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Letter to the Editor
Normotensive pheochromocytoma associated with ganglioneuroma
Feocromocitoma normotensivo asociado a ganglioneuroma
Gabriela Mintegui
Corresponding author
gabymin92@gmail.com

Corresponding author.
, Beatriz Mendoza, Carolina Guarneri
Clinics Hospital, School of Medicine, UdelaR, Montevideo, Uruguay
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pheochromocytoma is a catecholamine-secreting tumor of the adrenal medulla that causes arterial hypertension &#40;HT&#41; and increased morbidity&#46; It can be associated with an embryologically linked neurogenic tumor &#40;compound pheochromocytoma&#41;&#44; forming a mixed endocrine-neuroblastic tumor &#40;ganglioneuroma in 80&#37; of cases&#41; and represents 1&#8211;3&#37; of pheochromocytomas&#46; It is more frequent in adults between 40 and 60 years of age and they are usually larger tumors than conventional pheochromocytomas&#44; ranging between 4 and 6<span class="elsevierStyleHsp" style=""></span>cm&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Only 5&#8211;15&#37; of pheochromocytomas are normotensive within adrenal incidentalomas and occur mainly in large cystic tumors&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> The frequency of incidentally discovered normotensive pheochromocytoma increased due to better availability and accessibility of imaging procedures&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Thirty-five years old woman with no notable history who consulted for pain in the right hemiabdomen and hypogastrium&#44; without symptoms of peritoneal irritation&#46; Abdominal ultrasound showed a complex image in relation to the upper pole of the left kidney&#44; septate&#44; with vascularization&#44; measuring 92<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>72<span class="elsevierStyleHsp" style=""></span>mm&#46; The CT scan&#44; a well-defined&#44; mixed&#44; predominantly cystic&#44; rounded left adrenal tumor&#44; enhanced with contrast&#44; 85<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>90<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>72<span class="elsevierStyleHsp" style=""></span>mm &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; MR left SR tumor of 10<span class="elsevierStyleHsp" style=""></span>cm&#44; well defined&#59; it contacts the upper renal pole&#44; to which it displaces&#44; and the pancreas&#44; without infiltration elements&#46; In summary&#58; voluminous SR lesion with characteristics of a benign nature&#46; The patient did not present with hypertension&#44; headaches&#44; tachycardia or piloerection&#46; A basic paraclinical was requested that was normal and metanephrines in urine that were 8300<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;24<span class="elsevierStyleHsp" style=""></span>h &#40;normal value &#91;VN&#93; 80&#8211;130&#41;&#59; normetanephrines&#58; 1878<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;24<span class="elsevierStyleHsp" style=""></span>h &#40;VN 111&#8211;419&#41;&#59; total metanephrines 10<span class="elsevierStyleHsp" style=""></span>178<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;24<span class="elsevierStyleHsp" style=""></span>h &#40;VN 149&#8211;535&#41;&#46; Preparation prior to surgery with oral &#945;-adrenergic blocker &#40;terazosin 2&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41; and diet with 5<span class="elsevierStyleHsp" style=""></span>g of sodium per day&#46; Blood pressure monitoring did not show paroxysmal hypertension or hypotension&#46; He did not have tachycardia&#44; so he did not receive &#223;-blockers&#46; He underwent a left adrenalectomy in open surgery&#46; He did not present hemodynamic instability during it&#46; The histopathological characteristics of the 10<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>cm nodule tumor&#44; with multilocular cystic cavity&#44; the largest of 5&#46;5<span class="elsevierStyleHsp" style=""></span>cm with thin and smooth walls&#44; with serohematic content&#46; Remnant of the adrenal gland of 2<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; Microscopy&#58; adrenal parenchyma showing substitution by well-defined&#44; encapsulated proliferation of ganglioneuroma-like cells &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; Immunohistochemistry&#58; the tumor cells were positive for chromogranin A&#44; synaptophysin&#44; with a cytoplasmic granular pattern in S100 sustainable cells&#44; enolase in tumor cells and a schwann component&#44; CD34 in vascular structures&#44; neurofilaments in neuronal cells of the ganglioneuroma component&#46; They were negative for steroidogenic factor 1&#46; Ki 67 shows proliferative activity that in sectors reaches 2&#37; of the activity&#46; The final diagnosis&#58; ganglioneuroma-associated pheochromocytoma &#40;compound pheochromocytoma&#41;&#46; During the evolution&#44; the patient continued to be normotensive and metanephrines and normetanephrines were normal 3 months after surgery&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The classic triad of symptoms in patients with pheochromocytoma comprises episodic headache&#44; sweating&#44; and tachycardia&#46; About half have paroxysmal hypertension&#59; most of the rest have primary hypertension &#40;formerly called &#8220;essential&#8221; hypertension&#41; or normotension&#46; The explanation in the absence of hypertension and the circulating levels of catecholamines may involve several mechanisms such as &#40;1&#41; the &#8220;down-regulation&#8221; of receptors&#44; that is&#44; the decrease in the sensitivity of adrenergic receptors to catecholamines&#59; &#40;2&#41; individual differences in sensitivity of vessels to catecholamines&#59; &#40;3&#41; adaptive response with decreased blood volume&#44; secondary to sustained vasoconstriction&#59; &#40;4&#41; secretion of products with vasodilator action by the tumor&#44; which would modify the response of the vessels to catecholamines &#40;nitric oxide&#44; natriuretic peptide&#44; among others&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3&#44;4</span></a> Pheochromocytoma exceptionally has a malignant behavior and its cystic variant is usually even rarer&#46; This tumor usually does not present the typical clinical&#44; radiological&#44; and laboratory characteristics but is accompanied by nonspecific abdominal symptoms&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> The poor prognosis and&#47;or malignancy factors for this type of tumor are the same as those for conventional pheochromocytomas&#44; such as necrosis&#44; vascular invasion&#44; and high mitotic index&#46; The long-term prognosis of patients after resection of normotensive pheochromocytoma remains uncertain&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Cystic and compound pheochromocytomas are rare neoplasms&#44; mostly incidental findings&#44; and with HT&#46; This case shows a rare variant that&#44; despite presenting very high levels of catecholamines&#44; did not cause symptoms in the patient at any time of her clinical course&#46;</p></span>"
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ISSN: 23870206
Original language: English
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