2012
CASE REPORT BENIGN CCTL
HAN ET AL
(totaling 4 patients) in which Amplatzer devices were used in the airways to close bronchopleural and esophagorespiratory fistulas [4 – 6]. In this report, we describe the use of rigid bronchoscopy to deploy an Amplatzer device for closure of an acquired benign fistula between the airways and digestive tract. Because of the size of the deployment device, rigid bronchoscopy was necessary for simultaneous placement of the Amplatzer device and the telescopic camera into the airway. This enabled direct visualization of the deployment as well as confirmation that airway contamination had ceased once the fistula was closed.
References
FEATURE ARTICLES
1. Buskens CJ, Hulscher JB, Fockens P, Obertop H, van Lanschot JJ. Benign tracheo-neo-esophageal fistulas after subtotal esophagectomy. Ann Thorac Surg 2001;72:221– 4. 2. Mughal MM, Gildea TR, Murthy S, Pettersson G, DeCamp M, Mehta AC. Short-term deployment of self-expanding metallic stents facilitates healing of bronchial dehiscence. Am J Respir Crit Care Med 2005;172:768 –71. 3. Ranes JL, Budev MM, Murthy S, Mehta AC. Management of tracheomediastinal fistulas using self-expanding metallic stents. J Thorac Cardiovasc Surg 2006;131:748 –9. 4. Kramer MR, Peled N, Shitrit D, et al. Use of Amplatzer device for endobronchial closure of bronchopleural fistulas. Chest 2008;133:1481– 4. 5. Golbin JM, Prakash UB. Closure of a bronchopleural fistula using an Amplatzer Septal Occluder Device. Chest 2006;130(4 suppl):291S. 6. Rabenstein T, Boosfeld C, Henrich R, Ell C. First use of ventricular septal defect occlusion device for endoscopic closure of an esophagorespiratory fistula using bronchoscopy and esophagoscopy. Chest 2006;130:906 –9.
Benign Clear Cell Tumor of the Lung Bingqiang Han, MM, Gening Jiang, MD, Haifeng Wang, MM, Wenxin He, MM, Ming Liu, MM, and Nan Song, MM
Ann Thorac Surg 2010;89:2012– 4
C
lear cell tumor of the lung (CCTL) was primarily described by Liebow and Castleman in 1963 as an extremely rare benign pulmonary neoplasm [1]. This tumor is also known as a “sugar tumor” because of the abundant periodic acid-Schiff (PAS) staining positive for glycogen in cytoplasm. There have been only sporadic cases of this neoplasm reported in the world literature; therefore, we have limited information regarding this neoplasm. We present a case of CCTL and discuss the clinical and pathologic features of the tumor. A 51-year-old man was admitted to our hospital for an abnormal pulmonary nodule on the left lung, which was occasionally found on chest roentgenogram during routine examinations. A chest computed tomographic scan showed a solitary nodule of approximately 15 mm in diameter in the lingular segment of the left upper lobe, with round and smooth margins, uniform internal density, and a CT value of 15 Hounsfield Units. There was no sign of lobulation or spicules (Fig 1). Fiberoptic bronchoscopy showed no stenosis of the bronchi and was negative with brush biopsy. There were no significant findings in physical examinations and laboratory studies. The patient underwent wedge resection by videoassisted thoracoscopic surgery, but later a lobectomy was performed by thoracotomy because a frozen section study of the tumor was prone to borderline tumor or low-grade malignancy. Mediastinal lymph nodes (groups 5 to 7 and 9 to 11) were conventionally scavenged. Histologic examination revealed that the cytoplasm contained abundant glycogen granules observed by PAS staining. Light microscopy showed the tumor cells were round or irregular in shape and rich in transparent cytoplasm. The nuclei were small with uniform chromatin and without mitotic figures or necrosis. The tumor cells were arranged in a sheet with affluent capillaries and irregular sizes of thin-walled sinusoid (Fig 2). The immunohistochemical studies showed the tumor expressed human melanoma black (HMB)-45 (Fig 3), vi-
Department of Thoracic Surgery, Shanghai Pulmonary Hospital, Tongji University, Shanghai, China
Clear cell tumor of the lung is a rare benign tumor. Herein, we present a case of a 51-year-old man who had an abnormal pulmonary nodule occasionally found on chest roentgenogram. A computed tomographic scan showed a solitary nodule (approximately 15 mm in diameter) in the lingular segment of the left upper lobe. The patient underwent a lobectomy for the tumor. The pathologic report supported the diagnosis of clear cell tumor of the lung. The patient showed no evidence of recurrence or metastasis 2 years postoperatively. (Ann Thorac Surg 2010;89:2012– 4) © 2010 by The Society of Thoracic Surgeons Accepted for publication Oct 8, 2009. Address correspondence to Dr Jiang, Department of Thoracic Surgery, Shanghai Pulmonary Hospital, Tongji University, 507 Zheng Min Rd, Shanghai, 200433, China; e-mail:
[email protected].
