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━━ Background. Non-islet cell tumor hypoglycemia (NICTH) caused by non-pancreatic tumors is rare. Here, we report a case of NICTH that was associated with malignant solitary fibrous tumor (SFT) of the pleura, and was treated with glucocorticoid therapy. Case. A 72-year-old man presented with an abnormal shadow in the chest on medical health examination. A chest computed tomography (CT) scan confirmed the presence of a tumor shadow, approximately 12×9×8 cm in size, in the right upper lung field. A right upper lobectomy with chest wall resection and lymph node dissection were performed. Pathological findings of the resected specimens revealed malignant SFT arising from the pleura. Three years after the operation, multiple intrathoracic and inguinal lymph node recurrences were detected on CT. We decided to observe the natural course of recurrence without giving chemotherapy. Four years after the operation, he was admitted to our department with recurrent hypoglycemic attacks. Glucose infusion and intravenous hyperalimentation were not effective, and he experienced repeated hypoglycemic attacks. Based on the clinical course and examination results, we diagnosed NICTH associated with malignant SFT. After administration of oral glucocorticoids, the hypoglycemia improved. However, it was slightly difficult to regulate the dose of oral glucocorticoid. Conclusion. Glucocorticoid therapy is useful for treating NICTH with SFT and can be tailored to the individual’s needs. (JJLC. 2013;53:59-63) KEY WORDS━━Non-islet cell tumor hypoglycemia, Solitary fibrous tumor, Insulin-like growth factor, Steroid Reprints: Youko Kataoka, Department of Thoracic Surgery, Kyoto Medical Center Hospital, 1-1 Mukaihatacho, Fukakusa, Fushimiku, Kyoto-shi, Kyoto 612-8555, Japan (e-mail: oka319yoko@yahoo.co.jp). Received December 25, 2012; accepted February 18, 2013. 要旨━━ 背景. Non-islet cell tumor hypoglycemia (NICTH)は膵外腫瘍による空腹時低血糖症であり,頻度 は少ない.悪性胸膜孤立性線維性腫瘍(solitary fibrous tumor;SFT)によるNICTHにステロイド治療が奏効 した 1例を経験したので,文献的考察を加え報告する. 症例.72 歳,男性.4年前の健診で右上肺野に異常陰影 指摘,胸部CTで右肺尖部に 12×9×8 cm大の腫瘤陰影 を認め,右肺上葉・胸壁合併切除術を施行し,胸膜由来 の悪性 SFTと診断した.1年前に胸腹部CTで右胸腔内 に多発する腫瘤影,右鼠径リンパ節腫脹を認め,リンパ 節生検より悪性胸膜 SFT術後再発と診断,無治療で経過 観察としていた.今回は低血糖発作で救急搬送,経過と 精査より悪性胸膜 SFTによるNICTHと考えられた.高 カロリー持続点滴とブドウ糖投与で対応していたが,頻 回に低血糖発作を繰り返したため,ステロイド経口投与 を開始した.低血糖症は改善し,輸液を終了することが 1京都医療センター呼吸器外科;2洛和会音羽病院呼吸器外科; 3滋賀医科大学付属病院呼吸器外科. 別刷請求先:片岡瑛子,京都医療センター呼吸器外科,〒6128555 京都市伏見区深草向畑町 1-1 (e-mail: oka319yoko@yahoo.co. jp). 受付日:2012 年 12 月 25 日,採択日:2013 年 2 月 18 日. (肺癌.2013;53:59-63) 2013 The Japan Lung Cancer Society A Case of NICTH with Malignant SFT of the Pleura―Kataoka et al 60 Japanese Journal of Lung Cancer―Vol 53, No 1, Feb 20, 2013―www.haigan.gr.jp Figure 1. A: An extrapulmonary tumor, 12×9×8 cm in size arising from the pleura and compressing the lung. B: The tumor consisting of a nest of spindle or rounded cells. Mitotic activity was 10-16/HPF. A B できたが,経口摂取の状況や腫瘍の増大とともにステロ イドの投与内容の変更や投与量増量を要した.結論. SFTの NICTHに対する治療として,外科的治療が唯一 効果的であるといわれているが,本症例のように手術適 応外の場合,ステロイドが有用である.しかし,その投 与時間や投与量は工夫が必要である. 索引用語━━NICTH,孤立性線維性腫瘍,インスリン様 成長因子,ステロイド

A Case of Non-islet Cell Tumor Hypoglycemia with Malignant Solitary Fibrous Tumor of the Pleura