Presentation of two patients with malignant granulosa cell tumors, with a review of the literature

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WORLD JOURNAL OF SURGICAL ONCOLOGY

CASE REPORT

Open Access

Presentation of two patients with malignant granulosa cell tumors, with a review of the literature Ryousuke Tamura, Yoshihito Yokoyama*, Takeshi Yanagita, Yukiko Matsumura, Kazuhiro Abe, Ryousuke Taniguchi and Hideki Mizunuma

Abstract Granulosa cell tumors (GCTs) of the ovary account for 2 to 5 of ovarian malignancies. We present two patients with malignant ovarian adult GCT. In one patient, a combination of bleomycin, etoposide, and cisplatin was effective after initial surgery for malignant GCT. In the other, an aromatase inhibitor was effective for recurrent malignant GCT. We also review the literature for further management of this tumor. Because GCT of the ovary is rare, it will be necessary to elucidate the clinical phenotype and establish treatment protocols by accumulating and analyzing more patients. Keywords: Ovarian malignant granulosa cell tumor, BEP combination therapy, Aromatase inhibitor

Background Granulosa cell tumors (GCTs) of the ovary comprise 2 to 5% of malignant ovarian tumors [1,2], and 20 to 30% show malignant clinical and histopathological characteristics. The clinical outcome for tumors at stages I and II is excellent, whereas those at stages III and IV remains poor [3]. The 5-year survival rate for GCTs is approximately 80% overall, and some patients experience relapse 20 to 30 years after the initial surgery [4,5]. There are few reports on the clinical phenotype of malignant progression [6], and no standard therapy has yet been established [7]. We report our experience of two patients with malignant GCT, and review the literature. Case reports Patient 1

A 26-year-old woman presented to her doctor in December 2010 with abdominal pain and bloating. She had never been pregnant, but had a history of surgery for patent ductus arteriosus (at the age of 1 year) and also had type 2 diabetes. A pelvic neoplasm was suspected, and the patient was referred our department in January 2011 for detailed investigation and treatment. * Correspondence: [email protected] Department of Obstetrics and Gynecology, Hirosaki University Graduate School of Medicine, 5-Zaifu-cho, Hirosaki, Japan

Abdominal ultrasonography showed a mass, with hyperechogenicity. On magnetic resonance imaging (MRI) scan, an irregular, solid mass, 130 mm in size, was seen in the peritoneal cavity with associated pelvic ascites. Computed tomography (CT) showed enlargement of the left subclavian, para-aortic, and pelvic lymph nodes, and metastasis was thus suspected. No abnormality was seen with peripheral blood tests and biochemical tests, but the level of a tumor marker, CA125, was raised (233 U/ml; normal range 0 to 35 U/ml). A diagnosis of malignant ovarian tumor was made, and surgery was performed. The laparoscopic findings indicated that the mass was an irregular, solid tumor derived from the left ovary. Because multiple distant metastases were present, a left adnexectomy was performed. The cut surface of the tumor was yellowish and friable (Figure 1). On p