Now You See, Now You Don’t: A Case Of Spontaneous Regression of Pituitary Tumour

Jaafar, Ainul Syahrilfazli; shokri, Siti salwa Mohd; Paramasvaran, Sanmugarajah; Palaniandy, Kamalanathan; Fadzil, Farizal

doi:10.7759/cureus.9174

Cited by 2 publications

(2 citation statements)

References 6 publications

(16 reference statements)

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“…In the clinical case examples of the current study, we observed a decrease in tumor size in two patients with de novo Cushing disease due to macroadenomas (patients 3 and 4) following treatment with relacorilant. While spontaneous tumor regression cannot be ruled out (although extremely rare) (43), the changes in ACTH and cortisol levels that occurred in these patients during relacorilant treatment, characterized by early increases followed by reductions later during treatment, are consistent with the hypothesized inhibition of ACTH by endogenous somatostatin due to upregulation of SSTR2, as also seen in Patient 1. Together, these data suggest that relacorilant-mediated upregulation of SSTR2 provides more targets for somatostatin and somatostatin analogs, which can lead to tumor shrinkage in ACTH-secreting pituitary tumors.…”

Section: Discussionsupporting

confidence: 73%

Glucocorticoid Receptor Antagonism Upregulates Somatostatin Receptor Subtype 2 Expression in ACTH-Producing Neuroendocrine Tumors: New Insight Based on the Selective Glucocorticoid Receptor Modulator Relacorilant

et al. 2022

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Somatostatin exhibits an inhibitory effect on pituitary hormone secretion, including inhibition of growth hormone and adrenocorticotropic hormone (ACTH), and it can have antisecretory and antitumor effects on neuroendocrine tumors (NETs) that express somatostatin receptors. Although the precise mechanism remains unclear, the finding that glucocorticoids downregulate somatostatin receptor subtype 2 (SSTR2) expression has been used to explain the lack of efficacy of traditional SSTR2-targeting analogs in patients with ACTH-secreting NETs. Glucocorticoid receptor (GR) antagonism with mifepristone has been shown to reverse the glucocorticoid-induced downregulation of SSTR2; however, the effects of GR modulation on SSTR2 expression in ACTH-secreting NETs, particularly corticotroph pituitary tumors, are not well known. The current study presents new insight from in vitro data using the highly selective GR modulator relacorilant, showing that GR modulation can overcome dexamethasone-induced suppression of SSTR2 in the murine At-T20 cell line. Additional data presented from clinical case observations in patients with ACTH-secreting NETs suggest that upregulation of SSTR2 via GR modulation may re-sensitize tumors to endogenous somatostatin and/or somatostatin analogs. Clinical, laboratory, and imaging findings from 4 patients [2 ACTH-secreting bronchial tumors and 2 ACTH-secreting pituitary tumors (Cushing disease)] who were treated with relacorilant as part of two clinical studies (NCT02804750 and NCT02762981) are described. In the patients with ectopic ACTH secretion, SSTR2-based imaging (Octreoscan and 68Ga-DOTATATE positron emission tomography) performed before and after treatment with relacorilant showed increased radiotracer uptake by the tumor following treatment with relacorilant without change in tumor size at computed tomography. In the patients with Cushing disease who received relacorilant prior to scheduled pituitary surgery, magnetic resonance imaging after a 3-month course of relacorilant showed a reduction in tumor size. Based on these findings, we propose that GR modulation in patients with ACTH-secreting NETs upregulates previously suppressed SSTR2s, resulting in tumor-specific antisecretory and anti-proliferative effects. The effect of relacorilant on pituitary corticotroph tumors is being investigated in an ongoing phase 3 study (NCT03697109; EudraCT 2018-003096-35).

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Section: Discussionsupporting

confidence: 73%

Glucocorticoid Receptor Antagonism Upregulates Somatostatin Receptor Subtype 2 Expression in ACTH-Producing Neuroendocrine Tumors: New Insight Based on the Selective Glucocorticoid Receptor Modulator Relacorilant

et al. 2022

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“…Spontaneous disappearance of the tumor following PA is rarely observed. Spontaneous remission of endocrinopathy and regression of the tumor following an apoplectic event is a well-known phenomenon in cases of hormonally active pituitary adenomas; however, the resolution of a nonfunctioning adenoma following PA is rare [ 5 ]. Only 13 cases, including the present case, of spontaneous resolution of nonfunctional pituitary adenomas have been reported in 26 years [ 6 ].…”

Section: Introductionmentioning

confidence: 99%

Resolution of pituitary microadenoma after coronavirus disease 2019: a case report

et al. 2021

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Background This report describes the case of a patient whose pituitary microadenoma resolved after he contracted coronavirus disease 2019. To our knowledge, this is one of the first reported cases of pituitary tumor resolution due to viral illness. We present this case to further investigate the relationship between inflammatory response and tumor remission. Case presentation A 32-year-old man in Yemen presented to the hospital with fever, low blood oxygen saturation, and shortness of breath. The patient was diagnosed with coronavirus disease 2019. Past medical history included pituitary microadenoma that was diagnosed using magnetic resonance imaging and secondary adrenal insufficiency, which was treated with steroids. Due to the severity of coronavirus disease 2019, he was treated with steroids and supportive care. Three months after his initial presentation to the hospital, brain magnetic resonance imaging was performed and compared with past scans. Magnetic resonance imaging revealed changes in the microadenoma, including the disappearance of the hypointense lesion and hyperintense enhancement observed on the previous scan. Conclusions Pituitary adenomas rarely undergo spontaneous resolution. Therefore, we hypothesized that tumor resolution was secondary to an immune response to coronavirus disease 2019.

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Now You See, Now You Don’t: A Case Of Spontaneous Regression of Pituitary Tumour

Cited by 2 publications

References 6 publications

Glucocorticoid Receptor Antagonism Upregulates Somatostatin Receptor Subtype 2 Expression in ACTH-Producing Neuroendocrine Tumors: New Insight Based on the Selective Glucocorticoid Receptor Modulator Relacorilant

Glucocorticoid Receptor Antagonism Upregulates Somatostatin Receptor Subtype 2 Expression in ACTH-Producing Neuroendocrine Tumors: New Insight Based on the Selective Glucocorticoid Receptor Modulator Relacorilant

Resolution of pituitary microadenoma after coronavirus disease 2019: a case report

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