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Ann Thorac Surg 2007;83:236-240
© 2007 The Society of Thoracic Surgeons
Department of Thoracic and Vascular Surgery and Heart-Lung Transplantation, Marie Lannelongue Hospital, Le Plessis Robinson, France
Accepted for publication August 18, 2006.
* Address correspondence to Dr Fadel, Department of Thoracic and Vascular Surgery and Heart-Lung transplantation, Marie Lannelongue Hospital, 133 Avenue de la Résistance, 92350 Le Plessis Robinson, France (Email: fadel{at}ccml.com).
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Abstract |
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 Top Abstract IntroductionMaterial and MethodsResultsCommentReferences |
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METHODS: We reviewed all patients presenting with a pathologic diagnosis of PPL in our institution during the past 20 years. We analyzed the outcome and determined the impact of complete versus incomplete surgical resection on survival.
RESULTS: The study included 17 patients with PPL confined to the pulmonary parenchyma. Pathologic diagnosis was low-grade B-cell PPL in 14 patients, high-grade B-cell PPL in 2, and lymphomatoid granulomatosis in 1. A complete resection was performed in 8 patients presenting with low-grade B-cell PPL and in 2 patients with high-grade B-cell PPL. The overall survival of patients presenting with low-grade B-cell PPL was 63% at 10 years; however, survival at 10 year tended to be better when a complete resection was performed (87.5% vs 25%, respectively; p = 0.08). Gender, bilateral disease, or adjuvant therapy did not affect survival. Both patients presenting with high-grade B-cell PPL are alive and free of disease 22 and 36 months after the surgery, respectively. The patient presenting with lymphomatoid granulomatosis is alive after 2 years of follow-up.
CONCLUSIONS: PPL is a rare disease that may be localized or diffuse in one or both lungs. Surgery should be the treatment of choice in the localized form of PPL if complete resection can be achieved. A complete resection is associated with an excellent long-term survival of almost 90%. If the lesions are diffuse or involve both lungs, medical therapy should then be the treatment of choice.
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Introduction |
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TopAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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PPL is a rare entity accounting for only 0.5% to 1% of all lung malignancies and less than 1% of non-Hodgkin lymphomas [1]. Histologically, most PPL are classified as mucosa-associated lymphoid tissue (MALT) according to the latest classification of the World Health Organization (WHO) Classification [2], and can be referred to as BALTOMAs (for bronchial-associated lymphoid tissue affection) or pulmonary MALTOMA [3]. MALT lymphomas are low-grade B-cell lymphoma, formerly known as "pseudolymphoma" because of its indolent course. However, a higher-grade of B-cell lymphoma (large cell lymphoma) can occasionally be seen in association with MALT.
In the literature, MALT lymphoma accounts for 58% to 87% of all PPL [1]. Despite the similarities with gastric MALTOMAs, where a chronic antigenic stimulation related to Helicobacter pylori infection has been demonstrated, no such relationship has been established for low-grade B-cell PPL in the lung. High-grade B-cell PPL is less frequent, 11% to 19% of the PPLs in published series [4, 5], and has a worse prognosis than low-grade PPL. It usually occurs in immunosuppressed patients, is often seen in HIV patients or transplant recipients, and is related to Epstein-Barr virus infection [6]. Lymphomatoid granulomatosis is very rare (<1000 cases reported in the literature) and has a very variable course, ranging from spontaneous remission to death. Treatment is essentially medical, and the surgeon is only involved for diagnostic purposes [7, 8].
To increase the reports on this rare disease and to define the role of surgery in the treatment of PPL, we reviewed all patients presenting with a pathologic diagnosis of PPL in our institution during the past 20 years. We analyzed the outcome and determined the impact of a complete versus incomplete surgical resection on survival.
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Material and Methods |
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TopAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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The diagnosis of PPL was based on characteristic histologic and immunophenotypical features according to the Kiel classification and the WHO classification [2]. Immunophenotyping was determined by immunohistochemical staining, and clonality was characterized according to the type of immunoglobulin on the malignant cell surface. Malignant cells were tested for CD3, CD5, CD10, L26 (CD20), and UCHLl (CD45RO) antigen expression. The records of the patients fulfilling criteria for PPL were reviewed, and clinical data were collected as well as medical history, laboratory data, radiologic findings at the time of diagnostic, diagnostic procedure, treatment, and follow-up.
Duration of follow-up was calculated from the date of diagnosis to the date of death or to the last follow-up visit. Patient survival was estimated using the Kaplan-Meier method. Differences in survival were compared using the log-rank test. A value of p = 0.05 was considered significant.
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Results |
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Treatment
Sixteen patients had a thoracotomy (Table 2). One patient presenting with bilateral disease underwent a thoracoscopic lung biopsy. Patients presenting with low-grade B-cell PPL underwent wedge resections (n = 6), lobectomy (n = 6), or lobectomy associated with wedge resection (n = 2). Complete resection was achieved in 8 patients. The resection was incomplete in 6 patients because of diffuse involvement of both lungs (n = 3) or multiple lobes within the same lung (n = 3).
