Cushing's syndrome caused by mixed adrenal tumor of cortical origin associated with myelolipoma

Simó-Servat, Andreu; Pérez Maraver, Manuel; Caballero Corchuelo, Jordi

doi:10.1016/j.medcle.2017.03.012

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It must be differentiated from other adrenal gland tumors, such as adenoma, which has no myeloid component and no fat density in imaging tests.<a class="elsevierStyleCrossRef" href="#bib0025">1</a> Rarely associated with adrenal hormone hypersecretion, its diagnosis is radiological and there are fewer than 25 cases reported in the medical literature of myelolipomatous masses causing Cushing's syndrome.<a class="elsevierStyleCrossRef" href="#bib0030">2</a>We report the case of a 57-year-old female patient without any relevant history, who consulted for a 9kg weight gain over a 3-month period. Physical examination showed android obesity with a BMI of 36.3kg/m2, rounded facies, plethoric, kyphosis with fat accumulation in the supraclavicular fossae, wine-red stretch marks on the abdomen, muscle hypertrophy and bruising in the upper limbs. Under drug treatment for hypertension during one month. She also had recent-onset hyperglycemia, requiring insulin treatment.24h urinary free cortisol was 3.124nmol/day (VN 86-631). An abdominal computed tomography (CT) was requested simultaneously for evaluation of altered liver tests, showing decreased density liver in relation with steatosis, hepatic hypodense bilobar nodules compatible with cysts and a left adrenal mass 50mm, heterogeneous, with hypervascular nodular areas and small areas of fat density, indicative of myelolipoma (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).<elsevierMultimedia ident="fig0005"></elsevierMultimedia>The hormone study showed suppressed ACTH (<1.1pmol/l; VN 2-12) and lack of suppression of plasma cortisol. With 0.5mg of dexamethasone every 6h for 2 days: baseline of 709nmol/l (VN 155-678) and final of 933nmol/l. Cushing syndrome diagnosis was made independently of ACTH.To complete the adrenal lesion study, a magnetic resonance imaging was performed, reporting a 50mm×44mm nodule in the left adrenal gland with lipid content, nodular morphology and intense contrast uptake, compatible with myelolipoma. Additionally, a PET-CT scan was also conducted, showing a 40mm×47mm left adrenal mass, heterogeneous, with hypervascular nodular areas and small fat density areas with low carbohydrate uptake (maximum SUV 2.38g/ml), also compatible with myelolipoma; no other significant lesions where observed in the body scan.The patient was operated on by laparoscopic left adrenalectomy. Glucocorticoids replacement therapy was indicated after surgery to prevent adrenal insufficiency secondary to cortisol production suppression of the contralateral adrenal gland caused by the prior hypercortisolism. The patient had a progressive clinical improvement, being able to suspend the antihypertensive and antidiabetic treatment. Histopathology revealed a smooth-outer-surface 71g adrenal gland with a heterogeneous nodular lesion of 7cm×5cm that occupied the entire gland, with yellow and brown areas, some friable zones and other cystic. Normal adrenal tissue was not clearly identified. Histologically, it was a 2-component tumor: adrenal cortical (75%) and myelolipoma (25%). No vascular or capsular invasion criteria were observed. The immunohistochemical study showed that the adrenal cortical origin component was positive for synaptophysin, melan-A, calretinin and inhibin, and negative for chromogranin and cytokeratin AE1. It was described as an adrenocortical adenoma associated with myelolipoma.A dynamic corticotropin stimulation test was performed eight months after surgery, showing adrenal insufficiency (with a plasma cortisol peak of 187nmol/l). The replacement therapy was maintained.Adrenal myelolipoma is a benign tumor and, generally speaking, it is not associated with adrenal hormonal dysfunction. There have been very few cases of adrenal myelolipoma with Cushing's syndrome and adrenalectomy is the disease's curative treatment in all published cases, as well as in ours.<a class="elsevierStyleCrossRef" href="#bib0035">3</a> Myelolipomas usually occur as isolated adrenal lesions, but their association with other adrenal diseases, such as hyperplasia, adenoma or adrenocortical carcinomas has also been described, which can cause adrenal hyperfunction syndromes,<a class="elsevierStyleCrossRef" href="#bib0040">4</a> as in our case. A hormonal study should be conducted in the case of any adrenal mass, as even the lesions with radiological appearance of myelolipoma may be associated with hormonal hypersecretion." 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Med Clin (Barc). 2017;148:285–286." ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 864 "Ancho" => 998 "Tamanyo" => 125656 ] ] "descripcion" => array:1 [ "en" => "Abdominal computed tomography scan." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adrenal neoplasms" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "G. Low" 1 => "H. Dhliwayo" 2 => "D.J. Lomas" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.crad.2012.02.005" "Revista" => array:6 [ "tituloSerie" => "Clin Radiol" "fecha" => "2012" "volumen" => "67" "paginaInicial" => "988" "paginaFinal" => "1000" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22486993" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0030" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Myelolipomatous adrenal masses causing Cushing's syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "C. Lamas" 1 => "L.M. López" 2 => "E. Lozano" 3 => "M. Atienzar" 4 => "R. Ruiz-Mondéjar" 5 => "J.J. Alfaro" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1055/s-0029-1202274" "Revista" => array:6 [ "tituloSerie" => "Exp Clin Endocrinol Diabetes" "fecha" => "2009" "volumen" => "117" "paginaInicial" => "440" "paginaFinal" => "445" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19373749" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0035" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Mielolipoma asociado a adenoma adrenocortical: una causa infrecuente de síndrome de Cushing" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M.S. Corpas Jiménez" 1 => "R. Ortega Salas" 2 => "C. Tenorio Jiménez" 3 => "M.J. Molina Puerta" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.endonu.2013.04.002" "Revista" => array:6 [ "tituloSerie" => "Endocrinol Nutr" "fecha" => "2014" "volumen" => "61" "paginaInicial" => "e7" "paginaFinal" => "e9" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23916698" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0040" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Myelolipomatous foci in an adrenal adenoma causing Cushing's syndrome?" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "I. Vrezas" 1 => "P. Wentworth" 2 => "S.R. Bornstein" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Endocr Res" "fecha" => "2003" "volumen" => "29" "paginaInicial" => "67" "paginaFinal" => "71" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/12665319" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000014800000006/v1_201704120030/S2387020617301870/v1_201704120030/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000014800000006/v1_201704120030/S2387020617301870/v1_201704120030/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020617301870?idApp=UINPBA00004N"]

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Vol. 148. Issue 6.

Pages 285-286 (March 2017)

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