© 2010 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. A solitary nodule (approximately 15 mm in diameter) was observed in the lingular segment of the left upper lobe, with round and smooth margins and a uniform internal density of 15 Hounsfield Units (arrow). 0003-4975/$36.00 doi:10.1016/j.athoracsur.2009.10.017
Fig 2. Hematoxylin and eosin staining of the tumor cells (⫻200).
mentin, neuron-specific enolase, and CD34, and was focally positive for S-100 protein. However, the tumor cells were negative for cytokeratin, desmins, CD68, and thyroid transcription factor-1. The patient was discharged from the hospital after an uneventful recovery on postoperative day 6. At 2 years postoperatively there was no evidence of recurrence or metastasis.
Comment The primary CCTL is an extremely rare benign tumor. Liebow and Castleman briefly reported the first four examples [1], but they made a detailed study of 12 of these tumors in 1971 [2]. They found that cells of these tumors contained quantities of glycogen, but none of fat. A study of chemical composition of the tumors revealed 10,657 -moles of glycogen-derived hexose and 100 g of wet tissue, whereas 25.4 -moles of that in normal lung tissue. Thus far there have been approximately 50 cases of CCTL reported in the English literature [3–9]. The tumor usually occurs between the ages of 40 and 60 years, without any gender difference. Most patients in whom the tumor was found occasionally in routine examinations are asymptomatic, except for several cases with symptoms such as hemoptysis [4] or fever [5]. Located in the peripheral lung, the tumor looks like a coin on chest roentgenogram or computed tomographic scan, with a smooth margin of approximately 1 to 4 cm in diameter. In our case, a round pulmonary nodule was incidentally noticed in a routine examination. There were no preoperative symptoms. A clear cell tumor of the lung is also called a “sugar tumor,” because there are numerous glycogen granules in the cytoplasm. Therefore, PAS staining is necessary for a definitive diagnosis. In our case, the tumor was positive for PAS staining and negative for PAS staining after digested by amylase. We did not observe any mitotic figures or necrosis. According to immunoreactivity for HMB-45 and the
CASE REPORT
HAN ET AL BENIGN CCTL
2013
presence of pre-melanosomes observed by an electron microscope in both CCTL and angiomyolipoma of the kidney and liver, Bonetti and colleagues [7] put forward a notion of tumors showing perivascular epithelioid cell differentiation. The perivascular epithelioid cell family differentiation also involved lymphangiomyomatosis and a series of tumors with similarly morphological and phenotypical features. However, the histogenesis of CCTL is unclear. We analyzed some immunohistochemical studies with HMB-45, S-100 protein, vimentin, and others reported in previous literature [3–9]. The major immunohistochemical features of CCTL were HMB-45 (19 of 21), S-100 protein (17 of 20), vimentin (6 of 16), and neuron-specific enolase (NSE) (7 of 20). There was no reactivity for cytokeratin, epithelial membrane antigen, and chromogranin. The specific characteristics of CCTL are positive for PAS staining without mitotic figures or necrosis. We also noticed that HMB-45-positive, S-100 positive, and cytokeratin negative were also specific for CCTL. We believe that the joint application of these markers and PAS staining can distinguish CCTL from other pulmonary neoplasms more exactly, involving primary clear cell carcinoma of the lung and the pulmonary metastatic renal clear cell carcinoma. Some cases revealed that CD1a [3] and CD34 [6] contributed to a clear positive diagnosis, and the immunoreactivity for CD34 was also positive in our case. Most cases of CCTL (including our case) were usually diagnosed by thoracotomy. Clear cell tumor of the lung is a benign tumor of lung that may be cured by surgical resection. Lobectomies, segmentecties, and partial resections have been previously reported as treatments. There were also no reports of recurrence. However, Gaffey and colleagues [8] reported a case in which the patient had hepatic metastasis occur 17 years after the surgical resection. Therefore, follow-ups for as long as possible are recommended. Our patient has been followed-up for 2 years postoperatively without any sign of recurrence or metastasis. To our knowledge, it is the first case report of CCTL in China (written in English).
Fig 3. Tumor cells show positive immunohistochemical staining for HMB-45 (Streptavidin-perosidase [SP] ⫻200).
