A giant pelvic solitary fibrous tumor with Doege–Potter syndrome successfully treated with transcatheter arterial embolization followed by surgical resection: a case report

Abstract

Background Solitary fibrous tumor (SFT), a mesenchymal fibroblastic tumor with a hypervascular nature, rarely develops in the pelvis. Resection of a giant SFT occupying the pelvic cavity poses an increased risk of developing massive hemorrhage during resection, although surgical resection is the most effective treatment method for this tumor to achieve a potential cure. SFT rarely develops with Doege–Potter syndrome, which is known as a paraneoplastic syndrome characterized by non-islet cell tumor hypoglycemia (NICTH) secondary to SFT that secretes insulin-like growth factor-II (IGF-II). We present a case of a giant pelvic SFT with Doege–Potter syndrome, which was successfully treated with transcatheter arterial embolization (TAE) followed by surgical resection. Case presentation A 46-year-old woman presented with a disorder of consciousness due to refractory hypoglycemia. Images of the pelvis showed a giant and heterogeneously hypervascular mass displacing and compressing the rectum. Endocrinological evaluation revealed low serum levels of insulin and C-peptide consistent with NICTH. Angiography identified both the inferior mesenteric artery and the bilateral internal iliac artery as the main feeders of the tumor. To avoid intraoperative massive bleeding, super-selective TAE was performed for the tumor 2 days prior to surgery. Hypoglycemia disappeared after TAE. The tumor was resected completely, with no massive hemorrhage during resection. Histologically, it was diagnosed as IGF-II-secreting SFT. Partial necrosis of the rectum in the specimen was observed due to TAE. The patient was followed up for 2 years and no evidence of disease has been reported. Conclusions Preoperative angiography followed by TAE is an exceedingly helpful method to reduce intraoperative hemorrhage when planning to resect SFT occupying the pelvic cavity. Complications related to ischemia should be kept in mind after TAE, which needs to be planned within 1 or 2 days before surgery. TAE for tumors may be an option in addition to medical and surgical treatment for persistent hypoglycemia in Doege–Potter syndrome.

Magnetic resonance imaging features of a solitary fibrous tumor of the vulva: a case report

“…The promising results obtained so far warrant additional investigations. 37 , 38 Notably, the treatment of vulvar SFT still carries a risk of intraoperative blood loss and postoperative complications including surgical site infection and anal dysfunction 39 ; however, our patient did not develop these complications.…”

Case report: A giant lung leiomyosarcoma: From an accurate diagnosis to a successful surgery. A rare case and brief literature review

“…Preoperative TAE proved to be of paramount importance to prevent intraoperative bleeding. According to the literature, TAE can be considered a successful and effective technique in preventing intra- and postoperative blood loss, as demonstrated by Yuza and colleagues in surgical excision of giant pelvic solitary fibrous tumors ( 11 ). TAE has proved to be a powerful weapon for avoiding massive bleeding also in oncological orthopedic surgery, such as in resection and reconstruction of long bone hypervascular tumors ( 12 ).…”

Successful Surgical Treatment of a Recurrent Pelvic Solitary Fibrous Tumor of Uterine Origin Accompanied by Doege-Potter Syndrome: A Case Report

“…However, repeated surgeries for recurrent SFTs or too-large tumors can cause distressful adhesion, and complete surgical resection is considerably challenging [ 15 ]. To avoid a positive surgical margin and reduce operative blood loss, several studies have reported the utility of preoperative transcatheter arterial embolization, as SFTs are usually hypervascular tumors [ 5 , 25 ]. In our case, the main pelvic tumor had grown so large that the tumor had severe adhesion to the rectum and the left ureter, so it might have been wise to consider preoperative transcatheter arterial embolization followed by surgery.…”