Growing Teratoma Syndrome with Synchronous Gliomatosis Peritonei during Chemotherapy in Ovarian Immature Teratoma: A Case Report and Literature Review

Abstract

Coexistent growing teratoma syndrome (GTS) and gliomatosis peritonei (GP) arising during chemotherapy of ovarian immature teratoma (IMT) is extremely rare and can be misdiagnosed as recurrent or progressive disease. We present a 33-year-old woman diagnosed with GTS with synchronous GP during chemotherapy of IMT. She underwent ovarian cystectomy due to ovarian immature teratoma and chemotherapy were administered. The α-fetoprotein (AFP) concentration decreased from 28.7 ng/mL to normal after the second cycle. Four days after the third cycle of chemotherapy, ultrasound and CT revealed an 8-cm mass with negative tumor markers in the pouch of Douglas. An exploratory laparotomy was conducted, and a smooth round cystic-solid 8-cm mass was noted in the pouch of Douglas. Extensive peritoneal seeding glial nodules were also observed on the surface of the uterus, peritoneum, and omentum. The patient underwent a partial omentectomy, intact resection of the tumor, and resection of most of the glial nodules. Postoperative pathology demonstrated a pure mature cystic teratoma component in the mass, as well as diffuse GP involving the uterine serosa, peritoneum, and omentum; this diagnosis of GTS with synchorous GP should be considered in IMT patients with mass newly identified during chemotherapy while tumor markers are normal after treatment.

Keywords: gliomatosis peritonei; growing teratoma syndrome; immature teratoma; ovarian tumors; treatment.

Figure 1

Paraffin pathology confirmed that most of the right ovarian tumor was composed of mature teratoma with focal primitive neuroepithelial elements (HE staining, 20×).

Figure 2

Ultrasonic features of the mass. (A) A multilocular solid-cystic mass with a clear boundary was seen in the left adnexal area, in which lipid and hair-like hyper-echo mass was also observed. (B) Calcification can be seen in solid hypo-echo of the mass. (C) The CDFI revealed scattered, short strips of blood flow signals in the solid proportion of this mass. (D) Implant nodules can be seen on the intestinal surface.

Figure 3

A 9.5 × 6.2 cm mass was noted left posterior of the uterus with an enhancement of solid component but not cystic component, no enlarged retroperitoneal lymph node was noted (arrow, calcification).

Figure 4

Intraoperative views during exploratory laparotomy demonstrated a small myoma located at the posterior uterine wall, and multiple glial tissue (“GP”) seeding was noted at the uterine surface, vesical peritoneal reflection. The mass was located in the pouch of Douglas, with most proportion obscured by the uterus (RO, right ovary).

Figure 5

The tumor showed a smooth and irregularly lobulated gross appearance.

Figure 6

Pathology results of peritoneal seeding of GP and GTS (HE staining). Mature neuroglia presented with distinctive rounded, or ovoid nodules with well-defined boundaries involving omentum majus ((A), 200×), vesical peritoneal reflection ((B), 25×), peritoneum of ascending colon ((C), 50×), right ovarian cyst ((D), 50×), and posterior uterine wall ((E), 50×), while mature cartilage and squamous epithelium differentiation were observed in the mass ((F), 50×).