A rare case of recurrent ovarian cancer presenting as a round ligament metastasis
© Togami et al; licensee BioMed Central Ltd. 2011
Received: 19 August 2011
Accepted: 7 November 2011
Published: 7 November 2011
We report a rare case of recurrent ovarian cancer presenting as a round ligament metastasis. A 44-year-old woman presented with a lower abdominal mass. Computed tomography showed a pelvic mass. Primary surgery was performed. A histopathological examination showed an ovarian serous adenocarcinoma of Stage IIIb. The patient received 6 cycles of paclitaxel and carboplatin. Almost 2 years after the initial operation, the patient noticed a left inguinal mass. Computed tomography showed a left inguinal mass, 18 mm in size. An excisional biopsy was performed and the tumor was found to originate in the left round ligament. A histopathological examination showed serous adenocarcinoma and there was no evidence of lymph node tissue. Recurrence of ovarian cancer in the round ligament is extremely rare. This unique case suggests, however, that the round ligament in rare cases may be a recurrence site for ovarian cancer, and that accurate differentiation including confirmation by diagnostic imaging and excisional biopsy, is necessary for a definitive pathological diagnosis.
The majority of women with ovarian cancer present with advanced stage disease. A complete clinical remission after surgical cytoreduction and platinum-based chemotherapy can be achieved in 80-90% of these patients. Despite this, 70-90% of patients will develop recurrent disease . Fifty-five percent of the first relapse cases were found at the pelvis or abdomen . There was a wide variety among the other recurrent sites, such as, retroperitoneal nodes, liver or spleen, brain, and bone [2, 3]. We experienced a case with solitary recurrence at the left round ligament. To the best of our knowledge, this is the first report of recurrent ovarian cancer occurring in the round ligament.
Initially, the patient received 6 cycles of paclitaxel (175 mg/m2) and carboplatin (AUC = 6) for ovarian cancer in April 2009. In August 2009, her serum CA125 levels declined to 5 U/ml. She then received adjuvant radiotherapy (WB 50Gy/25 f) for breast cancer in October 2009. After that, she received routine follow-up from her gynecologic oncologist.
The majority of patients with ovarian cancer responds well to the initial treatment, but most of them will develop recurrent disease . Recurrent disease involves most frequently the pelvis or the abdomen [2, 3]. This case demonstrates a most unusual recurrence for ovarian cancer, presenting as a round ligament metastasis. The round ligament extends from the uterus, through the inguinal canal, and ends in the region of the mons pubis and labia majora. Embryologically, this is the female equivalent of gubernaculums testis and is predominantly composed of smooth muscle fibers, connective tissue, blood vessels, and nerves with a mesothelial coating .
Because round ligament recurrence of ovarian cancer is very rare, we performed a MEDLINE search of the English language literature, but no example could be found. Some unusual tumors involving the round ligament have been reported in the literature: dermoid cyst, endometriosis, mesothelial cyst, leiomyoma and leiomyosarcoma [6–8]. These tumors of the round ligament are very rare developmental disorders, which have been reported as case reports. Especially, leiomyoma is the most common tumor associated with the round ligament . Patil et al.  reported the clinicopathological features of 55 cases of women with inguinal smooth muscle tumors of women. Histologically, 23 tumors were considered as leiomyomas, and five tumors arose in the round ligament. In contrast to the leiomyomas, none of the leiomyosarcomas were associated with the round ligament. Indeed, leiomyosarcomas of the round ligament of the uterus are extremely rare and there is only 1 case report published of leiomyosarcoma arising in the round ligament of the uterus . Recurrent tumors of the round ligament are also rare. We found 1 case report of recurrent endometrial cancer stage Ia, originating in the round ligament . The 5 cm solid mass was located near the right superficial inguinal ring. Resection of the round ligament mass and dissection of the retroperitoneal node were performed, and one positive obturator node was found. They concluded that patients presenting with a round ligament recurrence should have a thorough work-up with pelvic lymph node evaluation. In our case, CT showed no evidence of pelvic lymph node swelling, so we performed only the round ligament mass resection to confirm the pathological diagnosis. Postoperative PET/CT scan revealed no hot spot, showing that metastasis to the round ligament was solitary.
Two possible pathways of metastasis to the round ligament have been considered: The lymphatic and the vascular pathway. Pathologically, the tumor showed vascular infiltration but no evidence of lymph node tissue, but it is difficult to determine whether the metastasis is pathway whether lymphatic or vascular. Ovarian cancer generally metastasizes via the lymphatic system or by peritoneal dissemination . Lymphatic vessels enter and travel along the round ligament to reach the inguinal region, and the most likely hypothesis is that the microscopic tumor metastasized to the round ligament through a lymphatic pathway.
There are some reports of solitary splenic metastasis of ovarian cancer after surgical remission [11–13]. In terms of treatment, splenectomy was performed in all cases and adjuvant chemotherapy was administered in most cases. The decision as to whether adjuvant chemotherapy is indicated must be carefully considered in each case.
This case presents an unusual example of a recurrence site for ovarian cancer. Although solitary ovarian cancer recurrence at the round ligament is extremely rare, it should be included in the differential diagnosis for any patient with a past history of ovarian cancer. The round ligament has the potential to be a site of occurrence of various tumors. Among them, benign tumors such as leiomyoma are often seen in the round ligament. This unique case suggests that the round ligament in rare cases may be a site of recurrence in ovarian cancer, and indicates that accurate differentiation, including confirmation with diagnostic imaging and excisional biopsy, are necessary because the subsequent treatment depends significantly on the pathological results.
Informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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