Unusual Cushing’s Syndrome and Hypercalcitoninaemia due to a Small Cell Prostate Carcinoma

Antonio, Balestrieri; Magnani, Elena; Nuzzo, Fiorella

doi:10.1155/2016/6308058

Cited by 8 publications

(9 citation statements)

References 22 publications

(28 reference statements)

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“…A partial response to somatostatin analogue has previously been reported in CS secondary to prostate neuroendocrine carcinoma (15). While somatostatin receptor scintigraphy can be helpful to identify neuroendocrine tumours such as bronchial carcinoids causing CS, given the likely aggressive underlying tumour in this setting, FDG-PET/CT would have been more appropriate (16, 17).…”

Section: Discussionmentioning

confidence: 81%

“…From both the literature ( 14 ) and our personal experience, somatostatin analogues may be of benefit in EAS, but despite the tumour demonstrating uptake on somatostatin receptor scintigraphy, there was no biochemical response. A partial response to somatostatin analogue has previously been reported in CS secondary to prostate neuroendocrine carcinoma ( 15 ). While somatostatin receptor scintigraphy can be helpful to identify neuroendocrine tumours such as bronchial carcinoids causing CS, given the likely aggressive underlying tumour in this setting, FDG-PET/CT would have been more appropriate ( 16 , 17 ).…”

Section: Discussionmentioning

confidence: 81%

See 1 more Smart Citation

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Elston

Crawford

Swarbrick

et al. 2017

Endocrine Connections

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Cushing’s syndrome (CS) due to ectopic adrenocorticotrophic hormone (ACTH) is associated with a variety of tumours most of which arise in the thorax or abdomen. Prostate carcinoma is a rare but important cause of rapidly progressive CS. To report a case of severe CS due to ACTH production from prostate neuroendocrine carcinoma and summarise previous published cases. A 71-year-old male presented with profound hypokalaemia, oedema and new onset hypertension. The patient reported two weeks of weight gain, muscle weakness, labile mood and insomnia. CS due to ectopic ACTH production was confirmed with failure to suppress cortisol levels following low- and high-dose dexamethasone suppression tests in the presence of a markedly elevated ACTH and a normal pituitary MRI. Computed tomography demonstrated an enlarged prostate with features of malignancy, confirmed by MRI. Subsequent prostatic biopsy confirmed neuroendocrine carcinoma of small cell type and conventional adenocarcinoma of the prostate. Adrenal steroidogenesis blockade was commenced using ketoconazole and metyrapone. Complete biochemical control of CS and evidence of disease regression on imaging occurred after four cycles of chemotherapy with carboplatin and etoposide. By the sixth cycle, the patient demonstrated radiological progression followed by recurrence of CS and died nine months after initial presentation. Prostate neuroendocrine carcinoma is a rare cause of CS that can be rapidly fatal, and early aggressive treatment of the CS is important. In CS where the cause of EAS is unable to be identified, a pelvic source should be considered and imaging of the pelvis carefully reviewed.

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Section: Discussionmentioning

confidence: 81%

Section: Discussionmentioning

confidence: 81%

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Elston

Crawford

Swarbrick

et al. 2017

Endocrine Connections

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“…Only 17% of cases have an elevated PSA as seen in our patient, which can mislead the diagnosis to prostate adenocarcinoma. We presented a case of SCC NEPC with ACTH-dependent CS as one presenting manifestation [6,11].…”

Section: Discussionmentioning

confidence: 99%

“…Neuroendocrine tumors are more frequently associated with paraneoplastic syndromes like paraneoplastic CS, which accounts for nearly 20% of all CS manifestations but is rarely associated with NEPC [6,11].…”

Section: Discussionmentioning

confidence: 99%

Cushing Syndrome as a Manifestation of Neuroendocrine Prostate Cancer: A Rare Presentation Within a Rare Tumor

Fernandes¹,

Santos²,

Reis³

et al. 2021

Cureus

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Neuroendocrine prostate cancer (NEPC) is a rare entity representing 1% of all prostate malignancies, associated with poor prognosis and often concomitant with paraneoplastic syndromes such as Cushing's syndrome (CS) with ectopic adrenocorticotropic hormone (ACTH) production.We present a case of a 56-year-old man with recent lower urinary tract symptoms hospitalized for pelvic pain, rectal tenesmus, and fatigue. A CT scan documented a large prostatic mass, adenomegalies, and hepatic lesions. Bone scintigraphy showed dispersed osteoblastic metastization. The patient had uncontrolled hypertension and hypokalemia that lead to the diagnosis of paraneoplastic ACTH-dependent CS. Prostate biopsy confirmed small cell NEPC. Potassium supplementation, anti-hypertensive medication, and metyrapone were initiated. The patient was proposed for palliative chemotherapy but died within a few days from a urinary tract infection.The authors aim to draw attention to a case of paraneoplastic CS, a rare manifestation, within the rarity that is NEPC.

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“…: not reported, Among the three cases for which reference was made to ectopic CRH secretion [10,20,27], Rickman and colleagues attributed elevated plasma levels of both CRH and ACTH in their case to ectopic CRH secretion despite the negative IHC results for CRH and ACTH [20]. In 13 of the prior cases, ACTH was confirmed to be the cause of Cushing's syndrome [5,22,26,[28][29][30][31][32][33][34][35][36][37] whereas this information was not documented in the remaining 10 cases [38][39][40][41][42][43][44][45]. In 4 of the 13 documented cases, the detection of ACTH was confirmed only by tissue immunoassay [28,29,32] or by the expression of POMC mRNA [36].…”

Section: Discussionmentioning

confidence: 99%

Molecular analysis and literature-based hypothesis of an immunonegative prostate small cell carcinoma causing ectopic ACTH syndrome

et al. 2019

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Ectopic ACTH syndrome (EAS) due to a prostate small cell carcinoma (SCC) is very rare with only 26 cases reported to date and has a poor prognosis. We here describe another case of this disorder that was clinically typical based on prior reports as it showed hypercortisolemia and severe hypokalemia with multiple metastasis. However, our current case of prostate SCC causing EAS is the first to display negative immunostaining for ACTH despite detectable POMC mRNA expression in the primary lesion. ACTH immunonegativity is thought to be associated with a more aggressive disease course and a poorer prognosis although there are few studies of the underlying mechanisms. We explored two possibilities for this finding in our current patient: aberrant POMC processing prevented immunodetection with an anti-ACTH antibody; and the ACTH content per cell was below the threshold for immunodetection due to its rapid secretion or low synthesis. The aberrant processing theory was thought to be less likely because of immunonegative findings even using anti-POMC/ACTH antibodies. As the plasma ACTH levels in our patient were comparable with those reported for previous immunopositive prostate EAS cases, we speculated that the depletion of ACTH may be caused not only by rapid secretion but also by low production levels as a sign of de-differentiation. De-differentiation may therefore explain the mechanism underlying the negative correlation between immunoreactivity for ACTH in EAS and disease aggressiveness. We believe that our present findings will be of use in future prospective studies aimed at confirming the mechanism of immunonegativity.

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Unusual Cushing’s Syndrome and Hypercalcitoninaemia due to a Small Cell Prostate Carcinoma

Cited by 8 publications

References 22 publications

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Cushing Syndrome as a Manifestation of Neuroendocrine Prostate Cancer: A Rare Presentation Within a Rare Tumor

Molecular analysis and literature-based hypothesis of an immunonegative prostate small cell carcinoma causing ectopic ACTH syndrome

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