Sclerosing peritonitis associated with luteinized thecoma and elevated serum CA 125 levels: case report

Abstract

CONTEXT: Thecomas are benign tumors that account for less than 1% of all ovarian neoplasms. The association of ovarian thecoma with sclerosing peritonitis is rare. CASE REPORT:We report the case of a 33-yearold woman, with a two-month history of increasing abdominal volume. Ultrasound showed a complex pelvic lesion and laboratory analysis detected elevated serum CA 125 levels. The patient underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy and peritoneal biopsy. Histopathological analysis revealed the presence of luteinized thecoma of both ovaries associated with sclerosing peritonitis.KEY WORDS: Thecoma. Peritonitis. Ovarian neoplasms. CA-125 antigen. Germinoma. INTRODUCTIONThecomas are rare neoplasms that usually occur in postmenopausal women. Luteinized thecoma of the ovary is characterized by a group of polygonal cells with clear, vacuolated and abundant lipid-fi lled cytoplasm linked to tumor estrogen activity.1 This neoplasm may be associated with multiple fi brotic nodular thickening of the peritoneum, which is named sclerosing peritonitis. Association of these two conditions is extremely rare and the etiology currently remains unclear. Patients may present with abdominal distension, intestinal obstruction, palpable unilateral or bilateral mass, weight loss, nausea and vomiting. 2The purpose of the present paper is to report on a rare association between thecoma of the ovary and sclerosing peritonitis with increased serum CA 125 levels. CASE REPORTA 33-year-old woman who had had five pregnancies, three deliveries and two abortions, sought medical treatment for a two-month history of increasing abdominal volume and weight loss of 4 kg in a month. On physical examination, a palpable mass was noted in the left iliac fossa with clinically apparent tumor infi ltration in the umbilical scar. Ultrasound revealed a complex pelvic lesion. Measurement of serum CA 125 detected elevated levels (316.9 U/ml). The patient underwent exploratory laparotomy, which showed an ovarian tumor with multiple implants in the peritoneal cavity, suggesting that it was malignant. There was also thickening of the peritoneum in the umbilical scar region.Intraoperative biopsy showed that the tumor was benign. However, based on the clinical and surgical findings, as well as the elevated serum CA 125 levels, we chose to perform total abdominal hysterectomy with bilateral salpingo-oophorectomy, Macroscopically, the outer surface of the right ovary appeared lobulated and grayish-white with granular areas, measuring 15 cm across its largest diameter. The sectioned surfaces of the right ovarian mass were solid, yellow and granular with mucin-containing cystic areas. The outer surface of the left ovary appeared undulating and was grayish-white, measuring 3.8 cm in its largest diameter. Sections through the left ovarian mass revealed a fi brillar and grayish-white surface.Histological examination of the ovaries identifi ed a benign tumor composed of rich fi bromatous stroma with rare isolated lutein cells and low mitotic...

Sex Cord–Stromal Tumors of the Ovary: An Update and Review. Part I — Pure Ovarian Stromal Tumors

“…8). Since then, only a limited number of cases have been reported in the literature mostly as case reports [143][144][145][146][147][148][149][150][151][152][153][154][155][156][157][158][159][160][161][162] with only one large series of 27 patients providing a comprehensive clinical, morphological, and immunohistochemical characterization of these lesions. 64 Although the term "luteinized thecomas" has prevailed in the literature, it is now favored by some that these are not neoplastic proliferations as they cause an expansion of the ovarian cortex with entrapment of preexisting structures, a feature not typically seen in neoplastic processes, and thus, the alternative term of thecomatosis has been proposed to denote its possible non-neoplastic nature.…”

“…Some patients have had a prior history of anticonvulsant or propranolol therapy or autoimmune disease, 64,150,151 and a few elevated CA-125 serum levels. 153 Rarely extraperitoneal involvement has been described including cutaneous 165 and extraperitoneal lesions. 148 Patients suffer from complications related to their peritoneal disease, most often bowel obstruction that in the past resulted in high morbidity and up to 30% mortality; symptoms may develop after the resection of ovarian lesions and may persist for months.…”

“…Hormonal manifestations are rare. Some patients have had a prior history of anticonvulsant or propranolol therapy or autoimmune disease, 64,150,151 and a few elevated CA-125 serum levels 153 . Rarely extraperitoneal involvement has been described including cutaneous 165 and extraperitoneal lesions 148 .…”

Sclerosing Peritonitis Associated With a Luteinized Adult Granulosa Cell Tumor

“…SP is a benign condition but patients may develop ascites, abdominal pain, or small bowel obstruction (5,6). Recurrent small bowel obstruction and surgical complications can cause significant morbidity and are potentially fatal (5)(6)(7)(8)(9)(10)(11).…”