References 1. Nakatsuka S, Yao M, Hoshida Y, Yamamoto S, Iuchi K, Aozasa K. Pyothorax-associated lymphoma: a review of 106 cases. J Clin Oncol 2002;20:4255– 60. 2. Narimatsu H, Ota Y, Kami M, et al. Clinicopathological features of pyothorax-associated lymphoma; a retrospective survey involving 98 patients. Ann Oncol 2007;18:122– 8. 3. Riehl G, Aubert A, Sandu C, Brichon PY. Malignant nonHodgkin’s lymphoma developing late after pneumonectomy. Eur J Cardiothorac Surg. 2006;30:948 –9. 4. Hashizume T, Aozasa K, Tomita Y, Matsushita K. Pyothoraxassociated T-cell lymphoma: a case report. Jpn J Clin Oncol 2003;33:145–7. 5. Fukino S, Fukata T, Inoue A, Hatazawa Y, Morio S. A surgical case of T-cell non-Hodgkin’s lymphoma originating in the wall affected by chronic tuberculous empyema following artificial pneumothorax. Nippon Kyobu Geka Gakkai Zasshi 1992;40: 113–7.
Giant Cell Tumor of the Sternum Emmanuele Abate, MD, Farzaneh Banki, MD, Jeffrey A. Hagen, MD, and Nancy Klipfel, MD Department of Surgery and Pathology, University of Southern California, Keck School of Medicine, Los Angeles, California
Primary giant cell tumors of the chest wall are extremely rare. To date, we believe that there have been no reported cases of sternal giant cell tumors in the thoracic literature. We report a case of an isolated giant cell tumor of the sternum in a 28-year-old man. The mass was resected and the sternum was reconstructed with methyl methacrylate prosthesis and bilateral pectoralis muscle advancement flaps. Excellent functional and aesthetic results were achieved. (Ann Thorac Surg 2009;88:645–7) © 2009 by The Society of Thoracic Surgeons
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The sternum was stable, and the sternal pain increased on coughing. He denied palpitations or left-sided chest pain. There were no other visible bony lesions. Laboratory evaluation revealed a calcium level of 10.3 mg/dL and an alkaline phosphatase of 79 U/L. A chest roentgenogram demonstrated a normal cardio-mediastinal silhouette. A computed tomographic scan of the chest showed a 6.4 ⫻ 4.3 ⫻ 4.4 cm expansile lytic lesion within the sternum with an intact cortex and no soft tissue abnormality (Fig 1). No other chest wall bone abnormalities were seen. The differential diagnosis included benign causes such as GCT, chondromyxoid fibroma (chondroid matrix), or aneurysmal bone cyst versus a chondrosarcoma. Fineneedle aspiration of the mass showed multi-nucleated giant cells, mononuclear plasmacytoid stromal cells, and rare benign skeletal muscle. The immunohistochemical stains for vimentin, S 100, pancytokeratin, desmins, and smooth muscle antigen revealed that the cells were immunoreactive only for vimentin, which supports the diagnosis of GCT. The mass was resected through a midline incision over the sternum. The tissue around the tumor was mobilized in a circumferential manner, and the sternal edges as well as the cartilages of the third, fourth, fifth, and sixth ribs were exposed. The manubrium was not involved. The superior and inferior margins of the tumor were resected using a sternal saw. Sections of the third, fourth, and fifth costal cartilages were excised medially. Both internal mammary arteries were identified and preserved. The involved sternum was lifted using bone hooks. The tumor involved the posterior plate of the sternum without involvement of any retrosternal tissue. The sternal defect was reconstructed with a sandwich of Prolene (Ethicon, Somerville, NJ) mesh and methyl
G
iant cell tumor (GCT) of the bone is a rare benign lesion that comprises approximately 5% of primary bone tumors in adults [1]. The GCT exhibits a slight female predilection and usually occurs in the third and fourth decades of life [2]. The GCTs typically arise in the metaphyseal-epiphyseal area most commonly in the distal femur, proximal tibia, and distal radius [3]. They have rarely been reported in other sites, such as the chest wall or sternum. In a review of 134 cases of chest wall tumors, Ochsner and colleagues [4] found only one benign sternal lesion. We report a case of GCT of the mid-sternum in a 28-year-old man. A 28-year-old man presented with a 1-year history of midsternal chest pain followed by a mass that slowly increased in size. On physical examination he had a round 6 ⫻ 6 cm hard, fixed, and tender mass in the mid-sternum without involvement of the manubrium.
