Spontaneously Ruptured Giant Splenic Cyst with Elevated Serum Levels of CA 19-9, CA 125 and Carcinoembryonic Antigen

Abstract

Splenic cyst is a relatively rare disease; however, the occurrence of complications associated with its rupture is even more rare. A 20-year-old female patient who had severe abdominal and left shoulder pain was admitted to our hospital. The patient’s abdomen was hard and tender to the touch and she presented with a high fever. The patient’s serum levels of the tumor markers carbohydrate antigen 19-9, cancer antigen 125 and carcinoembryonic antigen were high. Ultrasonography and computed tomography of the abdomen showed an 11-cm multilocular cystic lesion in the spleen and the presence of free intraperitoneal fluid. Peritonitis with ruptured splenic cyst was diagnosed, and the patient underwent an emergency laparotomy. The abdominal cavity was filled with purulent fluid. The cyst was localized to the spleen and had already ruptured. Total splenectomy and cyst resection were performed. The postoperative course was uneventful. The patient was discharged on day 9 following surgery. The histological findings showed the lesion to be a benign epidermoid cyst completely lined with inner stratified squamous epithelium with a capsule of connective tissue. In the immunostaining analyses, the squamous epithelium was positive for carcinoembryonic antigen. A ruptured splenic cyst causes sudden onset of severe peritonitis and elevation of serous tumor markers. An emergency operation is indicated as the treatment for a ruptured splenic cyst with peritonitis, after which a favorable outcome can be expected.

A diaphragmatic retroperitoneal cyst

“…4,5 Similar to our case, the CA19-9 returns to normal levels following resection of the cyst. A cyst in this location with raised CA19-9 has not been described before.…”

Schwannoma of the stomach with elevated preoperative serum carbohydrate antigen 19-9: report of a case

“…No abnormality of the biliary system or pancreatic duct system was shown by a preoperative magnetic resonance imaging examination in this case (data not shown). On the other hand, some reports have described high CA19-9 levels in patients with uncommon diseases, such as retrorectal cystic hamartoma, mediastinal mature teratoma, and splenic cysts [10][11][12]. Moreover, these surgically removed tumors were positive for CA19-9 by immunohistochemistry, and the serum levels decreased to the normal range about 1 month after tumor resection.…”

Huge splenic epidermoid cyst with elevation of serum CA19-9 level

“…In particular, cysts sized larger than 10 cm have a high risk of rupture (10), and therefore splenectomy should be performed. Moreover, when a malignant tumor is suspected, total splenectomy should be chosen because there have been some reports of splenic cyst accompanied with malignancy (11,12).…”