A True Splenic Cyst Producing Carbohydrate Antigen 19-9 and Cancer Antigens 50 and 125, but Not Interleukin 10

Abstract

A giant cystic lesion of the left upper abdomen associated with a smaller ovarian cyst in a young female patient is reported. Laboratory data revealed elevated serum levels of carbohydrate antigen 19-9 (CA 19-9), carcino-embryonic antigen (CEA), cancer antigens 50 and 125, and tissue polypeptide antigen. In contrast, the serum levels of interleukin 10, a cytokine involved in modulating immune responses and produced by many cancer histotypes, were normal. Since ovarian cancer or cystic adenocarcinoma of the tail of the pancreas were not ruled out, the patient underwent laparotomy. After splenectomy and ovariectomy, the tumour marker serum levels normalized. Histology and immunohistochemical analysis revealed a true splenic cyst with the inner epithelium strongly positive for CA 19-9 and CEA and high levels of cancer antigens in the fluid. The ovarian lesion was a serous cystadenoma. The inner epithelium showed no immunoreactivity for tumour markers which were not measurable in the fluid. True cysts of the spleen are rare; in a few cases, high serum levels of CA 19-9 and CEA have been reported. In such instances, cyst resection or splenectomy is indicated to rule out malignant lesions and to remove the cancer antigen producing epithelium. The reported case shows that the epithelium lining true splenic cysts may produce, besides CA 19-9 and CEA, other tumour markers, in particular cancer antigens 50 and 125. In addition, normal serum values of interleukin 10 correctly predicted the benign nature of the lesion.

CEA in Evaluation of Adnexal Mass: Retrospective Cohort Analysis and Review of the Literature

“…Abnormal CEA levels may mislead the diagnosis, as several benign disorders involving increased CEA were described in the literature, including rheumatoid arthritis (20), appendiceal mucocele (57), true splenic cyst (58) and a case report of a patient with bronchial asthma and mucoid impaction (59). Sasaki et al have suggested that CEA assessment could serve as a prognostic factor in predicting the future regression of simple ovarian cysts (60), and Pinto et al have shown that combination of CEA and Ca125 measurements could be helpful in the distinction of functional from epithelial ovarian cysts (61).…”

Spontaneous Rupture of a True Splenic Cyst Diagnosed by Laparoscopy

“…In addition, true cysts occur mainly in children and young adults [19], with approximately two-thirds of patients being female [20]. In the literature, approximately 190 reports pertaining to true splenic cysts have been documented, and of these, only a few describe spontaneous cyst rupture [20][21][22][23][24][25][26][27]. The origin of epithelial [true] cysts of the spleen is controversial.…”

“…Some authors attribute their development to the embryonic inclusion of epithelial tissue, whereas others refer to the inclusion of the mesothelial lining with subsequent squamous metaplasia [28,29]. In addition, there are recent reports in the literature describing high serum levels of tumor markers (CA 19-9, CEA, cancer antigens 50 and 125) [23][24][25]. These reports indicate that the production of CEA, CA 19-9, and, to a lesser extent, cancer antigens 50 and 125 is dependent on the epithelium lining of the true cysts.…”

“…The lowering of the serum levels of the tumor markers observed after cyst removal demonstrates the direct relationship between the increment of the serum tumor markers and the presence of the epithelial cyst [22,26]. On the other hand, the benign nature of the lesion may be predicted if normal serum levels of interleukin 10 (a cytokine involved in modulating the immune response and is produced by many cancer histiocytes) are found [23][24][25].…”