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Adjuvant chemotherapy was given to both patients with high-grade B-cell lymphoma. Six patients (43%) with low-grade B-cell lymphoma received adjuvant chemotherapy (n = 3), adjuvant radiation therapy (n = 2), or adjuvant chemoradiation therapy (n = 1). Eight patients with low-grade B-cell lymphoma who underwent complete resection did not receive any adjuvant therapy. The patient presenting with lymphomatoid granulomatosis did not receive any postoperative treatment after lung wedge resection.
Survival
The overall survival of patients presenting with low-grade B-cell PPL was 63% at 10 years; however, the 10-year survival rates reached 87.5% in patients who had complete resection and tended to be better than in patients who had an incomplete resection (10-year survival, 25%; p = 0.08; Fig 3). Gender, bilateral disease, or the administration of adjuvant therapy did not affect survival. During follow-up, 5 patients died, 1 from disease-related cause and 4 from unrelated causes. Nine patients with low-grade B-cell PPL are alive and free of disease. Both patients presenting with high-grade B-cell PPL are alive and free of disease at 22 and 36 months after the surgery, respectively. The patient presenting with lymphomatoid granulomatosis is alive after 2 years.
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Comment |
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TopAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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Chronic inflammatory factors have been advocated to explain the development of MALT lymphoma, such as H pylori infection in the gastric location [10]. Chronic infection, smoking, or autoimmune disease may play a role in the development of B-cell PPL. Interestingly, 2 of our patients had been previously treated for gastric MALT lymphoma.
Our data are consistent with the literature regarding patient age and the proportion of low-grade to high-grade PPL [4, 9, 11, 12]; however, we observed a larger proportion of symptomatic patients than in other series [13]. Both patients with high-grade lymphoma and 10 of 14 patients with low-grade lymphoma were symptomatic in our experience.
The diagnosis is often made at the time of surgery because radiologic signs are usually not specific and bronchoscopy has a low diagnostic yield [4]. The presence of lymphocytic alveolitis with more than 10% of B cells in bronchoalveolar lavage can be diagnostic [14]. Preoperative CT-guided fine needle aspiration biopsy allows diagnosis of PPL in only 25% of patients [8]. The diagnosis is thus often made intraoperatively on frozen section and confirmed postoperatively by immunostaining.
A preoperative or postoperative work-up should be performed in all patients, including abdominal and head and neck CT scans and a bone marrow biopsy. By definition, all patients should have negative results on extrathoracic imaging and the bone marrow study to meet the criteria for PPL. If significant (>1 cm) mediastinal lymph nodes are found on a preoperative chest CT scan, a mediastinoscopy should be performed first to obtain a diagnosis. If the diagnosis of PPL is made on frozen section, a mediastinal lymph node dissection should be performed at the time of surgery, even though in our experience the result was negative in most patients.
Although survival is worse for patients with high-grade PPL than in those with low-grade PPL, published reports show that more than half of patients with high-grade PPL can achieve survivals of 8 to 10 years [15]. Both of our patients with high-grade lymphoma are currently alive, with no recurrence 22 and 36 months after pneumonectomy performed to achieve complete resection. In both cases, the decision to perform a pneumonectomy was based on the possibility to perform a complete resection in the absence of response to chemotherapy.
The natural history of low-grade lymphoma appears to be slow, and its treatment is controversial [16]. Some authors advocate no therapy because of its indolent course, whereas others recommend surgery or chemotherapy, or both [1, 17]. Recent series did not demonstrate any difference in survival between surgery and chemotherapy or a combination [4, 9, 11, 12, 18]. The treatment was not always detailed, however, and the follow-up varied according to the series. In addition, the type of treatment may be influenced by the presence of multifocal or bilateral disease. The number of patients with multifocal disease varies between 24% and 70% according to the reported series [13, 19, 20].
We believe surgery should be the treatment of choice if a complete resection can be achieved. A complete resection was associated with a 10-year survival of almost 90% in our series, and survival tended to be better than in patients who underwent partial resection only. Patients presenting with diffuse involvement of one or both lungs should be treated with chemotherapy or chemoradiation therapy. Although some authors have recommended a pneumonectomy for multiple lesions of low-grade PPL involving one lung, in our view this option may be too aggressive because of the indolent course of the disease. We find, however, that a complete resection is an option in limited bilateral disease, as in one of our patients.
In conclusions, PPL is a rare disease that may be localized or diffuse in one or both lungs. Surgery should be the treatment of choice in the localized form of PPL if complete resection can be achieved. A complete resection was associated with a 10-year survival of almost 90% in our series. If the lesions are diffuse or involve both lungs, medical therapy should then be the treatment of choice.
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References |
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This article has been cited by other articles:
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  A. I. Hoeritzauer, L. Venkatraman, K. M. Manus, P. Kettle, S. Sah, and S. Elborn Bronchial associated lymphoid tissue (BALT) lymphoma presenting as chronic lung sepsis BMJ Case Reports, September 6, 2009; 2009(sep06_2): bcr0320091716 - bcr0320091716. [Abstract] [Full Text]
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