Diffuse large Bcell lymphoma presenting as ... - Wiley Online Library

Thoracic Cancer ISSN 1759-7706

CASE REPORT

Diffuse large B-cell lymphoma presenting as large anterior chest wall mass involving pleura and lung: A possible result of post-traumatic chronic inflammation Fouad Atoini1, Aziz Ouarssani2, Moulay Ahmed Hachimi3, Fatima Aitlhou2, Fatima Zohra Guenoun4, Charafeddine Elmejereb5, Mustapha Idrissi Rguibi2 & Mohammed Baaj6 1 2 3 4 5 6

Department of Thoracic Surgery, Military Hospital Moulay Ismail, Meknes, Morocco Department of Pulmonology, Military Hospital Moulay Ismail, Meknes, Morocco Department of Anesthesiology, Military Hospital Moulay Ismail, Meknes, Morocco Private Centre of Anatomopathology (Laboratory of GUENOUN), Meknes, Morocco Department of Anatomopathology, Military Hospital Moulay Ismail, Meknes, Morocco Department of Internal Medicine and Hematology, Military Hospital Moulay Ismail, Meknes, Morocco

Keywords blunt trauma; chest wall; chronic inflammation; diffuse large B-cell lymphoma; lymphoma. Correspondence Fouad Atoini, Department of Pulmonology and Thoracic Surgery, Military Hospital Moulay Ismail, Postal box: S 15, Meknes, Morocco. Tel: +212 661 44 38 46 Fax: +212 535 517 399 Email: [email protected] Received 5 June 2011; accepted 27 June 2011. doi: 10.1111/j.1759-7714.2011.00061.x

Abstract A 67 year-old-man was hospitalized due to chronic pain and a large mass on the anterior chest wall. His medical history showed chest trauma in 1970, the reconstitution of the scenario revealed there was blunt trauma with swelling and rib fracture on the same side. Physical examination revealed an isolated large anterior chest wall mass. Chest radiography showed two bilateral irregular masses, chest computed tomography showed a large right chest wall tumor with pleural effusion, nodules of the right upper lobe and tumor of the left lower lobe without mediastinal lymphadenopathy. Whole body exploration showed only the chest disease. Transthoracic biopsy showed inflammatory reaction. Surgical biopsy by anterior thoracotomy of the right mass was performed under general anesthesia. Histological and immunohistological analysis revealed lymphoid diffuse large cell proliferation with positive staining of CD 20, BCL-6 and MUM1, confirming the diagnosis of diffuse large B-cell malignant lymphoma. Chemotherapy based on CHOP-21 (cyclophosphamide, doxorubicin, vincristine, and prednisolone) was administered with good response after three cycles. The patient was discharged under surveillance in good condition after the end of chemotherapy. We report an infrequent neoplasm with an unusual and subtle clinical presentation.

A 67 year-old-man was admitted to our department for a painful large right anterior chest wall mass who had for 2 months in the context of weight loss of 5 kg. His medical history showed chest trauma and tobacco smoking (30 package/year) weaned 10 years prior to presentation. Questioning about the chest trauma which had occurred in 1970 found that the patient has suffered a fall from height of 2 m onto a metal object causing acute pain and swelling of the chest wall. This swelling had been neglected with the patient self medicating with analgesic treatment. No interventions were made at the time. The swelling was renitent with moder-

ate chronic chest pain certainly related to a rib fracture associated with hematoma of the chest wall. Clinical examination at admission revealed a 12x7 cm hard mass poorly defined and localized to the right anterior chest wall without inflammatory signs. The patient was in fairly good general condition and apyretic. The lymph nodes, subclavicular and cervical region were without anomaly, as were abdominal and pelvic examinations. Chest radiograph (Fig. 1) showed two abnormal irregular opacities localized to the right parahilar and in the left lower lobe. Chest computed tomography (CT) showed two abnormal masses with irregular contours, the first mass was larger and confined to the right chest wall with bone destruction and the old rib fracture (arrow in Fig. 2),

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© 2011 Tianjin Lung Cancer Institute and Blackwell Publishing Asia Pty. Ltd

Case report

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Diffuse large chest wall B-cell lymphoma

Figure 1 Chest radiograph showing two opacities localized to the right parahilar and left lower lobe.

this mass was associated with pleural effusion and nodules of the upper lobe, the second mass was localized to the left lower lobe; there was no lymphadenopathy after analysis of the mediastinum. On histology fine needle biopsy of the right mass showed an inflammatory reaction without malignancy. Laboratory tests showed an increased rate of C-reactive protein, alkaline phosphate and gamma GT. Specific antigens of the prostate and other tumor markers (cancer antigen (CA) 125, CA 19-9, CA 15-3, carcinoembryonic antigen, alpha-1-foetoproteine and chorionic gonadotropin) were normal. The patient underwent surgical biopsy of the mass. Anterior thoracotomy without opening of the intercostal space showed a large ill-defined whitish mass with bone lysis. Large biopsies were performed associated with curettage of

Figure 2 Chest computed tomography showing large chest wall tumor and the old defect of the rib (arrow) with pleural effusion, nodules of the right upper lobe and pulmonary contralateral tumor.

