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Eur J Cardiothorac Surg 2001;19:589-593
© 2001 Elsevier Science NL

Primary chest wall tumors: early and long-term results of surgical treatment

^a Department of Thoracic and Vascular Surgery, Evangelismos General Hospital, Athens, Hellas, Greece
^b Department of Pathology, Evangelismos General Hospital, Athens, Hellas, Greece

Received 10 October 2000; received in revised form 16 February 2001; accepted 28 February 2001.

Corresponding author. 34A Konstantinoupoleos strasse, 15562 Holargos, Athens, Greece. Tel.: +30-1-6510388; fax: +30-1-6547695
e-mail: kalliopiath{at}iname.com

	Abstract

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 
Objectives: A retrospective study of primary chest wall tumors (PCWTs/CWTs) was conducted to review their clinical, radiological and pathological features, as well as the early and long-term results of surgical management. Materials and methods: From 1986 through 1996, 41 patients (18/44% male, 23/56% female, aged 15–78 years) with PCWTs were treated in our department. Results: Twenty-three patients (nine male, 14 female, mean age 36 years) had a benign CWT: enchondroma, five patients; fibrous dysplasia, four patients; neurilemoma, three patients; osteochondroma, two patients; granular cell tumor, two patients; fibroma, two patients; lipoma, two patients; fibrolipoma, one patient; eosinophilic granuloma, one patient; aneurysmal bone cyst, one patient. Eighteen patients (nine male, nine female, mean age 59 years) had a malignant CWT: plasmacytoma, five patients; chondrosarcoma, two patients; osteosarcoma, two patients; fibrosarcoma, two patients; desmoid tumor, two patients; leiomyosarcoma, one patient; malignant fibrous histiocytoma, one patient; tendon sheath sarcoma, one patient; hemangiosarcoma, one patient; neurinosarcoma, one patient. The ribs were involved in 21 patients, the sternum in five patients, and the soft tissue in 17 patients. Distinction between benign and malignant CWT was not possible using radiographic criteria alone, and diagnosis was always confirmed histologically. Surgical treatment consisted of wide resection in 29 patients (15 benign/14 malignant CWTs), with the use of synthetic mesh in five cases, and excisional or incisional biopsy in 12 patients. There was no perioperative mortality. Two patients with a benign CWT (fibroma, one patient; neurilemoma, one patient) had a local recurrence at 13 and 26 months after resection, respectively, and underwent wide resection (recurrence rate 8.7%). Follow-up at 3–13 years revealed one non-tumor-related death in patients with benign CWT (overall mortality rate 4.3%) and no other local recurrence. The overall 5- and 10-year survival in patients with malignant CWT was 33.3%. Conclusions: We believe that all CWTs should be considered malignant until proven otherwise. Wide resection with tumor-free margins is required in order to provide the best chance for cure in both benign and malignant lesions.

Key Words: Chest wall • Primary tumors • Chest wall tumors • Primary chest wall tumors

	1. Introduction

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 
Primary chest wall tumors (PCWTs, CWTs) are a heterogeneous group of tumors developing in the bones and soft tissues of the thoracic cage. These various tumors are often considered as a group because the diagnostic and therapeutic problems they pose are similar. Altogether, PCWTs make up from less than 1% to 2% of all primary tumors [1,2].

A retrospective study of PCWTs was conducted to review their clinical, radiological and pathological features, as well as the early and long-term results of surgical management.

	2. Material and methods

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 
During the period from January 1986 to December 1996, 41 patients with PCWT were diagnosed and treated at Evangelismos General Hospital in Athens. Patients with metastatic tumors of the chest wall or direct invasion from intrathoracic neoplasms to the chest wall were excluded. We retrospectively reviewed the clinical presentation, radiological features, pathological findings, surgical treatment and early and long-term results. Follow-up was completed for all patients. The mean follow-up time was 7.8 years (range 3–13 years).

