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Ann Thorac Surg 2006;81:1214-1218
© 2006 The Society of Thoracic Surgeons

Original article: General thoracic

Non-Hodgkin's Lymphoma Presenting as a Large Chest Wall Mass

Po-Kuei Hsu, MD a , Han-Shui Hsu, MD b , * , Anna Fen-Yau Li, MD, PhD a , Liang-Shun Wang, MD a , Biing-Shiun Huang, MD, PhD a , Min-Hsiung Huang, MD a , Wen-Hu Hsu, MD a

a Division of Thoracic Surgery, Department of Surgery, Taipei Veterans General Hospital and National Yang-Ming University School of Medicine, Taipei, Taiwan
b Department of Pathology, Taipei Veterans General Hospital and National Yang-Ming University School of Medicine, Taipei, Taiwan

Accepted for publication November 21, 2005.

* Address correspondence to Dr Han-Shui Hsu, Division of Thoracic Surgery, Department of Surgery, Taipei Veterans General Hospital, No. 201, Sec. 2, Shih-Pai Rd, Taipei, Taiwan (Email: hsuhs{at}vghtpe.gov.tw).


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Malignant lymphoma presenting as a solitary chest wall mass is not frequently seen. Only a few case reports have been found in the English literature. The treatment for primary chest wall lymphoma remains unclear.

METHODS: From 1991 to 2004, of 157 patients with initial presentation of isolated chest wall mass, non-Hodgkin's lymphoma was diagnosed in 7 of them. Patients with tumors arising from axillary lymph nodes or mediastinal lymphadenopathy with chest wall extension were excluded in the study. The clinical manifestation, management, and outcome of these patients were reviewed.

RESULTS: There were 1 female and 6 male patients with a mean age of 66.5 years. The mean largest diameter of the mass was 10.3 cm. Four of these 7 patients had the chest wall lymphoma as the only site of disease. The other 3 patients had other organ involvement including lung, bone, or liver. The pathologic diagnoses were malignant lymphoma in 2 patients and diffuse large B-cell lymphoma in 5 patients. Three patients with chest wall lymphoma as the only site of disease had tumor excision followed by adjuvant chemotherapy. No recurrence or metastasis was noted for these 3 patients. The mean follow-up period was 102 months. The other patient with chest wall lymphoma as the only site of disease, who had chemotherapy as the initial treatment, remained free of disease for 6 months after treatment. The other 3 patients with other organ involvement who were managed with chemotherapy with or without radiotherapy died of disease after a mean survival of 20 months.

CONCLUSIONS: Malignant lymphoma presenting as a large chest wall mass is not common. Although the primary treatment of choice for lymphoma with or without chest wall involvement is chemotherapy, surgery followed by adjuvant chemotherapy can provide satisfactory outcome for some patients in whom the chest wall lymphoma was the only site of disease.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 References
 
Chest wall tumors arise from a wide variety of benign and malignant etiologies, and provide the clinician with a diagnostic and therapeutic challenge. The classification of chest wall tumors includes primary tumors, adjacent tumors with local invasion, metastatic lesions, and nonneoplastic disease. The majority of chest wall lesions are the result of metastasis or invasion from adjacent malignancies [1]. Among primary chest wall tumors, chest wall lymphoma is uncommon, accounting for less than 2% of chest wall soft tissue tumors [2, 3]. Few cases of primary malignant lymphoma arising from the pleura, the rib or the sternum have been reported [4–7]. Malignant lymphoma presenting as a solitary chest wall mass is not frequently seen [7, 8]. Only a few case reports have been found in the English literature. The treatment for primary chest wall lymphoma remains unclear. Here we report our experience in the management of 7 patients with non-Hodgkin's lymphoma presenting as a solitary chest wall mass.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
A retrospective survey of the admission data in Taipei Veteran General Hospital from 1991 to 2004 was conducted. There were 157 patients with an initial presentation of a chest wall mass. Among these 157 patients, primary non-Hodgkin's lymphoma was diagnosed in 7 of them. Diagnostic methods included fine-needle aspiration, incision biopsy, and excision biopsy. Patients with tumors arising from axillary lymph nodes or mediastinal lymphadenopathy with chest wall extension were excluded in the study. Complete workup included blood profiles, imaging studies (roentgenogram and computed tomography scan of chest), and radionuclide bone scan. After diagnosis was confirmed, all patients received bone marrow biopsy for lymphoma staging. The clinical manifestation, management, and outcome of these patients was recorded. Follow-up information was obtained from medical record or from telephone contact. Our Institutional Review Board approved this study and granted an exemption from informed consent on November 9, 2005.


