Non-islet cell tumor hypoglycemia associated with recurrent carcinosarcoma of the ovary

Abstract

Objective: To describe the first reported case of non-islet cell tumor hypoglycemia (NICTH) associated with carcinosarcoma of the ovary.

Methods: We report the clinical course, imaging, and pathologic findings of our patient and review relevant literature.

Results: A 48-year-old woman had a surgery to remove ovarian masses, which turned out to be carcinosarcoma of the ovary, stage IIIc; however, she declined postoperative adjuvant chemotherapy. Six months later, she became unconscious with severe hypoglycemia. A large pelvic mass was found and thought to represent a recurrence. Serum insulin and C-peptide were undetectable. Morning cortisol was mildly elevated. Thyroid stimulating hormone, amylase, lipase, and renal and hepatic functions were normal. While insulin-like growth factor (IGF)-I was low, IGF-II was inappropriately elevated. Increased IGF-II/IGF-I ratio was suggestive of NICTH in light of the large pelvic tumor. She required frequent meals, dextrose boluses, and continuous infusions, oral prednisone, and glucagon continuous infusion to prevent recurrent hypoglycemic attacks. Chemotherapy with carboplatin and paclitaxel was initiated, and glucose control started to improve. After 4 cycles of the chemotherapy, the tumor regressed substantially and was surgically removed. She had 3 more cycles of postoperative chemotherapy. Although the reported median survival of this aggressive neoplasm is less than 2 years, this patient has been free of recurrent disease and hypoglycemia for 6 years.

Conclusion: This is the first study to report NICTH in a patient with carcinosarcoma of the ovary. Clinicians should be aware of NICTH as a cause of hypoglycemia especially in a patient with a tumor or history of tumor.