Letter to the Editor
Adrenocortical oncocytoma: 3 case reports
Oncocitoma suprarrenal: presentación de 3 casos
Meritxell Viñes Raczkowski
, Elena Mena Ribas, Guillermo Serra Soler
Corresponding author
Servicio de Endocrinología y Nutrición, Hospital Universitario Son Espases, Palma, Balearic Islands, Spain
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Right: macroscopic aspect of the adrenal mass showing the rupture of the tumour capsule (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Oncocytic cell tumours or oncocytomas are a rare subtype of adrenocortical neoplasms. They are classified according to Lin–Weiss–Bisceglia (LWB) criteria: major criteria (>5 mitoses per 50 high-power fields, atypical mitoses or venous invasion) and minor criteria (size > 10 cm or weight > 200 g, tumour necrosis, capsular or sinusoidal invasion). The existence of any major criteria classifies the tumour as malignant, the existence of any minor criteria as a lesion of uncertain malignant potential, and the absence of all of them as a benign lesion. Despite its low frequency, 3 cases have been diagnosed recently in our centre.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The first clinical case was a 56-year-old male, with no past history of interest, who presented to the emergency department with left flank pain associated with vomiting of several hours’ duration. An urgent abdominal CT scan was requested (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), which showed retroperitoneal bleed and a heterogeneous mass of 10 × 10 × 8 cm in the left adrenal region, with signs of active haemorrhage dependent on the adrenal artery. Once the initial symptoms had stabilised, the patient was admitted for further investigation. During history-taking, the patient reported that he had been asymptomatic until the current episode. The hormonal study showed DHEAS of 314.9 μg/dl (21–199), with the rest of the hormonal tests being normal. The patient underwent left adrenalectomy by laparotomy. Histological examination revealed an oncocytic tumour of uncertain malignant potential according to LWB criteria. After surgery he was asymptomatic, with normal DHEAS and no signs of recurrence in the control CT scan.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second case was a 39-year-old woman, active smoker, who came to the emergency department for a 3-month history of right hypochondrium pain. Urgent abdominopelvic ultrasound showed a mass that appeared to be liver-dependent, heterogeneous with hyperechogenic areas. She was referred to Gastroenterology with a request for an outpatient thoracic–abdominal–pelvic CT. The CT scan showed a heterogeneous mass located in the right subhepatic space, dependent on the right adrenal gland, measuring 11 × 9.6 × 11.6 cm, well defined and with no plane of separation with the liver margin, inferior vena cava and right kidney, structures that it compressed and displaced. It showed density values >40 UH, hypervascularity in the arterial phase and hypodensity in the portal phase due to probable necrosis. The patient was referred to Endocrinology to complete the study. She reported hirsutism in the face, linea alba and limbs for the last 4 months, as well as increased libido, clitoromegaly and irritability.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The hormone analysis showed DHEAS of 1179 μg/dl (47–248); androstenedione 12.7 ng/ml (0.4–3.4); testosterone 1.56 ng/ml (0.13–0.53). Other hormones were normal. The condition was diagnosed as a possible androgen–secreting adrenal carcinoma and anti-androgen treatment with spironolactone was started. The patient underwent right adrenalectomy and prophylactic lymphadenectomy by laparotomy. The pathology report described an oncocytic adrenocortical carcinoma according to the LWB criteria, as it met all the main criteria. One year after surgery, the patient remained asymptomatic, with normal androgen levels and no signs of recurrence.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Finally, the third clinical case was a 51-year-old woman, with no past history of interest, with a left adrenal incidentaloma of 13 mm detected in an abdominopelvic CT scan performed for uterine fibroid. She was asymptomatic during history-taking. The follow-up abdominal MRI at one year showed no changes, suggesting an adenoma with very little fat content (atypical adenoma).</p><p id="par0030" class="elsevierStylePara elsevierViewall">The hormonal study ruled out functionality. It was decided to follow up with radiology. The control CT scan (3 years later due to the COVID-19 pandemic) showed a 23 × 28 mm lesion, homogeneous and well-defined. Given the increase in size, it was decided to perform a laparoscopic left adrenalectomy. The anatomical pathology study described an oncocytic tumour of uncertain malignant potential according to LWB criteria. The patient showed no signs of recurrence on the follow-up CT scan 2 years after surgery.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion, we believe that oncocytomas should be considered in the differential diagnosis of an adrenal mass. They do not have specific radiological features that allow them to be distinguished from pheochromocytoma or adrenal carcinoma. They are usually large tumours and frequently diagnosed incidentally.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Most are non-functional, although in some cases they produce hormone secretion, most commonly virilisation or Cushing’s syndrome.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> The clinical presentation of the cases reported was very different: incidentaloma, tumour mass effect (haemorrhage) and abdominal pain associated with hyperandrogenism. Treatment is surgical and the definitive diagnosis is histological.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Immunoreactivity for vimentin, synaptophysin, alpha-inhibin, melan-A and calretinin is characteristic.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols established in their work centre for accessing clinical history data in order to be able to make this publication available to the scientific community.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">No funding has been received from any entity.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 988 "Ancho" => 2675 "Tamanyo" => 270339 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Left: abdominal CT scan performed in the emergency department, showing the adrenal mass (arrow). Right: macroscopic aspect of the adrenal mass showing the rupture of the tumour capsule (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adrenocortical oncocytic neoplasm: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "J.J. Kanitra" 1 => "J.C. Hardaway" 2 => "T. Soleimani" 3 => "T.J. Koehler" 4 => "M.K. McLeod" 5 => "S. 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