ga-DOTA-TATE in 3 cases of recurrent central hyperthyroidism (SUVmax 6.6–14.3) and 2 cases of prolactinoma (SUVmax 5.5 and 11.3). Conclusion Absent [68Ga]Ga-DOTA-TATE activity in the pituitary fossa is useful in excluding pituitary disease in recurrent Cushing’s. Recurrent pituitary thyrotropinomas and prolactinomas showed moderate to high pituitary activity. In addition, in Cushing’s syndrome, [68Ga]Ga-DOTA-TATE is useful for detection of ectopic sources of ACTH production, especially where anatomic imaging is negative.

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A Patient with Thyrotropinoma Cosecreting Growth Hormone and Follicle-Stimulating Hormone with Low α-Glycoprotein: A New Subentity?

Abstract: This case of plurihormonal thyrotropinoma is unique in having hypersecretion of TSH, GH, and FSH with low alpha-subunit. Such a combination may represent a new subentity of TSHomas.

Cited by 9 publications

References 16 publications

The Value of Ga68-DOTATATE PET/CT in Diagnosis and Management of Suspected Pituitary Tumors

The Value of Ga68-DOTATATE PET/CT in Diagnosis and Management of Suspected Pituitary Tumors

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

The value of [68Ga]Ga-DOTA-TATE PET/CT in diagnosis and management of suspected pituitary tumors

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