   Results
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 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Table 1 shows the clinical manifestation of these 7 patients. There were 1 female and 6 male patients with a mean age of 66.5 years (range, 27 to 80). All patients presented with huge chest wall masses. The mean largest diameter of the mass was 10.3 cm. In 3 patients the masses were associated with painful sensation. No obvious enlarged lymph node could be found during physical examination in all patients. Elevated lactate dehydrogenase (LDH) level was found in 5 patients. The mean LDH level was 246 u/L (range, 148 to 562 u/L). Chest radiographs showed pleural-based soft tissue shadow (Fig 1). A computed tomography scan of the chest revealed a huge tumor mass arising from chest wall (Fig 2). Osteolytic destruction of the ribs was found in 3 patients. Only 1 of these 3 patients had painful sensation in the presentation. Lung and liver involvement was found in 1 patient. Radionuclide bone scan study showed increased radioactivity of the mass lesion in 6 patients and multiple bone metastasis in 3 patients.


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  		Table 1. Clinical Data of 7 Patients With Malignant Lymphoma Presenting as Solitary Chest Wall Mass
 

Figure 1
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  		Fig 1. A 27-year-old man presented with left side chest wall mass without tenderness. Chest radiograph showed a soft-tissue shadow about 5 cm in diameter over the left lower chest wall (arrow). Pathology examination revealed a diffuse large B-cell lymphoma.

 

Figure 2
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  		Fig 2. A 62-year-old man presented with a large right side chest wall mass. Computed tomography scan of the chest disclosed a soft-tissue mass lesion with osteolytic bone destruction at the right 11th rib (arrow). Pathology examination proved a diffuse large B-cell lymphoma.

 
Fine-needle aspiration was performed in 6 patients. Only 1 patient had a definite diagnosis of malignant lymphoma. The diagnostic rate was only 16.6%. One patient had incision biopsy for diagnosis without needle aspiration. For the other 5 patients, diagnosis was obtained by excision biopsy in 3 and incision biopsy in the other 2 patients. The pathologic diagnoses were malignant lymphoma in 2 patients and diffuse large B-cell lymphoma in 5 patients (Fig 3). The lymphoma cell was positive for LCA (leukocyte common antigen) and L26 (CD20) and negative for CD3 and UCHL1 (CD45) marker in the immunohistochemical analysis. There was no Hodgkin's disease in our study. All patients received bone marrow biopsy for complete staging, and none had malignant cells found in the bone marrow.


Figure 3
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  		Fig 3. (A) Microscopic examination showed malignant lymphoma made up of large cells with vesicular nuclear and prominent nucleoli, consistent with the diagnosis of diffuse large B-cell lymphoma (hematoxylin and eosin; original magnification x400). (B–D) The tumor cells were immunocytochemically positive for LCA and L26, and negative for CD3 (original magnification x400).

 
For 3 patients with the chest wall lymphoma as the only site of disease, complete surgical resection followed by chemotherapy was carried out. One patient had a solid tumor in the lower left chest wall with the involvement of the 8th to the 10th ribs. The chest wall defect (15 x 12 cm) was repaired with a latissimus dorsi muscle flap without prosthesis. Another patient had a huge tumor in the right posterior chest wall with the involvement of the 6th to the 10th ribs. The chest wall defect (20 x 15 cm) was also repaired with a muscle flap. The other patient who had a huge tumor in the lower right chest wall invading the 9th to the 12th ribs, latissimus dorsi, and serratus posterior muscle received wide resection. Two-layer Marlex mesh (Bard, Cranson, RI) was used to repair the chest wall defect (15 x 12 cm). The surgical margins were at least 2 cm away from the tumors in these 3 patients. The adjuvant chemotherapy regimen was CHOP (C: cyclophosphamide, H: doxorubicin, O: vincristine, P: prednisolone) in 2 patients and CEOP (E: epirubucin) in 1 patient. No recurrence or metastasis was noted at the end of the study. All 3 patients are still alive. The mean follow-up period was 102 months. The other 4 patients were managed with medical treatment after the diagnosis was made. Chemotherapy regimen was CHOP in 3 patients and COP in 1 patient. Tumor shrinkage was observed in 3 of them. Two patients died of chemotherapy-related complications (sepsis due to central line infection in 1 patient, and neutropenic pneumonia in the other) 7 months after treatment initiated. One patient died of tumor recurrence 46 months after treatment. The other patient with chest wall lymphoma as the only site of disease, who had chemotherapy as the initial treatment owing to old age and substantial operative risk, remained free of disease for 6 months after treatment.


   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
The incidence of thoracic involvement, including mediastinal lymphadenopathy, lung parenchyma, and chest wall, in patients with lymphoma was not unusual according to Press and colleagues [8]. In their report, 4 of 250 patients with lymphoma had the chest wall as the only site of disease (1.6%), consisting of 3 Hodgkin diseases and 1 non-Hodgkin's lymphoma. However, some authors reported that in few cases of primary lymphoma from the rib or sternum, non-Hodgkin's lymphoma, particularly large cell lymphoma is more common [6, 9, 10]. In this study, we reported 7 cases of non-Hodgkin's lymphoma presenting as a large chest wall mass with male predominance and mean age of 66.5 years, including 4 patients in whom chest wall lymphoma was the only site of disease. The mean largest diameter of these tumors was 10.3 cm. In patients with chest wall tumors, extensive chest wall resection with reconstruction was needed, which carried some operative risk in elderly patients.

