Non-Islet Cell Tumor Hypoglycemia at the Second Recurrence of Malignant Solitary Fibrous Tumor in the Retroperitoneum and Pelvis: A Case Report

Abstract

An 83-year-old man underwent complete resection of a large malignant solitary fibrous tumor in the retroperitoneum in 2006 and of a local recurrent tumor in 2010. In 2011, he was admitted to our hospital because of hypoglycemia. His serum glucose level was very low, and his levels of insulin and C-peptide were low. Furthermore, he had a recurrent tumor in the retroperitoneum and pelvis. Immediately after the third surgery for complete resection, he had no hypoglycemic episodes and his serum glucose level was within the normal range. Immunoblotting analysis revealed a high-molecular-weight form of insulin-like growth factor II in the patient’s serum and in the protein extract obtained from the resected specimen in 2011. To our knowledge, this is the first report of non-islet cell tumor hypoglycemia caused by a malignant solitary fibrous tumor secreting a high-molecular-weight form of insulin-like growth factor II at the second recurrence.

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

“…Several studies have confirmed the secretion of HMW-IGF-II using immunoblotting analyses of perioperative serum [ 15 , 22 , 23 ]. In the present case, an immunoblotting analysis revealed that the strong band of HMW-IGF-II in the preoperative serum had disappeared after tumor resection, which is compatible with the clinical improvement of hypoglycemia after surgery.…”

Late recurrence of a malignant hypoglycemia-inducing pelvic solitary fibrous tumor secreting high-molecular-weight insulin-like growth factor-II: A case report with protein analysis

“…Low IGF-I levels in patients with NICTH are attributed to chronic attenuation of GH secretion, due to the negative feedback of IGF-II ( 9 ). Several studies have shown that NICTH due to HMW-IGF-II can be diagnosed using western blot analysis ( 10 – 12 ). Hata et al ( 10 ) examined HMW-IGF-II by using preoperative and postoperative blots and demonstrated that HMW-IGF-II disappeared postoperatively, which is considered to be a useful therapeutic index.…”

“…Several studies have shown that NICTH due to HMW-IGF-II can be diagnosed using western blot analysis ( 10 – 12 ). Hata et al ( 10 ) examined HMW-IGF-II by using preoperative and postoperative blots and demonstrated that HMW-IGF-II disappeared postoperatively, which is considered to be a useful therapeutic index. In the present case, perioperative western blot analysis was also performed, which revealed the disappearance of HMW-IGF-II, but also the appearance of several unidentified protein bands.…”

“…Additionally, it is possible that certain inoperable cases are encountered, which are due to a poor general condition of the patient, metastasis or severe infiltration to other tissues and organs. Several studies have indicated that the length of time between the first and second recurrence is shorter compared with the time between the initial diagnosis and the first recurrence in frequently-recurring SFTs, regardless of whether surgery was performed ( 7 , 10 ). Baldi et al ( 7 ) suggested that the inherent limitations of surgery may result in unavoidable recurrence or metastasis, since the anatomy of the retroperitoneum and pelvis make complete resection challenging.…”

Integrally calcified solitary fibrous tumor in the retroperitoneum: a case report and review of the literature

“…SFT in the retroperitoneum is rare, and 31 cases have been reported including this case for the last 15 years [ 9 , 14 , 16 , 18 , 20 – 24 , 26 – 42 ] (Table 1 ). The English language literature was extracted from PubMed from 2000 to 2015 using the following Medical Subject Heading terms: “solitary fibrous tumor” and “retroperitoneum”.…”