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Non-Islet Cell Tumor Hypoglycemia Associated With Recurrent Carcinosarcoma Of The Ovary
Institution:1. The John A. Hartford Foundation Center of Excellence in Geriatrics, Department of Geriatric Medicine, John A. Burns School of Medicine, University of Hawaii, the;2. Department of Obstetrics, Gynecology & Women’s Health, John A. Burns School of Medicine, University of Hawaii, the;3. Kuakini Medical Center, and the;4. Miki Medical Associates, Honolulu, Hawai.;1. Boston Children’s Hospital, Division of Endocrinology, Boston, Massachusetts;2. Case Western Reserve University School of Medicine, Cleveland, Ohio;3. Boston University School of Medicine, Division of Endocrinology, Diabetes, and Nutrition, Boston, Massachusetts.;1. Department of Endocrinology and Metabolism, Cleveland Clinic Foundation, Cleveland, Ohio;2. Department of Quantitative Health Sciences, Cleveland Clinic Foundation, Cleveland, Ohio;1. Department of Gastroenterology, Christian Medical College and Hospital, Vellore, Tamil Nadu, India;2. Department of Endocrinology, Christian Medical College and Hospital, Vellore, Tamil Nadu, India.;3. DDepartment of Radiology, Christian Medical College and Hospital, Vellore, Tamil Nadu, India;4. Department of Surgical Endocrinology, Christian Medical College and Hospital, Vellore, Tamil Nadu, India.;1. Division of Endocrinology, “V. Fazzi” Hospital, Lecce, Italy;2. Endocrine Unit and Thyroid Diseases Center, IRCCS-Arcispedale Santa Maria Nuova, Reggio Emilia;3. Department of Endocrinology and Metabolism, Thessaloniki, Greece;4. Department of Pathology,“V. Fazzi” Hospital, Lecce, Italy;5. Institute of Pathology, Locarno, Switzerland;6. Department of Nuclear Medicine and Thyroid Centre, Oncology Institute of Southern Switzerland, Bellinzona, Switzerland;7. Department of Pathology, IRCCS Arcispedale Santa Maria Nuova, Reggio Emilia, Italy;1. Department of Endocrinology, The Children’s Hospital of ZheJiang University School of Medicine, HangZhou, China.;1. Division of Endocrinology, Diabetes, and Metabolism, Department of Medicine, The University of Jordan, Jordan University Hospital;1. Department of Family and Preventive Medicine, University of California, San Diego, La Jolla, California
Abstract:ObjectiveTo describe the first reported case of nonislet cell tumor hypoglycemia (NICTH) associated with carcinosarcoma of the ovary.MethodsWe report the clinical course, imaging, and pathologic findings of our patient and review relevant literature.ResultsA 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.ConclusionsThis 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 (Endocr. Pract. 2013;19:e83-e87)
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