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bone. Histological examination showed a diffuse large cell proliferation of lymphoid appearance. Immunohistochemical analysis using CD3, CD20, CD10, BCL6, and MUM1 revealed an intense positive membrane staining in a honeycomb pattern for CD 20 and moderate to intense and diffuse nuclear staining for BCL 6 and MUM1, thereby confirming the diagnosis of diffuse large B-cell malignant lymphoma (DLBCL) of the subtype non-germinal center phenotype (Fig. 3). Staging, including abdominal and pelvic CT, was normal. The patient underwent chemotherapy administered at 3 week intervals for a total of six cycles, based on CHOP-21: cyclophosphamide 750 mg/m2 i.v. day 1; doxorubicin 50 mg/m2 i.v day 1; vincristine 1 mg/m2 i.v day 1; and prednisolone 100 mg days 1–5. Evaluation after three cycles revealed the disappearance of the tumor on chest radiograph and chest-CT showed an objective response > 90% (Fig. 4). The patient was discharged to follow up after the end of chemotherapy.

Discussion Trauma as a predisposing factor of tumors has been a controversial subject. Barnett et al.1 claimed that prolonged or repeated inflammation caused by trauma may induce cellular atypia leading to neoplasm. Although some researchers argue that the inflammatory process leads to carcinogenesis.2 Non-Hodgkin’s lymphoma can have multiple etiologies, including immunodeficiency, viral infections (such the Epstein Barr virus), radioactive contaminations, and hybrid genes resulting from translocation.3 There have been several reports in which lymphoma developed after blunt trauma, the majority were described in the head and neck;3–5 in the chest wall, the literature presents cases of lymphoma in patients with a long history of chronic tuberculous pyothorax or a history of artificial pneumothorax, and most cases are reported in Japan.6 These lymphoma have been designated pyothorax-associated lymphoma and show features of DLBCL similar to those developed from other chronic inflammatory conditions,7 such as the use of metallic implants in bones and joints,8 chronic osteomyelitis,9 chronic venous ulcer9 and the use of surgical mesh implant.10 In the recent 2008 world health classification of tumors of hematopoietic and lymphoid tissues, DLBCL associated with chronic inflammation was defined as a distinct entity.11 In many reported cases, lymphomas were diagnosed 1 to 2 months after trauma,3,4 but the etiological role of trauma in the development of lymphoma is not well understood. After chronic inflammation, DLBCL has been diagnosed tardily, like those associated with tuberculous pyothorax; in a recent Chinese report by Loong et al.,7 the authors describe four cases of DLBCL associated with chronic inflammation, one of those patients had a long history of knee trauma, with a total knee replacement for flexion deformity of the right knee due


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Figure 3 (a) Microscopic examination showed a diffuse large cell proliferation of lymphoid appearance (hematoxylin and eosin ¥40). (b) Positive membrane staining of CD 20. (c) Positive nuclear staining of BCL6. (d) Positive nuclear staining of MUM1.

to septic arthritis 32 years prior to DLBCL and replacement of the prosthesis 16 prior. In our case, the age of trauma was 41 years, history-taking of the scenario showed that the patient suffered a rib fracture with chest wall hematoma but this injury was neglected, treated with analgesic only and without medical supervision. The course of post-traumatic chronic inflammation can lead to tumor proliferation, but the beginning and the exact timing of this course cannot be determined. The argument in favor post-traumatic chronic inflammation leading to tumor in our case is: 1 The delay of lymphoma after post-traumatic chronic inflammation. 2 Primary localized lymphoma of the chest wall with pleural effusion, the homolateral pulmonary nodules and the contralateral pulmonary mass was considered metastasis. 3 The occurrence of lymphoma at the same side of the old trauma (right anterior chest wall)

4 The histology of DLBCL with a consistent non germinal center cell immunophenotype similar to the finding in pyothorax-associated lymphoma,12 the prototype of DLBCL associated with chronic inflammation. In conclusion our patient was diagnosed with DLBCL of the anterior chest wall with locoregional involvement. The long history of trauma with certain post-traumatic chronic inflammation led us to propose this mechanism in the category of DLBCL associated with chronic inflammation. This case highlights an unusual and subtle clinical presentation, and highlights the importance of anamnesis especially in elderly and the importance of supervising thoracic trauma in the long term; persistent swelling after chest trauma should not be overlooked.



Disclosure No authors report any conflict of interest.

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Figure 4 Chest radiograph and computed tomography showing objective response after three cycles of chemotherapy with CHOP-21 (cyclophosphamide, doxorubicin, vincristine, prednisolone).

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6 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. 7 Loong F, Chan AC, Ho BC et al. Diffuse large B-cell lymphoma associated with chronic inflammation as an incidental finding and new clinical scenarios. Modern. Pathology 2010; 23: 493–501. 8 Cheuk W, Chan ACL, Chan JKC et al. Metallic implant-associated lymphoma. A distinct subgroup of large B-cell lymphoma related to pyothorax-associated lymphoma? Am J Surg Pathol 2005; 29: 832–6. 9 Copie-Bergman C, Niedobitek G, Mangham DC et al. Epstein-Barr virus in B-cell lymphomas associated with chronic suppurative inflammation. J Pathol 1997; 183: 287–92. 10 Fujimoto M, Haga H, Okamoto M et al. EBV-associated diffuse large B-cell lymphoma arising in the chest wall with surgical mesh implant. Pathol Int 2008; 58: 668–71.


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11 Chan JKC, Aozasa K, Gaulard P. DLBCL associated with chronic inflammation. In: Swerdlow SH, Campo E, Harris NL et al. (eds). WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. IARC Press, Lyon 2008; 245–6.

12 Petitjean B, Jardin F, Joly B et al. Pyothorax-associated lymphoma. A peculiar clinicopathologic entity derived from B cells at late stage of differentiation and with occasional aberrant dual B- and T-cell phenotype. Am J Surg Pathol 2002; 26: 724–32.



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