Ann Thorac Surg 2009;88:642-645. doi:10.1016/j.athoracsur.2008.11.072
© 2009 The Society of Thoracic Surgeons
Case Reports
A Surgical Case of Pyothorax-Associated Lymphoma of T-Cell Origin Arising From the Chest Wall in Chronic Empyema
Mario Santini, MDa,*,
Alfonso Fiorello, MD, PhDa,
Giovanni Vicidomini, MD, PhDa,
Luigi Busiello, MDa,
Alfonso Baldi, MDb
a Thoracic Surgery Unit, Second University of Naples, Naples, Italy
b Department of Biochemistry, Section of Pathology, Second University of Naples, Naples, Italy
Accepted for publication November 24, 2008.
* Address correspondence to Dr Santini, Chirurgia Toracica–Seconda Università di Napoli, Piazza Miraglia, 2, Naples, I-80138, Italy (Email: mario.santini{at}unina2.it).
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Abstract
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We report a surgical a case of pyothorax-associated lymphoma of T-cell origin arising from the chest wall and developing on pleural sequelae of therapeutic pneumothorax for pulmonary tuberculosis. The tumor was removed with resection of the fifth to eighth ribs. The chest wall defect repaired with a Marlex (Phillips Sumika Polypropylene Co, Houston, TX) prothesis. The histologic, immunohistochemical, and genotypic features were conclusive for a diagnosis of T-cell non-Hodgkin lymphoma. The patient received postoperative chemotherapy and is doing well after 15 months.
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Introduction
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Pyothorax-associated lymphoma (PAL) is a lymphoproliferative disorder that develops in the pleural cavity of patients who have long histories of pyothorax. Although the pathogenesis, clinical features, and optimal treatment have not been clarified, PAL represents an entity distinct from other malignant lymphomas [1]. We report a surgical a case of PAL of T-cell origin arising from the chest wall and developing on pleural sequelae of therapeutic pneumothorax for pulmonary tuberculosis.
In March 2007, a 78-year-old man was admitted to another hospital because of chest pain and asthenia. He had undergone artificial pneumothorax for pulmonary tuberculosis 53 years before. He had a long history of pyothorax that was left untreated. The results of all laboratory examinations were normal.
A contrast-enhanced chest tomography (CT) scan showed chest wall invasion adjacent to chronic pleural effusions, which was visualized as nonenhancing homogeneous soft-tissue density mass (Fig 1A). No extrathoracic abnormalities were seen. A radionuclide bone scan showed increased radioactivity of the mass lesion. Fine-needle aspiration supposed a diagnosis of non-small cell lung cancer (NSCLC), and the patient was transferred to our institution for surgical treatment.
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Fig 1. (A) Contrast-enhanced computed tomography shows chest wall invasion adjacent to chronic pleural changes, which is visualized as nonenhancing homogeneous soft-tissue density mass. (B) The chest wall defect was repaired with Marlex prothesis.
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Because the radiologic imaging findings were highly suggestive of lymphoma, we used incision biopsy rather than thoracotomy. Histologic analysis showed a proliferation of round cells, suggestive of a lymphoid proliferation; however, immunohistochemical studies were not definitive for a diagnosis of lymphoma. Consequently, we performed thoracotomy through the fifth intercostal space that revealed a tumor arising from chronic empyema that involved the sixth and seventh ribs. The chest wall tumor was removed with resection of fifth to eighth ribs and an empyema peel. The surgical margins were at least 3 cm away from the tumor. The chest wall defect (13 x 12 cm) was repaired with a Marlex prothesis (Phillips Sumika Polypropylene Co, Houston, TX) and muscle flaps (Fig 1B).
Histology showed the tumor was composed of a lymphoid mature cell population with an admixture of cells with slightly irregular and indented nuclear contours. Immunohistochemical testing showed these cells were positive for the T-cell markers CD3 and CD5. Among the neoplastic cells, we found few lymphoid cells that were positive for the B-cell markers CD20 and CD79a (Fig 2). Southern blotting revealed T-cell receptor gene rearrangement. No immunoglobulin light chain gene rearrangements were noted, and the Epstein-Barr virus (EBV) genome was not detected in the tumor cells. The histologic, immunohistochemical, and genotypic features were conclusive for a diagnosis of PAL of T-cell origin.
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Fig 2. Histologic and immunohistochemical analysis of the tumor: (A) the cell population was essentially composed of small lymphoid cells (hematoxylin-eosin; original magnification x 20). (B) Most of the neoplastic cells were positive for CD5, a T-cell marker (avidin-biotin complex; original magnification x20). (C) Few lymphoid cells were positive for CD20, a B-cell marker (avidin-biotin complex; original magnification x20).
