Necrosis of a phaeochromocytoma associated with spontaneous remission of diabetes and hypertension

Zanin, Lucia; Rossi, Gianpaolo; Poletti, Alessandro; Piotto, Andrea; Chiesura-Corona, M; Pessina, Achille C.

doi:10.1111/j.1365-2265.1993.tb02417.x

Cited by 14 publications

(8 citation statements)

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“…2 ). This finding was similar to those of previously reported cases of adrenal pheochromocytoma with a large central area of pure necrosis without hemorrhage or tumor rupture [ 5 – 7 , 9 – 11 ]. In our patient, the necrotic material, containing morphologically ghost cells, yielded positive immunoreactivity for chromogranin A. Granulation tissue was adjacent to the necrotic area, accompanied by the presence of numerous hemosiderin-laden macrophages and histiocytes with vascular proliferation.…”

Section: Discussionsupporting

confidence: 92%

Histopathological analysis of spontaneous large necrosis of adrenal pheochromocytoma manifested as acute attacks of alternating hypertension and hypotension: a case report

et al. 2016

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BackgroundPheochromocytomas are rare catecholamine-producing neuroendocrine tumors. Hypertension secondary to pheochromocytoma is often paroxysmal, and patients occasionally present with sudden attacks of alternating hypertension and hypotension. Spontaneous, extensive necrosis within the tumor that is associated with catecholamine crisis is an infrequent complication of adrenal pheochromocytoma, but its pathogenesis remains unclear.Case presentationA 69-year-old Japanese man developed acute-onset episodic headaches, palpitations, and chest pains. During the episodes, both marked fluctuations in blood pressure (ranging from 40/25 to 300/160 mmHg) and high plasma levels of catecholamines were found simultaneously. Radiological findings indicated a 4-cm left adrenal pheochromocytoma. These episodic symptoms disappeared within 2 weeks with normalization of plasma catecholamine levels. Two months later, the patient underwent adrenalectomy. Microscopic examinations revealed pheocromocytoma with a large central area of coagulative necrosis. The necrotic material was immunohistochemically positive for chromogranin A. Granulation tissue was adjacent to the necrotic area, accompanied by numerous hemosiderin-laden macrophages and histiocytes with vascular proliferation. Viable tumor cells, detected along the periphery of the tumor, demonstrated pyknosis, and the Ki-67 labeling index was 2 % in the hot spot. No embolus or thrombus formation was found in the resected specimen harboring the whole tumor. The Pheochromocytoma of the Adrenal gland Scaled Score was 2 out of 20. The patient’s postoperative course was unremarkable for > 7 years.ConclusionsPresumed causal factors for the extensive necrosis of adrenal pheochromocytoma in previously reported cases include hemorrhage into the tumor, hypotension induced by a phentolamine administration, embolic infarction, high intracapsular pressure due to malignant growth of the tumor, and catecholamine-induced vasoconstriction. In the present case, histopathological and clinical findings suggest that under conditions of chronic ischemia due to catecholamine-induced vasoconstriction, an acute infarction occurred after sudden attacks of alternating hypertension and hypotension. Over the subsequent 2 weeks, repetitive massive release of catecholamines from the infarcts into circulation likely accelerated infarction progression by causing repeated attacks of alternating hypertension and hypotension and resulted in the large necrosis. This case highlights the need for physicians to consider acute spontaneous tumor infarction accompanying episodic catecholamine crisis as a rare but severe complication of pheochromocytoma.

