• Takaya Suzuki, Hirotsugu Notsuda, Hisashi Oishi, Hiromichi Niikawa, Tatsuaki Watanabe, Yui Watanabe, Ken Onodera, Tetsuto Takeda, Ringo Sugawara, Masafumi Noda, Kanako Sakurai, Mototsugu Nagao, Izumi Fukuda, and Yoshinori Okada.
• Department of Thoracic Surgery, Institute of Development, Aging and Cancer, Tohoku University.
• Tohoku J. Exp. Med. 2024 May 23; 263 (1): 111611-16.

AbstractNon-islet cell tumor hypoglycemia (NICTH) is one of the paraneoplastic syndromes manifesting severe hypoglycemia caused by aberrant production of high-molecular-weight insulin-like growth factor 2 (big-IGF2). Two surgical cases of extremely large thoracic solitary fibrous tumors (SFT) with unusual history of NICTH are presented. One case manifested severe hypoglycemia after four years of the first complete surgical resection of the tumor with potential malignant transformation, and the other case showed severe hypoglycemia after ten years of the first detection of the tumor. Meticulous laboratory testing, including serum endocrinological tests and western immunoblotting before and after surgery was performed, and both cases were diagnosed as NICTH. Both patients underwent open thoracic surgery. The patients showed normal glucose and hormone levels immediately after the resection of responsible tumors with elevated blood insulin concentration. SFTs are generally considered benign; however, life-threatening hypoglycemia can happen regardless of treatment. Careful follow-up of the tumor growth is warranted.

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