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

Discussion

Dysgerminomas are tumors derived from germ cells, which constitute 3 to 5 percent of malignant ovarian tumors. They are rare tumors that affect younger patients, and treatment should therefore be as unaggressive as possible and aim to preserve fertility. Most dysgerminomas are unilateral and occur predominantly on the right side, but up to 12 percent of cases may be bilateral.[1] The combination of conservative surgery and chemotherapy achieves good results even in advanced stage disease. In the early stages, it is generally recommended that the patient undergo conservative treatment (unilateral salpingo-oophorectomy; USO) and platinum-based adjuvant chemotherapy. However, with stage IA tumors treatment with surgery alone is likely sufficient. Nevertheless, dysgerminoma of the ovary has a propensity to metastasize to the pelvic and para-aortic lymph nodes in the absence of other evidence of metastatic disease; biopsies of these structures are particularly important.[2] In a recent review of dysgerminomas of the ovary, Vicus et al.[3] reported that from 65 cases in their institution during a period of 35 years, 38 (58.5 percent) cases presented with stage IA disease and in five of them (13.1 percent) recurrences occurred after performing USO (unstaged), all within 19 months of the primary diagnosis. However, none of the 15 patients who received adjuvant treatment experienced tunorrecurrence, and three women who had received adjuvant chemotherapy had successful pregnancies. Shamim[4] also reported a case of a 30-year-old woman presenting with a successful pregnancy outcome after fertility-sparing surgery and chemotherapy for dysgerminoma. Our patient has done well without adjuvant treatment.

Diagnosing ovarian germ cell tumors is challenging.[5] Following clinical examination and imaging tests (ultrasound, CT and magnetic resonance imaging (MRI)), the finding of increased serum levels of β-hCG, α-FP and LDH suggest the diagnosis pre-operatively. Although there is considerable variation in the production of these markers, the majority of endodermal sinus tumors produce α-FP, and most choriocarcinomas and dysgerminomas produce β-hCG and LDH respectively. However, some dysgerminomas also produce β-hCG.[1]

The case presented here had the unusual coincidence of being associated with a tubal pregnancy, which increased β-hCG and provoked a slight hemoperitoneum, thus raising doubts about the diagnosis. In fact, once the ovarian tumor had been diagnosed, the tubal ectopic pregnancy was not suspected and was only found at the time of surgery. Interestingly, the corpus luteum was present in the right ovary, as was the dysgerminoma, while the ectopic pregnancy was implanted in the contralateral tube. It is possible that the tumor favored the ectopic pregnancy on the contralateral side (external hyperemigration). In the literature, we have found few cases of tubal pregnancy associated with ovarian carcinoma and only one associated with dysgerminoma.[6] This case concerned a patient who presented with a dysgerminoma in the left ovary and a right interstitial pregnancy; a corpus luteum was present in the ovary with the dysgerminoma. An emergency laparotomy was performed due to accidental breakage of the ectopic pregnancy with acute hemoperitoneum. The treating clinicians performed left adnexectomy and cuneiform resection of the right uterine horn with salpingectomy. The authors reviewed the literature and did not find any other similar cases. We were also unable to find any similar published cases at present.

There are cases of gonadoblastoma and tubal pregnancy,[7,8] and there is a series of 23 cases of ovarian carcinoma during pregnancy from Dgani et al.,[9] including four dysgerminomas at stage IA associated to intra-uterine gestation. There were three cases of ovarian carcinoma and right tubal pregnancy, but two were serous carcinomas and one was a granulosa tumor; all three patients had advanced illness.

In our patient's case, it is also interesting that after the right adnexectomy and the left salpingostomy, which were performed during laparotomy, our patient became pregnant with a normal intra-uterine pregnancy and carried it to term; she later also had another normal pregnancy. It is clear that despite the surgery that was performed, our patient's fertility was effectively preserved. However, our report on the challenges of diagnosis and treatment in this case can help clinicians to better understand and manage these pathologies.

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