A Case of Malignant Solitary Fibrous Tumor of the Retroperitoneum with Hypoglycemia

Yoshinori Morimoto¹⁾²⁾, Hiromi Iwagaki²⁾, Sadayuki Horiki²⁾, Kazuyuki Kawamoto¹⁾, Mototaka Niwano¹⁾, Tepunn Paku¹⁾, Yasuo Yoshida¹⁾, Tadashi Ito¹⁾, Keizo Ogasawara¹⁾ and Noriaki Tanaka²⁾

Department of Surgery, Kurashiki Central Hospital¹⁾
Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine and Dentistry²⁾

A 19-year-old man admitted for an early morning unconsciousness attack in April 1995 had a serum glucose level of 41 mg/dl, insulin of 0.4 μU/ml, insulin-like growth factor (IGF)-I of 20 ng/ml, IGF-II of 640 ng/ml, and an IGF-II/IGF-I ratio of 32. Abdominal computed tomography (CT) showed a large well-defined 13 cm tumor in the right pelvic cavity. A laparotomy based on a diagnosis of retoroperitoneal tumor with nonislet-cell tumor hypoglycemia, showed that the tumor originated in the pelvic retroperitoneum and was fed by the right internal iliac artery. The encapsulated resected specimen was 13.5×11.0×5.4 cm. The cross-section showed yellowish white solid and multicystic components with hemorrhaging and necrosis. Histologically, patternless arrangements of spindle cells were seen against a collagenous background. Neoplastic cells stained positive for CD34, and mitotic cells were seen in 4/10 high-power fields. Histology indicated a malignant solitary fibrous tumor (SFT) of the retroperitoneum. After resection, problems with hypoglycemia ceased. Since surgery, he developed local recurrence, and multiple metastases to the lung, thyroid, bone, liver, and brain gradually detected. Despite surgical resection, chemotherapy, and radiotherapy, he died 11 years postoperatively. We discuss malignant SFT of the retroperitoneum with hypoglycemia-a rarity in Japan-together with combined therapies for this patient.

Key words
malignant solitary fibrous tumor, nonislet-cell tumor hypoglycemia, retroperitoneum

Reprint requests
Yoshinori Morimoto Department of Surgery, Kurashiki Central Hospital
1-1-1 Miwa, Kurashiki, 710-8602 JAPAN

Accepted
June 27, 2007

To read the PDF file you will need Abobe Reader installed on your computer.