FEATURE ARTICLES
Ann Thorac Surg 2010;89:2012– 4
2014
CASE REPORT CHETCUTI ET AL MASSIVE HEMOTHORAX SECONDARY TO RENAL CARCINOMA
References 1. Liebow AA, Castleman B. Benign “clear cell tumors” of the lung. Amer J Path 1963;43:13a– 4a. 2. Liebow AA, Castleman B. Benign clear cell (“sugar”) tumors of the lung. Yale J Biol Med 1971;43:213–22. 3. Adachi Y, Kitamura Y, Nakamura H, et al. Benign clear (sugar) cell tumor of the lung with CD1a expression. Pathol Int 2006;56:453– 6. 4. Santana AN, Nunes FS, Ho N, Takagaki TY. A rare cause of hemoptysis: benign sugar (clear) cell tumor of the lung. Eur J Cardiothorac Surg 2004;25:652– 4. 5. Gora-Gebka M, Liberek A, Bako W, Szumera M, Korzon M, Jaskiewicz K. The “sugar” clear cell tumor of the lung-clinical presentation and diagnostic difficulties of an unusual lung tumor in youth. J Pediatr Surg 2006;41:e27–9. 6. Hirata T, Otani T, Minamiguchi S. Clear cell tumor of the lung. Int J Clin Oncol 2006;11:475–7. 7. Bonetti F, Pea M, Martignoni F, et al. Clear cell (“sugar”) tumor of the lung is a lesion strictly related to angiomyolipoma–the concept of a family of lesions characterized by the presence of the perivascular epithelioid cells (PEC). Pathology 1994;26:230 – 6. 8. Gaffey MJ, Mills SE, Zarbo RJ, Weiss LM, Gown AM. Clear cell tumor of the lung. Immunohistochemical and ultrastructural evidence of melanogenesis. Am J Surg Pathol 1992;15: 307– 8. 9. Gal AA, Koss MN, Hochholzer L, Chejfec G. An immunohistochemical study of benign clear cell (‘sugar’) tumor of the lung. Arch Pathol Lab Med 1991;115:1034 – 8.
FEATURE ARTICLES
Massive Hemothorax Secondary to Metastatic Renal Carcinoma Karen Chetcuti, MRCS, James Barnard, MRCS, Spiros Loggos, MD, Mohamed Hassan, MRCS, Vivek Srivastava, MRCS, Faisal Mourad, MRCS, Zyad Makhzoum, MRCP, and Mohamad N. Bittar, FRCS C-Th
Ann Thorac Surg 2010;89:2014 – 6
A
spontaneous hemothorax is a rare clinical occurrence. The differential diagnosis of a major hemothorax that has not resulted from trauma can include vascular, idiopathic, malignant, and spontaneous causes. Spontaneous bleeding caused by metastatic renal cell carcinoma has been described in the gastrointestinal tract, cerebellopontine angle, and the pancreas, but we believe that our case describes the first patient with spontaneous bleeding in the thorax caused by metastatic renal cell carcinoma. A 78-year-old man was brought to the emergency room after suddenly collapsing at home. He had complained of a dull ache in his chest just an hour prior, which was during a walk in the countryside. The ambulance crew had found him with an unrecordable blood pressure and he was resuscitated with fluids on his way to the hospital. His hemoglobin was 10.0 g/dL and his clotting was normal. The patient’s past medical history included a right nephrectomy 12 years previously for renal cell carcinoma. He was also an ex-smoker and took regular aspirin. A chest roentgenogram revealed a massive right hemothorax with a mediastinal shift to the left side (Fig 1). A pleural tap produced fresh blood and therefore a right-sided chest drain was inserted that immediately drained more than 1 L of fresh blood. A computed tomographic scan confirmed the presence of a massive right-sided hemothorax with significant mediastinal shift to the left. Active arterial bleeding was noted from a hypervascular pleural-based lesion that was lying adjacent to the fifth rib in the anterolateral position. Several other hypervascular pleural and diaphragmatic-based lesions were also seen and observed to be extending through the diaphragm and abutting the liver, which
Lancashire Cardiac Centre, Victoria Hospital, Whinney Heys Road, Blackpool, Lancashire, United Kingdom
We present the case of a 78-year-old man with a past history of nephrectomy for renal cell carcinoma who was brought to the emergency room in hypovolemic shock after suddenly collapsing at home. A chest roentgenogram taken on admission showed a large pleural effusion on the right with a mediastinal shift to the left. A contrast computed tomographic scan of his chest excluded aortic dissection. An emergency right thoracotomy was performed, which revealed a chest wall tumor that was bleeding profusely. The tumor was biopsied after the bleeding was controlled. The histologic report revealed metastatic renal cell carcinoma, which is a rare cause for a spontaneous massive hemothorax. (Ann Thorac Surg 2010;89:2014 – 6) © 2010 by The Society of Thoracic Surgeons Accepted for publication Oct 1, 2009. Address correspondence to Dr Chetcuti, Cardiac Audit Office, Lancashire Cardiac Centre, Victoria Hospital, Whinney Heys Rd, Blackpool, FY3 8NR, United Kingdom; e-mail:
[email protected].
© 2010 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Chest roentgenogram showing a right hemothorax associated with a mediastinal shift to the left side. 0003-4975/$36.00 doi:10.1016/j.athoracsur.2009.10.009