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The patient received combination chemotherapy with six cycles of cyclophosphamide, doxorubicin, vincristine, and prednisolone (CHOP). His chest pain disappeared. The patient is currently alive 15 months after the operation.
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Comment
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Pyothorax-associated lymphoma is distinct from other malignant lymphomas and is defined as non-Hodgkin lymphoma of exclusively B-cell phenotype developing in the pleural cavity of patients with chronic pyothorax [1]. Induced pneumothorax created for tuberculosis is considered a risk factor because it leaves chronic nonhealing inflammation in the pleural cavity, which could subsequently result in the growth of PAL [2]. Narimatsu and colleagues [2] reported that the interval between the start of the chronic pyothorax and the onset of PAL is 22 to 55 years, similar to the 53 years in our patient who had a history of asymptomatic chronic pyothorax that started immediately after artificial pneumothorax for pulmonary tuberculosis. EBV may also be a further etiologic factor [1], but it was not detected in the tumor cells of our patient. Chest pain is a common symptom of PAL but is unusual in cases of chronic pyothorax. Radiographic evaluations are inconclusive for a definitive diagnosis, which is obtained by immunohistochemical and genotypic studies [1]. The subject case of PAL is rare due to its treatment, histology, and prognosis.
Although PAL responds to chemotherapy and radiotherapy, surgical treatment was used because the patient was previously diagnosed in another hospital as having NSCLC. The clinical and radiologic findings were highly suggestive of PAL, however, so an incisional biopsy was performed that was not converted to a full thoracotomy until the results of histologic and immunohistochemical studies were available. The immunohistochemical studies failed to establish a definitive diagnosis of lymphoma, and decortication and excision of the tumor was performed with a combined resection of four ribs. Reconstruction of the chest was completed with a Marlex prothesis to prevent scapula tip herniation into chest cavity during breathing. Although using Marlex prothesis presented an infection risk from pleural inflammation, the patient's postoperative course was unremarkable.
Most cases of PAL are B-cell type, and T-cell type is extraordinary [1]. In our patient, the histologic, immunohistochemical, and genotypic features allowed a diagnosis of T-cell non-Hodgkin lymphoma. Another patient with PAL of T-cell type origin in a Western country was reported by Rihel and colleagues [3] from France.
In a series of 98 patients from Tokyo, Narimatsu and colleagues [2] reported that 81 of 98 patients received aggressive treatments and that response rates to chemotherapy, radiotherapy, and chemoradiotherapy were 56%, 71%, and 83%, respectively. Median follow-up of survival was 39 months. Nakatsuka and colleagues [1] confirm that the overall prognosis of PAL after chemotherapy and radiotherapy is poor, with a 5-year survival of 21.6%. Regarding the prognosis of PAL of T-cell origin after chemotherapy and radiotherapy, the studies are too small to make a definite conclusion. Rihel and colleagues [3] used chemotherapy and radiotherapy, with 6 years of survival in follow-up. This contrasts with Hashizume and colleague [4], who applied only radiotherapy with resulting 2 months of survival. Fukino and colleagues [5] report a surgical case of non-Hodgkin lymphoma (T-cell PAL) treated postoperatively with combination chemoradiotherapy; the patient died 161 days later. In contrast, our patient received only chemotherapy (6 cycles of CHOP) postoperatively, and was alive at 15 months of follow-up.
In conclusion, PAL is a known entity and should always be suspected when there is a chest wall mass in chronic pyothorax, especially after iatrogenic pneumothorax for Koch lung. In selected patients in whom the chest wall is the only site of disease, surgical excision, followed by chemotherapy, may provide a favorable outcome.
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References
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- Nakatsuka S, Yao M, Hoshida Y, Yamamoto S, Iuchi K, Aozasa K. Pyothorax-associated lymphoma: a review of 106 cases J Clin Oncol 2002;20:4255-4260.[Abstract/Free Full Text]
- Narimatsu H, Ota Y, Kami M, et al. Clinicopathological features of pyothorax-associated lymphoma; a retrospective survey involving 98 patients Ann Oncol 2007;18:122-128.[Abstract/Free Full Text]
- Riehl G, Aubert A, Sandu C, Brichon PY. Malignant non-Hodgkin's lymphoma developing late after pneumonectomy Eur J Cardiothorac Surg 2006;30:948-949.[Abstract/Free Full Text]
- Hashizume T, Aozasa K, Tomita Y, Matsushita K. Pyothorax-associated T-cell lymphoma: a case report Jpn J Clin Oncol 2003;33:145-147.[Abstract/Free Full Text]
- 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;40:113-117.[Medline]
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Abstract
Introduction
