Polycystic ovary syndrome with a Sertoli-Leydig cell tumor: A case report

Pérez de Arenaza Pozo, Víctor; Miguelez González, María; Moya Orduñez, Luis Daniel

doi:10.1016/j.medcle.2024.04.025

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Positive staining for calretinin, malan-A, inhibin and WT1; negative for vimentin." ] ] ] "textoCompleto" => "Androgenic group of tumors originated mainly from sex-cord stromal ovarian tumors (SCSTs) are including steroid cell tumors, Leydig tumors, granulosa cell tumors, Sertoli cell tumors, Sertoli-Leydig cell tumors (SLTC), gonadoblastomas, and some other rare forms as ovarian metastases from neuroendocrine tumors.<a class="elsevierStyleCrossRefs" href="#bib0030">1,2</a>Sertoli cell tumors, SLTC and Leydig cell tumors, they account for less than 1% of all ovarian tumors. Compared to other SCST that occur in postmenopausal women, average age at presentation of SLTC is 25 years with a wide range (1–84 years).<a class="elsevierStyleCrossRef" href="#bib0035">2</a>Clinically, these tumors often manifest signs of hyperandrogenism (as they are known to secrete testosterone) or symptoms of a pelvic mass (solid or solid cystic, less frequently cystic).<a class="elsevierStyleCrossRef" href="#bib0040">3</a>A 29-year-old woman with a history of grade 3 obesity, polycystic ovary syndrome (PCOS), and lupus. She was under gynecological follow-up for PCOS and receiving desogestrel with normal gynecological exams, referred to Endocrinology for obesity management. During the consultation, she exhibited a non-Cushing morphotype, with a BMI of 41.5, a weight of 117kg, and a height of 168cm. During the anamnesis, the presence of facial hair protruding from the mask was observed, completing the examination, which revealed a Ferriman-Galway score of >8.Blood tests were requested for the next appointment, which showed testosterone levels of 211ng/mL (8.4–48.1), estimated free testosterone of 33.16pg/mL (0.7–3.6), delta-4 androstenedione of 1.22ng/mL (0.3–3.3), dehydroepiandrosterone sulfate (DHEA-S) of 47.60μg/dL (98.8–340), SHBG 35.20nmol/l (32.4–128). 17-OH progesterone 1.8ng/mL, insulin of 85.80μUI/mL (2.6–24.9), HOMA index of 19.43 (<2.5) and a normal low-dose dexamethasone suppression test. Treatment with drospirenone and metformin was initiated, and an ovarian ultrasound was requested, revealing numerous bilateral cysts similar to previous ultrasounds. In the following consultation, signs of hyperandrogenism persisted, leading to the initiation of spironolactone. Due to testosterone levels above 150ng/mL, a pelvic magnetic resonance imaging was requested.The March 2023 MRI showed enlargement of the right ovary, suggestive of an ovarian tumor (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The endocrinology committee decided that the most probable diagnosis was a SCSTs delaying bariatric surgery. In May 2023, the ultrasound revealed a solid mass in the right ovary measuring 50mm×38.8mm with well-defined borders and intratumoral Doppler flow (score 2–3). In August 2023, a laparoscopic right oophorectomy was performed, and the specimen was sent for pathological examination. A well-differentiated neoplasm was found with densely arranged tubular structures with stromal strands between them, grouped in nodules. Luteinized stromal cells and other cells with eosinophilic cytoplasm, rounded nuclei, and grouped suggestive of Leydig cells were found in the stroma, confirming the diagnosis of a SLTC (pTNM (8va ed.2017)/FIGO(2018): pT1a/(IA) L0 V0). Study of gene DICER-1 was performed to rule out a DICER-1-syndrome but resulted negative. The risk of having a germline DICER-1 mutation after a diagnosis of ovarian SLCT can be as high as 69%.<a class="elsevierStyleCrossRefs" href="#bib0040">3–5</a><elsevierMultimedia ident="fig0005"></elsevierMultimedia>During follow-up, a computed tomography (CT) of the neck, chest, abdomen, and pelvis was performed in the postoperative period, and no pathological changes were found. The serum testosterone levels are 14ng/dl and the clinical symptoms of hyperandrogenism disappeared. It is important to note that after the surgery, spironalctone was withdrawn and drosperinone was maintained. Nowadays, she is wating for bariatric obesity surgery.In summary, women with PCOS have serum testosterone concentrations that varies between 45 and 60ng/dL. In case of an elevated above 150ng/dL an ovarian androgen-secreting tumor ovarian hyperthecosis or adrenal tumors is suspected. Treatment is surgical (fertility-sparing surgery in patients of reproductive age) and a long-term follow-up is recommended because of the risk of recurrence (median time to relapse of 4–6 years). It is important to considered for genetic counseling for DICER-1 syndrome in any women with SLTC.