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Section: Discussionsupporting

confidence: 92%

Histopathological analysis of spontaneous large necrosis of adrenal pheochromocytoma manifested as acute attacks of alternating hypertension and hypotension: a case report

et al. 2016

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“…Laparoscopic adrenalectomy has recently become the method of choice in surgical treatment of various adrenal gland pathologies, including but not limited to phaeochromocytomas [ 27 , 28 ]. According to different authors, one should expect a somewhat higher, though still acceptable, rate of complications and conversion to the open method in the case of phaeochromocytoma surgeries [ 29 – 31 ].…”

Section: Discussionmentioning

confidence: 99%

The effect of surgical treatment of phaeochromocytoma on concomitant arterial hypertension and diabetes mellitus in a single-centre prospective study

Pogorzelski¹,

Toutounchi²,

Krajewska³

et al. 2014

CEJU

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IntroductionPhaeochromocytoma is one of the numerous causes of secondary hypertension. Furthermore, phaeochromocytoma may first present with type 2 diabetes mellitus. The objective of our study was to evaluate the effects of adrenalectomy on patient recovery with regards to normotension and well–controlled glycaemia.Material and methodsThe retrospective analysis involved 67 patients with phaeochromocytoma operated between 2006 and mid-2012. The pre–operative diagnoses were made in the departments of internal medicine and endocrinology. Based on laboratory tests and diagnostic imaging, we were able to confirm the diagnosis of phaeochromocytoma in 42 (62.7%) patients. We verified the influence of adrenalectomy on the level of patient recovery, with regards to normotension and glycaemic control: arterial pressure and fasting glycaemia levels were obtained on the day of hospital discharge, at follow–up 3 months post–operatively and 1 year after surgical intervention.ResultsOf the 67 patients operated for phaeochromocytoma, 48 (71.6%) were treated laparoscopically, whereas 19 (28.4%) underwent open adrenalectomy. Arterial hypertension was recorded in 53 (79.1%) cases. Furthermore, among this group, diabetes mellitus coexisted in 21 (31.3%) cases. Postoperatively, 70% of cases of arterial hypertension and 90% of type 2 diabetes mellitus were cured. Additionally, a high rate of patients reported a quantitative reduced use of antihypertensive medicines.ConclusionsIn the majority of patients, surgical treatment of symptomatic phaeochromocytoma leads to a regression of arterial hypertension, or a reduction of the number or doses of medicines taken in one's treatment, and glucose–intolerance symptoms.

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“…Spontaneous remission of tumors with a common neural crest embryological origin as paragangliomas has been described as well. Regression of pheochromocytomas after initial presentation with hypertensive crises or shock has been reported [9-11], as well as spontaneous regression [12], Another known entity is spontaneous regression in a subset of pediatric neuroblastomas, especially those detected with mass screening and with biologically favorable characteristics [13,14]. …”

Section: Discussionmentioning

confidence: 99%

Case of spontaneous regression of carotid body tumor in a SDHD mutant: a discussion on potential mechanisms based on a review of the literature

et al. 2012

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BackgroundHead and neck paragangliomas are tumors associated with the parasympathetic nerve system and typically show an indolent growth pattern. Therefore a conservative management strategy is considered in selected cases.Methods and resultsWe present a case of a female patient who presented in 2003 with bilateral carotid body tumors and a tympanic tumor, associated with a mutation in the succinate dehydrogenase -sub-unit-D (SDHD). She was operated on the right carotid body tumor and the tympanic tumor. Thereafter the follow-up was performed with MR examinations at 2-year intervals. After an initial stable phase, over the last 3 years a spontaneous near-total regression of the contralateral carotid body tumor was observed, with only subtle rest-abnormalities visible in 2011.ConclusionsThe present case underlines the indolent growth pattern of head and neck paragangliomas and for the first time describes a rare manifestation of spontaneous regression of a carotid body tumor. The literature was reviewed to discuss this phenomenon.

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Necrosis of a phaeochromocytoma associated with spontaneous remission of diabetes and hypertension

Cited by 14 publications

References 14 publications

Histopathological analysis of spontaneous large necrosis of adrenal pheochromocytoma manifested as acute attacks of alternating hypertension and hypotension: a case report

Histopathological analysis of spontaneous large necrosis of adrenal pheochromocytoma manifested as acute attacks of alternating hypertension and hypotension: a case report

The effect of surgical treatment of phaeochromocytoma on concomitant arterial hypertension and diabetes mellitus in a single-centre prospective study

Case of spontaneous regression of carotid body tumor in a SDHD mutant: a discussion on potential mechanisms based on a review of the literature

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