Ovarian Dysgerminoma and Synchronic Contralateral Tubal Pregnancy Followed by Normal Intra-uterine Gestation

A Case Report

Lourdes Montesinos; Pedro Acién; Monserrat Martínez-Beltrán; María-José Mayol

Disclosures

J Med Case Reports. 2012;6(399) 

In This Article

Case Presentation

A 24-year-old nulliparous Spanish woman first presented seven years ago to the obstetric-gynecologic emergency service (OGES) of our hospital with a history of pelvic pain, weight loss, metrorrhagia after menstrual delay, urinary infection, positive β-hCG test result and the finding of a right ovarian tumor upon examination.

She underwent menarche at age nine, had frequent menstrual delays and had been taking oral contraceptives (OC) since age 17. She had a history of tobacco and drug (not parenteral) use. Prior to her presentation, she had not undergone regular gynecological exams but had presented several times to the OGES. Two years earlier, she presented after two months of amenorrhea. A pregnancy test was negative, and a transvaginal ultrasound examination (TVU) showed a 2cm anechoic tri-chamber image in the right ovary. Her blood sedimentation rate (BSR) was normal at 14mm (normal value (nv), <25mm), and the following tests for tumor markers were negative: β-hCG <1.2mU/mL (nv, 0 to 5mU/mL); cancer antigen (CA)-19-9 15.87U/mL (nv, 0 to 40U/mL); carcinoembryonic antigen (CEA) 0.97ng/mL (nv, 0 to 5ng/mL); CA-125 14.53U/mL (nv, 0 to 35U/mL); α-fetoprotein (α-FP) 1.05ng/mL (nv, 0 to 15ng/mL). Our patient returned to the OGES for pelvic pain and urinary symptoms six months later. The results of the TVU exam and the blood work were similar to those from our patient's previous presentation (BSR 17mm, CA-19-9 17.76U/mL, CEA 0.95ng/mL, CA-125 12.23U/mL). Again, our patient did not attend for subsequent outpatient follow-up.

When she returned to OGES seven years ago she presented with persistent metrorrhagia and symptoms of a urinary infection, as mentioned above. A pregnancy test was weakly positive and an altered urinary sediment confirmed urinary tract infection. A TVU did not reveal an intra-uterine pregnancy, but a right adnexal hyperechoic dotted mass was observed. It appeared to be heterogeneous and of medium density and measured 7×6cm. Our patient underwent repeat blood analysis and her tumor markers were as follows: BSR 29mm, β-hCG 105U/mL, CA-19-9 15.34U/mL, CEA 0.99ng/mL, CA-125 13.73U/mL, α-FP 1.06ng/mL. She was told to take urinary antibiotics, repeat the analysis and return to our outpatient center two weeks later. She reported experiencing weight loss and minor metrorrhagia for one month and had discontinued her OC four months prior to her visit. A vaginal examination showed a normal uterus and a large right adnexal tumor extending from the Douglas cul-de-sac to the right iliac fossa. A TVU exam showed a normal uterus with no evidence of endometrial pathology, a normal left ovary, low to moderate ascites and a large pelvic tumor. The tumor measured over 13cm, was poly-lobed and had heterogeneous densities with increased vascularity, arising from the right ovary. An endometrial biopsy was performed and revealed secretory changes without villi or trophoblastic elements. Repeated blood tests showed the following: a normal hemogram, BSR 8mm and normal blood chemistry with the exception of lactate dehydrogenase (LDH) 2349U/L (nv, 135 to 214U/L) and β-hCG 242U/mL; α-FP, CA-19-9, CEA and CA-125 levels were normal. A chest radiograph was normal, and abdominal and pelvic computed tomography (CT) and Doppler sonography showed a solid right ovarian mass of 13×8cm with diffuse vascular mapping with low resistance index and fluid in the Douglas cul-de-sac (Figure 1).

Figure 1.

Imaging studies. (A) Uterus and hemoperitoneum transvaginal ultrasound images. (B) Right ovarian tumor and Doppler ultrasonography showing a diffuse vascular mapping with low resistance index. (C) Right ovarian tumor on pelvic computed tomography scan. (D) Tumor and uterus on pelvic computed tomography scan.

With the diagnosis of a right ovarian tumor compatible with a germ cell tumor, which was thought likely to be a dysgerminoma, laparotomy was performed three weeks later. During the laparotomy procedure, we found a solid, poly-lobed and irregular right ovarian mass of roughly 14cm in size, as well as an ectopic pregnancy in the ampullar region of the left tube (Figure 2). There was a small amount of hemoperitoneum, approximately 50 to 100cm3. A right adnexectomy and a left linear salpingostomy were performed. Her postoperative course was uncomplicated and she had menstruation after surgery.

Figure 2.

(A) Laparotomic observation. Right ovarian tumor and uterus. (B) After right adnexectomy: left tubal pregnancy, left ovary and uterus. (C) Ovarian tumor and tissue from ectopic pregnancy.

The histopathologic diagnosis was as follows: (1) left tubal ectopic pregnancy; (2) negative results for peritoneal fluid cytology; and (3) right ovarian dysgerminoma 12cm in size, with areas of tumor necrosis and infiltration of the tunica albuginea but with an unruptured capsule. Histopathological images are shown in Figure 3. A normal residual ovary was identified with the presence of the corpus luteum. Immunohistochemical analysis gave the following results: keratin was weakly positive; desmin, vimentin, and neuron specific enolase were all negative; and the proliferative index was high (>50 percent). The right fallopian tube had a normal appearance. On absence of peritoneal tumor spread on thorough inspection, the International Federation of Gynecology and Obstetrics (FIGO) stage was likely consistent with a stage IA.

Figure 3.

Histopathological images. (A) Dysgerminoma, 20×, hematoxylin and eosin stain. (B) Chorial villi tissue and fibrinohemorrhagic material, 4×, hematoxylin and eosin stain.

After discussing the case at our Gynecological Tumors committee, it was decided to administer three chemotherapy cycles in spite of likely stage IA dysgerminoma due to no biopsy of pelvic and para-aortic lymph nodes, with our patient transferred to the medical oncology service. However, a new positive pregnancy test postponed the start of chemotherapy. Our patient returned to the OGES five weeks after surgery because she had experienced no menstruation, her β-hCG was 588U/mL, and a TVU confirmed the presence of an early intra-uterine pregnancy. Our patient was informed of the situation, and she decided to continue the pregnancy. A new TVU confirmed the presence of a normal intra-uterine pregnancy of gestation five to six weeks, with a normal yolk sac and embryo button. Four weeks later the embryo measured 24mm, which is consistent with a gestational age of roughly nine weeks, indicating that our patient became pregnant less than three weeks after surgery. The pregnancy evolution was normal, although there was some delay of fetal growth at the end of gestation. Our patient gave birth to a baby boy, born at term with a weight of 2600g. Chemotherapy was subsequently dismissed.

Our patient moved to another city and again became pregnant. She delivered one year later at the Baza Hospital via Caesarean section due to intra-uterine growth retardation and fetal distress. From that point to the present day, our patient has been followed up in our hospital Oncology and Gynecology departments. She remained asymptomatic and with normal test results (β-hCG negative; LDH = 302U/L in 2009, 155U/L in 2010 and 150 in 2011 and 2012; α-FP and other tumor markers were all normal) and normal imaging studies (TVU and CT).

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