Accepted for publication Dec 16, 2008. Address correspondence to Dr Banki, Department of Surgery, Division of Thoracic and Foregut Surgery, University of Southern California, 1510 San Pablo, #514, Los Angeles, CA 90033; e-mail: fbanki@surgery. usc.edu.
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Computed tomographic scan presentation of the giant cell tumor of the sternum (arrow) in a 28-year-old man. 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2008.12.059
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Fig 2. (A) Tumor resection showing numerous giant cells and mononuclear stromal cells. (Hematoxylin & eosin, ⫻100.) (B) Giant and mononuclear cells showing similar oval nuclei with open chromatin and small nucleoli. (Hematoxylin & eosin, ⫻400.) (C) The tumor focally penetrates through the bone into adjacent adipose tissue. (Hematoxylin & eosin, ⫻40.)
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methacrylate. The prosthetic plates were fixed to the sternum with sternal wires superiorly and inferiorly. The plate was fixed to the remaining cartilaginous portions of the third, fourth, and fifth ribs using No. 1 Prolene interrupted stitches (Ethicon). Once the plate was placed in the sternal defect, the edges of the mesh were sewed to the surrounding tissue with No. 1 Prolene to keep the edges of the mesh in place and to allow the growth of tissues to cover the edges of the mesh. The plate was covered by bilateral pectoralis muscle advancement flaps. The specimen consisted of a single ellipsoid sternal tumor that measured 5 cm from anterior to posterior, 8.5 cm from left to right, and 12.5 cm from superior to inferior. The histopathologic examination of hematoxylin and eosin stained slides showed numerous medium to large multi-nucleated osteoclast-type giant cells in a background of mononuclear stromal cells (Fig 2A). These two populations have the same oval nuclei with open chromatin and small nucleoli (Fig 2B). No significant atypia and only rare mitoses were present. The tumor focally penetrated through the bone into the adjacent adipose tissue (Fig 2C). The specimen margins were negative for tumor. His hospital course was uncomplicated, and the patient was discharged on postoperative day 5. At 5-months follow-up, he is free of symptoms, has a stable sternum, and no evidence of tumor recurrence, and he is performing his daily activities without difficulties.
Comment Benign tumors of the sternum are extremely rare. The most common differential diagnosis includes chondroma,
fibrous dysplasia, lipoma, fibroma, and aneurysmal bone cyst [5]. Malignant lesions such as chondrosarcoma, solitary plasmacytoma, metastatic carcinoma, lymphoma, Ewing’s tumor, and desmoid tumor should be excluded because the treatment of malignant tumors differs from the benign tumors and includes a more radical resection. Limited experience and difficulties in distinguishing benign from malignant cartilaginous lesions of the chest wall are considerable challenges in their diagnosis and management. Radiologic features are not always diagnostic of malignancy, but presence of cortical destruction and soft tissue swelling is indicator of malignancy [6]. A plain chest roentgenogram, as well as a computed tomographic scan is essential for diagnosis. Advances in pathologic techniques, including the use of special stains and immunocytochemistry may help in the diagnosis. The histologic morphology on large biopsies or resection specimens is reliably diagnostic for giant cell tumors. However, interpretation of small biopsies or fine-needle aspiration specimens might be limited due to the various giant cell-containing bone tumors, including osteosarcoma, chondroblastoma, and aneurysmal bone cyst. Immunohistochemical stains can help eliminate some lesions from the differential diagnosis. Therapeutic options are intralesional curettage for small tumor or wide radical resection, and reconstruction for more extensive or recurrent tumors, or both. The best method of reconstruction is to use synthetic mesh such as Marlex (Marlex Pharmaceuticals Inc, New Castle, Delaware), Prolene (Ethicon), or polytetrafluroethylene in combination with methyl methacrylate. The reported long-term local control of giant cell tumor of bone after surgical resection ranges from 73% to 100% [3]. Radiation therapy has no role in treatment of completely resected tumors.
