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J Thorac Cardiovasc Surg 2006;132:713-714
© 2006 The American Association for Thoracic Surgery
Brief Communication |
Department of Thoracic Surgery, Shin-Kokura Hospital, Federation of National Public Service Personnel Mutual Aid Associations, Kitakyusyu, Japan.
Received for publication May 10, 2006; accepted for publication May 12, 2006. * Address for reprints: Ryoichi Nakanishi, MD, PhD, Department of Thoracic Surgery, Shin-Kokura Hospital, Federation of National Public Service Personnel Mutual Aid Associations, 1-3-1 Kanada, Kokurakita-ku, Kitakyusyu 803-8505, Japan (Email: ryoichi{at}med.uoeh-u.ac.jp).
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Solitary fibrous tumor of the pleura (SFTP) is a rare neoplasm that accounts for 10% of pleural neoplasms. Although the majority of SFTPs have benign histologic features, approximately 12% of them are malignant.1
Although more than half of patients with SFTP are free of symptoms, large tumors cause respiratory symptoms such as dyspnea, cough, hemoptysis, and chest pain as a result of bronchial compression, atelectasis, and obstructive pneumonia. Furthermore, systemic symptoms, such as hypoglycemia and hypertrophic osteoarthropathy, and nonspecific symptoms, such as fever and fatigue, are occasionally found in patients with SFTP.
The incidence of hypoglycemia has been reported to be approximately 4%. Although insulin-like growth factor II (IGF-II) is considered to be related to the hypoglycemia, there are few reports manifesting this relationship on the basis of specific data.1 We herein describe a rare case of malignant SFTP with hypoglycemia closely associated with serum IGF-II.
Clinical Summary
A 77-year-old man was admitted with cold sweating, cough, and dyspnea. He had symptoms of finger clubbing and hypertrophic osteoarthropathy.
Laboratory analyses on pulmonary functions at admission are described in Table 1. Chest radiographs revealed a large consolidation in the right middle and lower lung fields although a small consolidation had been pointed out in the same fields 15 years before. Computed tomographic scan of the chest with intravenous contrast demonstrated the abnormal giant mass slightly compressing the heart (Figure 1).
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The tumor measured 10.9 x 9.8 x 9.4 cm. On histologic examination, the tumor exhibited a classic "pattern-less" pattern of SFTP, composed of a cellular proliferation of spindle-shaped cells accompanied by dense collagenous stroma and hemangiopericytomatous vasculatures. In terms of increased mitotic activity, high cellularity, and the presence of necrosis, this tumor was considered a low-grade malignancy from the England criteria.2 The surgical margins of pleura, vessels, and bronchus were free of tumor cells.
Hypoglycemia improved in association with decreased serum IGF-II early after the operation. Respiratory symptoms of the patient decreased gradually and pulmonary function improved (Table 1). The postoperative course was uneventful except for air leak. The patient is alive 9 months after the operation without signs of recurrence.
Discussion
The nonislet cell tumors, including SFTP, fibrosarcoma, mesothelioma, leiomyosarcoma, and hemangiopericytoma, often cause fasting hypoglycemia.3 Tumor-derived IGF-II is thought to be a hypoglycemic agent. IGF-II is a polypeptide growth factor composed of 67 amino acid residues and shares a high degree of sequence homology with insulin.4 The mechanism by which IGF-II induces hypoglycemia is not fully understood. Recent studies have shown a large molecular form, designated big IGF-II, which is probably an incompletely processed molecule of IGF-II and may play a key role in the pathogenesis of nonislet cell tumor hypoglycemia.5 IGF-II produced by the tumor may act on insulin receptors, thereby inducing hypoglycemia. The fact that the hypoglycemia reverted to normal in association with decreased serum IGF-II after resection of the tumor suggests this speculation, although the normal value of serum IGF-II is unclear.
de Perrot and colleagues1 histologically classified SFTP as benign and malignant types. Recurrences were reported in approximately 10% of the benign groups, although the recurrence rate in malignant groups of SFTP is high. The histologic changes of malignancy are closely associated with prognosis of this disease. In most cases chemotherapy and radiotherapy are not effective, especially in malignant SFTP.
In conclusion, a complete resection improves both hypoglycemia associated with serum IGF-II and relevant respiratory symptoms and would have a good effect on prognosis.
References
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