Metachronous contralateral cystadenoma after ovariectomy for
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Although ovarian masses are rare in children and adolescent, representing approximately 1-1.6 % of all neoplasms, metachronous benign ovarian tumors are not seldom reported in this population, but the association between a stromal tumor and metachronous contralateral epithelial tumors has not been previously described.

The girl presented with isosexual precocious puberty (metrorrhagia, telarche) and a large abdominopelvic mass (14x10x16 centimeters) at the age of 5 years and 10 months. Inhibin A (538 ng/L) and B (> 977 ng/L), neuron-specific enolase (NSE – 87.2 mcg/L) and 17OHprogesterone (8.7 nmol/L) were elevated, while estradiol, luteinizing hormone (LH) and follicle-stimulating hormone (FSH) level were normal. The patient underwent a lower midline laparotomy: the mass was originating from the left ovary without evidence of normal tissue. An ovariectomy was performed, together with omentectomy and incidental appendectomy, with no suspicious areas observed in the contralateral ovary and no tumor spillage. Histology revealed a juvenile granulosa cell tumor (JGCT), capsulated and limited to the ovary. Postoperative abdominal ultrasound scan (US), performed quarterly, were normal and laboratory tests revealed a normalized levels of hormones and markers, with clinical remission of the signs of precocious puberty within two months.

One year after surgery, an 18-mm fluid formation in the right ovary, consistent with a follicular cyst, was observed at US scan. Due to the benign sonographic characteristics of the cyst, an initial conservative approach was planned, using repeat US and marker evaluation every six months. At that time, the US showed an enlargement of the cyst (40 mm), characterized by a multivacuolar appearance, with a small portion of healthy ovarian tissue around. A blood test revealed a slightly elevated inhibin A level (3.1 ng/L), while NSE and CA-125 were between normal ranges. Surgical exploration was planned through a mini-invasive approach, using the Alexis Laparoscopic System®, at which time the cyst was resected, sparing macroscopically healthy ovarian tissue. Additional biopsies on the tumor’s bed were performed. Histology demonstrated a completely excised benign mucinous cystadenoma (MCA), with negative Margins. Four months after surgery, an abdominal MRI scan showed a normal-sized right ovary (22x24x16 cm) without suspicious areas of local recurrence. Currently, the patient is alive with no signs of recurrence, two years after the second surgery.

Source: https://www.jpagonline.org/article/S1083-3188(20)30292-8/fulltext?rss=yes
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