References 1. Gamberi G, Serra M, Ragazzini P, et al. Identification of markers of possible prognostic value in 57 giant cell tumors of bone. Oncol Reports 2003;10:351– 6. 2. Zhen W, Yaotian H, Songjian L, et al. Giant cell tumor of bone. The long-term results of treatment by curettage and bone graft. J Bone Joint Surg Br 2004;86-B:212– 6. 3. Mendenhall W, Zlotecki R, Scarborough M, Parker Gibbs C, Mendenhall N. Giant cell tumor of bone. Am J Clin Oncol 2006;29:96 –9. 4. Ochsner A Jr, Lucas GL, Mc Garland GB Jr. Tumors of the thoracic skeleton: review of 134 cases. J Thorac Cardiovasc Surg 1966;52:311–21. 5. Athanassiadi K, Kalavrouziotis G, Rondogianni D, Loutsidis A, Hatzimichalis A, Bellenis I. Primary chest wall tumors: early and long-term results of surgical treatment. Eur J Cardiothorac Surg 2001;19:589 –93. 6. Sabanathan S, Salama FD, Morgan WE, Harvey JA. Primary chest wall tumors. Ann Thorac Surg 1985;39:4 –15.
Primary Pulmonary Meningioma Presenting with Hemoptysis on Exertion Nobuhiro Izumi, MD, Noritoshi Nishiyama, MD, Takashi Iwata, MD, Koshi Nagano, MD, Takuma Tsukioka, MD, Shoji Hanada, MD, and Shigefumi Suehiro, MD Department of Thoracic Surgery, Osaka City University Hospital, Osaka, Japan
An 18-year-old woman presented with hemoptysis during long-distant running. A chest computed tomographic scan revealed a well-demarcated, round-shaped, solitary mass (3.0 cm in diameter) in the left pulmonary hilum. The bleeding was getting more often and a left upper lobectomy was carried out to avoid critical airway bleeding. A solid proliferation of short spindle cells with a remarkable whorl pattern was demonstrated histologically. Immunohistochemical studies showed positive for vimentin and S-100, and focally positive for epithelial membrane antigen. Primary pulmonary meningioma was thus diagnosed. The patient is well without recurrent disease 15 months after surgery. (Ann Thorac Surg 2009;88:647– 8) © 2009 by The Society of Thoracic Surgeons
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hemoptysis on exertion that was successfully treated by surgical resection. An 18-year-old woman presented with hemoptysis on exertion, especially during long-distant running. She was otherwise healthy and had no history of pulmonary or neurologic disturbances. A chest roentgenogram showed a solitary mass of 3.0 cm in diameter in the left pulmonary hilum. A chest computed tomographic scan revealed a well-demarcated, round-shaped, and homogenous lesion without showing any cavity lesions or densities of fatty or calcified contents, or both, in the left upper lobe adjacent to the pulmonary artery and vein (Fig 1). Brain and abdominal computed tomographic scans demonstrated no evidence of metastasis. Values of carcinoembryonic antigen and squamous cell carcinomaassociated antigen in serum were within normal ranges. Diagnostic bronchoscopy or computed tomographicguided needle biopsy of the tumor was refrained from because of possible massive bleeding from the hilar vessels or the hemorrhagic tumor itself during the procedures. Furthermore, the bleeding was getting easier and more frequent; therefore, pulmonary resection was scheduled to prevent further acute bleeding, without confirming definitive histologic diagnosis. Preoperative differential diagnoses were hamartoma, sclerosing hemangioma, or a metastatic tumor of unknown origin. A left upper lobectomy was carried out through a small axillary thoracotomy of 5 cm with video assistance. Diffuse telangiectasis was seen on the visceral pleura of the upper lobe. A resected specimen showed an elastic soft, well-demarcated tumor, measuring 33 mm in diameter. The cut surface was yellowish white, soft, and fragile. An intraoperative frozen section revealed a spindle-cell tumor partially containing atypical epithelial components. A definitive diagnosis of benign or malignant disease was not obtainable; therefore, mediastinal dissection was also undertaken in case of possible epithelial malignancy. Postoperative histologic examination showed the lesion consisted mainly of a solid proliferation of short-spindle
P
rimary pulmonary meningioma (PPM) is an extremely rare entity [1]. Since Erlandson described the first case in 1981, only 33 cases have been reported in the English literature [2]. Most of the cases were asymptomatic. We report herein a case of PPM presenting with
Accepted for publication Dec 15, 2008. Address correspondence to Dr Iwata, Department of Thoracic Surgery, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, 545-8585, Japan; e-mail:
[email protected].
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Chest computed tomographic scan shows a solitary, well-demarcated, round-shaped and homogenous 3.0 cm diameter tumor in the hilum of the left upper lobe, adjacent to the pulmonary artery and vein. No cavity lesion or any density of fatty or calcified contents are seen. 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2008.12.058
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