Luteinized fibrothecomas of the ovary associated with sclerosing peritonitis in a patient with systemic lupus erithematosus
© Springer-Verlag 2007
Received: 24 October 2006
Accepted: 5 April 2007
Published: 1 June 2007
Sclerosing peritonitis is a peritoneal subserosal fibrosis that has been associated with luteinized thecomas of the ovary. Ascites, peritoneal thickening and adnexal masses were found in a 38-year-old woman. She had been an intravenous drug abuser and had systemic lupus erytematosus. At laparotomy the association of peritoneal diffuse fibrosis and bilateral luteinized fibrothecomas was diagnosed. The postoperative course was fatal due to small bowel obstruction. The concomitant finding of ascites, peritoneal thickening and adnexal masses suggested the diagnosis of ovarian cancer. Although the patogenesis is unknown, predisposing causes of sclerosing peritonitis have been described such as intravenous drug abuse and systemic lupus erithematosus.
KeywordsOvarian luteinized fibrothecomas Ovarian fibrothecomas Luteinized fibrothecomas Sclerosing peritonitis Systemic lupus erithematosus
Sclerosing peritonitis (SP) is a peritoneal subserosal fibrosis that has been associated with luteinized thecomas of the ovary (LTO) [1, 2]. It has also been described associated with certain drugs and illnesses, and as a complication of chronic ambulatory peritoneal dialysis . Some authors have suggested the association of SP and autoimmune diseases such as systemic lupus erythematosus (SLE) . Most of the cases described were, at first, misdiagnosed as disseminated peritoneal malignancy from an ovarian carcinoma [1, 2]. In this paper we report the first case of this rare association in a patient with SLE.
A 38-year-old non-gravida woman was admitted to the hospital complaining of acute abdominal pain, nausea and abdominal swelling. She also complained of weight loss of 7 kg over the past year. Ten years ago she had been an intravenous drug abuser, and she also had SLE.
The postoperative course was complicated by small bowel obstruction that improved with conservative management. Three months after hospital discharge the patient died due to small bowel obstruction and extensive peritoneal adhesions although glucocorticoid treatment and new surgical interventions had been performed.
SP is a rare inflammatory process that leads to the deposition of a thick fibrous membrane on the peritoneum. The serosa of the small bowel is the most frequently affected site and clinical manifestations are usually abdominal mass and/or small bowel obstruction .
In our case the concomitant finding of ascites, peritoneal thickening and adnexal masses strongly suggested the diagnosis of ovarian cancer. Although the pathogenesis is unknown, predisposing causes of SP have been described [1–3]. The patient’s case report had two predisposing causes of SP: she had been an intravenous drug abuser and she had SLE.
SP treatment should be conservative surgical removal of the thickened peritoneum, lysis of adhesions, and partial resection of the small bowel. When associated with LTO the treatment must include the removal of the tumor/s. Glucocorticoid treatment has been tried in another case associated with LTO with favourable results avoiding the surgery . Most patients require more than one surgical intervention as the postoperative course is usually complicated with the formation of new fibrosis and more adhesions [1, 2].
In summary, we present a new case of bilateral LTO with SP that differs from those previously reported by having several possible predisposing causes of SP (SLE and being a past drug abuser). The possible diagnostic confusion with malignancy, the way the illness can degrade the quality of life, and the potentially lethal surgical complications emphasize the importance of increasing awareness of this unusually recognized entity.
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