<a class="elsevierStyleCrossRefs" href="#bib0045">4,5</a>Ethical considerationThe patient's consent has been obtained and recorded for the publication of the case.FundingWe have not received any financial aid or funding.Conflict of interestWe have no conflict of interest." "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical consideration" ] 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:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1194 "Ancho" => 1480 "Tamanyo" => 206744 ] ] "descripcion" => array:1 [ "en" => "Findings from the pathological analysis: well-differentiated neoplasia characterized by the proliferation of dense tubular structures within stromal strands, featuring luteinized stromal cells and other exhibiting eosinophilic cytoplasm and rounded nuclei. Positive staining for calretinin, malan-A, inhibin and WT1; negative for vimentin." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Ovarian Sertoli cell tumor with immature prepubertal-like Sertoli cell component: a case report and literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "D. Bužinskienė" 1 => "E. Šidlovska" 2 => "G. Vilutytė" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.3390/medicina58111638" "Revista" => array:5 [ "tituloSerie" => "Medicina (Kaunas)" "fecha" => "2022" "volumen" => "58" "paginaInicial" => "1638" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/36422177" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Androgen-secreting ovarian tumors" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "D. Macut" 1 => "D. Ilić" 2 => "A. Mitrović Jovanović" 3 => "J. Bjekić-Macut" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1159/000494906" "Revista" => array:5 [ "tituloSerie" => "Front Horm Res" "fecha" => "2019" "volumen" => "53" "paginaInicial" => "100" "paginaFinal" => "107" ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Update on ovarian sex cord-stromal tumors" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "Z. Ordulu" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.cll.2023.03.001" "Revista" => array:6 [ "tituloSerie" => "Clin Lab Med" "fecha" => "2023" "volumen" => "43" "paginaInicial" => "245" "paginaFinal" => "274" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/37169445" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Assessment of cardiovascular risk and prevention of cardiovascular disease in women with the polycystic ovary syndrome: a consensus statement by the Androgen Excess and Polycystic Ovary Syndrome (AE-PCOS) Society" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R.A. Wild" 1 => "E. Carmina" 2 => "E. Diamanti-Kandarakis" 3 => "A. Dokras" 4 => "H.F. Escobar-Morreale" 5 => "W. Futterweit" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1210/jc.2009-2724" "Revista" => array:6 [ "tituloSerie" => "J Clin Endocrinol Metab" "fecha" => "2010" "volumen" => "95" "paginaInicial" => "2038" "paginaFinal" => "2049" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20375205" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Spectrum of DICER1 germline pathogenic variants in ovarian Sertoli-Leydig cell tumor" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "E. De Paolis" 1 => "R.M. Paragliola" 2 => "P. Concolino" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.3390/jcm10091845" "Revista" => array:5 [ "tituloSerie" => "J Clin Med" "fecha" => "2021" "volumen" => "10" "paginaInicial" => "1845" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/33922805" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000016300000008/v1_202410240712/S2387020624004455/v1_202410240712/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/23870206/0000016300000008/v1_202410240712/S2387020624004455/v1_202410240712/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020624004455?idApp=UINPBA00004N"]

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Vol. 163. Issue 8.

Pages 429-430 (October 2024)

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