Whether patients with chest wall lymphoma as the only site of disease should receive surgical resection is unclear. Patients with lymphoma are usually treated with chemotherapy or local irradiation. Hodgson and associates [11] reported the outcome of 324 patients with clinical stage I-II Hodgkin's lymphoma treated with chemotherapy and local irradiation. They found that patients with chest wall invasion had poor local control and survival. However, Ryan and coworkers [9] in their series, reported that 1 patient with primary soft tissue lymphoma of the anterior chest wall involving the first four ribs remained free of disease 49 months after surgical resection. In another case report from Lones and colleagues [6], a 71-year-old man with large cell lymphoma arising from the rib was successfully managed with surgical resection and local irradiation without tumor recurrence. In 2000, Nishiyama and associates [12] also reported a 77-year-old woman who had a painless huge mass over the anterior chest wall. Surgical excision was performed with an uneventful recovery. The postoperative diagnosis of the tumor was non-Hodgkin's lymphoma. Unfortunately, the patient died of local and distant recurrence 9 months after operation. In our series, 3 of the 4 patients with isolated chest wall lymphoma as the only site of disease were managed with surgical resection and adjuvant chemotherapy. No tumor recurrence was recorded during the follow-up period. The other patient who received chemotherapy for the initial treatment remained free of disease 6 months after treatment. For 3 patients having chest wall lymphoma with other focal lesions like bone or liver, only chemotherapy plus radiation were given. The outcome for these patients was poor.

The role of fine-needle aspiration or nonexcision biopsy for the diagnosis of primary chest wall tumor was undetermined [1, 3, 13, 14]. Caution is warranted when using fine-needle biopsy as a diagnostic modality as diagnosis by cytologic specimen is sometimes unsatisfactory [15, 16]. A wrong diagnosis from inadequate tissue specimens may even lead to less than optimum treatment and a worse prognosis [17]. Meanwhile, the risk of tumor implantation made fine-needle biopsy an unpopular tool in the diagnosis of primary chest wall tumors [18, 19]. Some authors suggest that all patients with primary chest wall tumors should receive at least excision biopsy, whereas patients highly suspected of having malignancy should receive wide radical resection or subsequent resection for safe margins [20–25]. Nonexcision biopsy should be reserved for patients in whom metastatic origin and hematologic disease is suspected, in whom aggressive surgical resection is less beneficial [26, 27]. In our study, the diagnosis was obtained in only 1 of 6 patients with chest wall lymphoma having fine-needle aspiration (16.7%). Most of the patients received, at least, incision biopsy to make the definite diagnosis. For 3 patients without other focal lesions, surgical excision was not only diagnostic but also therapeutic as the malignant potential of the tumors was highly suspected. Adjuvant chemotherapy was given when the diagnosis of lymphoma was confirmed. The outcome for these 3 patients was excellent.

Lymphoma is considered extranodal if it presents with the main bulk of disease in an extranodal site. The gastrointestinal tract is the most common site of extranodal lymphoma followed by the Waldeyer‘s ring. Lymphomas of bone and soft tissue are relatively rare [28]. In our study, most of the patients have B-cell lymphoma. The diffuse large B-cell lymphoma constitutes about 30% of all non-Hodgkin's lymphoma. The B-cell symptoms (fever, night sweats, body weight loss) are observed in 30% of patients and serum LDH is elevated in more than half the patients. Disseminated disease is noted in about 40% of patients, and bone marrow involvement is found in only 10% to 20% of patients [29]. For extranodal non-Hodgkin's lymphoma, local therapy can be used for local control or curative intent. The recognition of distant metastasis mandates the use of systemic chemotherapy. Although the main modalities used in the treatment of non-Hodgkin's lymphoma are irradiation and chemotherapy, surgical resection could be used in the diagnosis and management of some selected extranodal locations [28]. Because tumor burden is a important predictor for survival in patients with lymphoma, surgery as a front-line therapy in patients with resectable early-stage diffuse large-cell lymphoma has been proposed. Romagurea and associates [30] reported that surgical debulking is associated with improved survival in stage I-II diffuse large-cell lymphoma.

In conclusion, primary chest wall lymphoma presenting with a large chest wall mass is not common. The diagnostic rate using fine-needle aspiration for the diagnosis of chest wall lymphoma is low. Although the primary treatment of choice for lymphoma with or without chest wall involvement is chemotherapy, surgery followed by adjuvant chemotherapy can provide satisfactory outcome in some patients in whom the chest wall lymphoma was the only site of disease.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

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