A patient with recurrent severe hypoglycaemia attacks due to a large, irresectable retroperitoneal ... certain non-islet cell tumours such as leiomyosar- intensive ...
T he British Journal of Radiology, 70 (1997 ), 306–308
© 1997 The British Institute of Radiology
Case report
Radiotherapy for hypoglycaemia associated with large leiomyosarcomas K KISHI, MD, T SONOMURA, MD and M SATO, MD Department of Radiology, Wakayama Medical College, 7-Bancho-27, Wakayama City 640, Japan Abstract. A patient with recurrent severe hypoglycaemia attacks due to a large, irresectable retroperitoneal leiomyosarcoma was treated with radiotherapy (60 Gy). The blood glucose level gradually and steadily improved as the cumulative radiation dose was increased. Weaning of hyperalimentation was started when the cumulative dose reached 21.6 Gy. The patient became completely free from hypoglycaemic attacks despite no significant diminishment of local tumour size and untreated multiple lung metastases. The patient was discharged and the attacks did not recur until expiration as a result of tumour bleeding. This case report supports the clinical usefulness of radiation therapy in treating hypoglycaemia induced by non-islet cell tumour.
Introduction Severe hypoglycaemia may be associated with certain non-islet cell tumours such as leiomyosarcoma, fibrosarcoma or mesothelioma [1–3]. The first choice of treatment is surgical tumour resection. Radiotherapy, or chemotherapy, is indicated in unresectable cases [3, 4 ]. Although hypoglycaemia is thought to disappear with effective surgical treatment of the primary tumour, a few clinical papers have supported the effectiveness of radiotherapy or chemotherapy [2]. We report a case of a severe hypoglycaemia as a result of a large recurrent leiomyosarcoma in which the hypoglycaemia was successfully controlled by radiotherapy.
Case report A 72-year-old male was referred for radiotherapy to a large recurrent leiomyosarcoma occupying his lower abdomen and pelvis, the primary retroperitoneal tumour having been resected 15 years previously. The initial hypoglycaemia attack, occurring 9 years after the first resection, led to further surgery for a recurrent tumour attached to the bladder. The next hypoglycaemic attack occurred 1 year later and led to a total vesicoureterectomy to remove refractory tumour. 2 years prior to the most recent presentation the patient experienced shoulder stiffness and lumbago, and a diagnosis of recurrence in the abdomen, with multiple metastases in the lungs, was made. Twicedaily oral 600 mg tegafur-uracil was started. However, 14 months later a severe hypoglycaemia Received 5 June 1996 and in final form 8 October 1996, accepted 25 October 1996. 306
attack ( blood glucose level of 17 mg dl−1) occured and, following this, hypoglycaemia became severe and uncontrollable despite hyperalimentation and intensive feeding. At the time of admission to our hospital, the patient showed extraordinary appetite, good performance status, a body-weight of 68 kg, and lower leg oedema due to tumour compression of iliac veins by a large abdominal mass. A CT scan revealed a large abdominal mass of 18×15×15 cm (Figure 1a) and multiple bilateral lung metastases (Figure 1b). Haematochemical analysis showed mild anaemia and hypoproteinaemia. Insulin-like growth factor-I (IGF-I) was in the normal range and measurement of IGF-II was not available in our hospital laboratory. The laboratory values, Alb, 3.1 g dl−1; TP, 6.1 g dl−1 and Somatomedin C, 0.17 U, were slightly lower than normal levels. Other haematological and biochemical parameters, including Insulin and Glucagon were in the normal range. Hyperalimentation was set at a rate of 75 kcal 50 ml−1 h−1 at night with extra food at midnight, 3 am and 5 am, and of 45 kcal 50 ml−1 h−1 in the daytime. Despite this, a hypoglycaemia attack occurred at midnight and the dosage was therefore increased to 105 kcal h−1 at night and 60 kcal h−1 in the daytime, totalling approximately 5500 kcal per day. The daily profile of blood glucose level on the sixth day was 153, 119, 84, 139, 88, 127 and 138 mg dl−1 for before and after meals and before sleep, respectively. Radiotherapy was commenced the next day. The gross abdominal tumour was treated by a 22×17 cm field using anteroposterior and posteroanterior 10 MV opposed photon beams, delivering 1.8 Gy fractions daily to a total of 59.8 Gy. At 21.6 Gy the daily profile was 114, T he British Journal of Radiology, March 1997
Case report: Radiotherapy for hypoglycaemia associated with leiomyosarcomas
(a)
(b)
Figure 1. (a) Abdominal X-ray CT images show a giant intrapelvic mass of inhomogenous density extending into the mid abdominal space. ( b) Chest X-ray CT images show multiple pulmonary metastases.
106, 78, 117, 105, 101 and 110, and the patient complained of nausea and full-stomach feeling. However, these symptoms disappeared by decreasing the glucose dose rate to 40 kcal h−1 at night. At 41.4 Gy the nausea appeared again, and the glucose dose rate was further decreased to 30 kcal h−1 at night. At 43.2 Gy, when the glucose dose was set at 20 kcal h−1 , the daily blood glucose profile was stationary. After reducing the glucose dose, the venous infusion was stopped when 55.8 Gy was reached. At this stage the patient no longer experienced hypoglycaemia attacks and the daily profile 1 week later was stable. Despite this marked effect, there was no significant reduction in the size of the abdominal tumour, even after boost treatment with hyperthermia, percutaneous ethanol injection, and transcatheter arterial embolization. The patient was discharged and maintained the same good performance status for 7 months during which time he had no further hypoT he British Journal of Radiology, March 1997
glycaemic attacks. He died from massive tumour bleeding.
Discussion The treatment for a non-islet tumour such as a leiomyosarcoma, causing fasting hypoglycaemia is surgical resection [1, 2]. However, primary or recurrent tumours may be inoperable and then an alternative remedy may be indicated. Chemotherapy is usually not effective for such tumours [2 ]. Radiotherapy is an alternative to surgery although these tumours may not be highly radiosensitive [ 4]. In the present case, the hypoglycaemic condition of the patient gradually disappeared as the cumulative radiation dose increased. The first improvement was obtained when the dose reached 20 Gy. The hypoglycaemia disappeared after radiotherapy of 59.8 Gy to the abdominal tumour despite no 307
K Kishi, T Sonomura and M Sato
remarkable shrinkage of the tumour and the presence of untreated large metastatic tumours in the lungs. In other words, the balance of glucose metabolism was recovered after treatment to a major part of the tumour. This means that at least one functional element, associated with fasting hypoglycaemia, of this tumour was radiosensitive, although no standard dose-fractionation schedule has been established for hypoglycaemia associated with such large non-islet tumours. This case report indicates the value of radiotherapy in this situation.
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References 1. Karn CR. The riddle of tumour hypoglycemia revisited. Clin Endocrinol Metabol 1980;9:335–60. 2. Kiu MC, Liaw CC, Ng KT, Ho YS. Leiomyosarcoma associated hypoglycemia—report of two cases. Chang Gung Med J 1990;13:237–41. 3. Papaioannou AN. Tumors other than insulinoma associated with hypoglycemia. Surg Gynecol Obstet 1966;123:1093–109. 4. Cox JD. The lung and thymus. In: Moss WT, Cox JD, editors. Radiation Oncology; rationale, technique, results. St Louis: Mosby, 1989, 292.
T he British Journal of Radiology, March 1997
