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Letter to the Editor
Adrenal schwannoma: A case report
Schwannoma suprarrenal: descripción de un caso
Clara Fuentes
Corresponding author
, Leire Garaizabal, Octavio Pérez
Servicio de Endocrinología, Hospital Universitario Araba, Vitoria-Gasteiz, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A schwannoma is a benign&#44; encapsulated tumor originating in the Schwann cells of the peripheral sympathetic nervous system&#44; the cranial nerves of the head and neck&#44; and the upper or lower limbs&#46; Its occurrence in an adrenal gland is unusual&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Most of these tumors are benign&#44; with malignant ones being more common in cases of Von Recklinghausen&#8217;s disease&#46; They are more prevalent among women than men &#40;1&#46;2&#58;1&#41;&#44; and the mean age of onset is 49 years&#46; Most are discovered as incidental findings&#44; and their symptoms seem to be related to the size of the tumor&#44; such as abdominal pain caused by the mass effect&#46; Hormonal test findings tend to be normal&#46; Computed tomography &#40;CT&#41; images tend to show a well-defined&#44; encapsulated&#44; homogeneous mass and magnetic resonance imaging &#40;MRI&#41; reveals a heterogeneous mass&#44; with either cystic degeneration or hemorrhagic areas with a hypointense T1 signal and a hyperintense T2 signal&#44; which&#44; therefore&#44; lead to a differential diagnosis of pheochromocytoma or adrenal carcinoma&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We hereby present the case of a 55-year-old woman with hypertransaminasemia secondary to alcohol consumption&#46; A requested MRI revealed a heterogeneous&#44; left adrenal mass with a diameter of 3&#46;5&#160;&#215;&#160;3&#160;cm&#44; with some irregular areas of hyperintense T2 signal and others of hypointense cystic degeneration&#47;central necrosis&#46; The lesion contained no adipose or hemorrhagic material&#44; and exhibited no signs of invasion of the adjacent structures or adenopathies&#46; A differential diagnosis of either pheochromocytoma or adrenal carcinoma was proposed as the main option&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was referred to the Endocrinology Department for further examination&#46; She reported a three-year history of arterial hypertension&#44; with a worse control of her blood pressure levels over the previous year &#40;mean 160&#47;100&#160;mmHg&#41;&#44; owing to which she is currently under treatment with two antihypertensive drugs&#46; A hormonal study was requested to determine the concentrations of renin mass&#44; aldosterone&#44; urinary free cortisol&#44; plasma cortisol after administering 1&#160;mg of dexamethasone&#44; urinary metanephrines&#44; dehydroepiandrosterone sulphate&#44; total testosterone&#44; androstenedione&#44; and 17-hydroxyprogesterone&#44; all of which fell within normal limits&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient was subsequently referred to the Surgery Department to undergo a left adrenalectomy and reach a definitive pathological diagnosis&#46; This procedure was performed laparoscopically in March 2017&#44; with the pathological study of the samples obtained yielding a diagnosis of a schwannoma limited to the adrenal gland&#46; The macroscopic description was reported as an adrenal gland measuring 8&#46;5&#160;&#215;&#160;4&#160;&#215;&#160;3&#160;cm&#44; with a 3&#46;5&#160;&#215;&#160;3&#46;2-cm nodule delimited by a capsule&#46; The nodule corresponded to a solid mass with whitish-grayish cystic areas&#46; The microscopic description reported a tumor in the adrenal medulla that was displacing its cortex and was comprised by a proliferation of spindle cells with a fascicular pattern&#44; elongated nuclei&#44; and an ill-defined cytoplasm&#44; with areas of increased cellularity&#44; nuclear &#8220;pseudo-palisading&#8221;&#44; myxoid areas with poor cellular representation&#44; and blood vessels with thick and hyalinized walls&#46; Areas of ischemia&#44; cystic degeneration&#44; and polymorphous nuclei without mitosis were also observed&#46; The mitotic index was less than 1 mitosis&#47;10 high power fields &#40;HPFs&#41;&#44; and the Ki-67 index was 5&#37;&#46; In addition&#44; an immunohistochemistry study revealed a positive result for the S-100 protein and a negative one for actin&#44; caldesmon&#44; desmin&#44; AE1-AE3&#44; and inhibin&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">After the procedure&#44; the patient was re-examined in the Endocrinology Department with the aid of a Holter blood pressure device&#44; which revealed mean blood pressure levels of 107&#47;74 mmHg and&#44; therefore&#44; a need for decreasing her antihypertensive medication&#46; A physical examination revealed no &#8220;<span class="elsevierStyleItalic">caf&#233; au lait</span>&#8221; spots&#44; but several axillary freckles were detected in a region where&#44; according to the patient&#44; several&#44; small&#44; pedunculated&#44; benign tumors had previously been removed&#46; The corresponding pathology report could not be retrieved from her medical history&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">To conclude&#44; schwannomas are solid&#44; encapsulated tumors&#44; frequently presenting with areas of bleeding&#44; necrosis&#44; and cysts&#44; which are generally located in the cranial and cervical cutaneous nerves&#44; with their presence in the retroperitoneum and adrenal glands being rare&#46; Because CT or MRI images rarely allow their preoperative differentiation from pheochromocytomas or adrenal carcinomas&#44;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> preoperative hormonal testing is advised to rule out an increase in metanephrine levels requiring prior treatment with alpha-blockers&#46; Only a single case of a schwannoma associated with increased levels of urinary catecholamines in the laboratory tests has been described&#46; Pathological studies often reveal hemorrhagic and cystic areas within the tumors&#46; In addition&#44; despite not being pathognomonic&#44; positivity for the S-100<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> protein in the immunohistochemistry studies is a fundamental finding&#46; The clinical evolution of this condition is generally benign&#44; except in cases associated with Von Recklinghausen&#8217;s fibromatosis&#44; where malignancy is more frequent&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The patient was also examined in the Ophthalmology Department to have an optic glioma ruled out owing to the suspected diagnosis of type 1 neurofibromatosis&#44; although she did not fulfill the criteria for genetic testing&#46;</p></span>"
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ISSN: 23870206
Original language: English
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