Diagnostic work-up included a detailed history, a thorough physical examination and postero–anterior and lateral chest radiographs in all cases. A thoracic CT scan has been routinely performed since 1989, when CT became available, to delineate the extent of the bone, soft tissue, pleural and mediastinal involvement, helping us to plan surgical resection. A radionuclide bone scan was ordered in case of a bony CWT, suspected for malignancy, to rule out bone metastases. MRI was not necessary, since no case of spinal cord, mediastinal, or thoracic outlet involvement existed.

Our policy in treating PCWTs was, in general, as follows. For relatively small tumors requiring no major resection, excisional biopsy was performed, and if malignancy was proved during surgery, wide resection was performed. In large tumors (more than 4 cm), we proceeded either to incisional biopsy under local anesthesia, which would determine our operative plan at a second time, or, in case of bony tumors, incisional biopsy under general anesthesia was converted to wide resection of the tumor.

Wide resection of a malignant CWT included the affected rib with at least a 4–5 cm free margin proximally and distally to the tumor, portions of the ribs immediately above and below the tumor, the adjacent muscles, and the underlying pleura. Any other tissue adherent to the tumor was also excised. For sternal CWTs, the size and location of the tumor determined the extent of the resection. Lesions at the upper third of the sternum necessitated resection of the manubrium and most of the sternal body, as well as the medial ends of the clavicles and the adjacent sternocostal cartilages (subtotal sternectomy, two cases in our series). Lesions at the middle third of the sternum were treated with resection of the sternal body, with preservation of the manubrium and the xiphoid process (partial sternectomy, one case of ours).

Chest wall closure was achieved without difficulty in most cases. In four cases, partial transposition of latissimus dorsi (n=2) or pectoralis major muscles (n=2) was necessary to cover large defects of the lateral chest wall or sternum. Synthetic prostheses were used to cover larger chest wall defects (larger than 7x7 cm) and in one case of subtotal sternectomy, Marlex mesh with methylmethacrylate (‘sandwich’) was used in three cases, and a polytetrafluorethylene (PTFE) patch was used in two cases. The middle axillary line was the borderline for distinguishing anterior from posterior chest wall, and anterior from posterior parts of a rib.

	3. Results

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 
There were 18 male (44%) and 23 female (56%) patients, ranging in age from 15 to 78 years (mean 45 years). The histologic type of the PCWTs is shown in Table 1. Swelling and pain were the two most common symptoms, with many patients complaining of both (Table 2). Seven patients (17%) were asymptomatic, the tumor being discovered during routine radiographic examination or investigation for unrelated reasons. The duration of symptoms for benign CWTs was from 3 months to 4 years (mean 21 months), and for malignant CWTs from 4 weeks to 13 months (mean 9 months).

3.1. Benign CWTs
Twenty-three of the 41 patients (56%) had benign CWTs (Table 1). Nine were male (39.2%) and 14 were female (60.8%), with an age range of 15–63 years (mean 36 years). Fifteen patients (65.2%) had a bony or cartilaginous tumor (six male, nine female, mean age 32 years), and the remaining eight patients (three male, five female, mean age 44 years) had soft tissue CWT. Most of the bony tumors were located at the ribs (13/15, 86.6%), with almost equal distribution between anterior and posterior halves of the rib (7:6). The majority of the soft tissue tumors were located at the anterior chest wall (5/8). The maximum diameter of a benign CWT ranged from 2.5 to about 25 cm (mean 13 cm).

Surgical treatment consisted of incisional biopsy (n=1), excisional biopsy (n=7), or wide resection (n=15). In two cases (9.9%), a Marlex mesh with methylmethacrylate or a PTFE patch were used for the chest wall reconstruction. There was no perioperative morbidity or mortality. Two patients (8.7%), one with fibroma and the other with a huge neurilemoma, came back 13 and 26 months after excision with a palpable mass at the site of operation. They were submitted to wide resection. Histologic examination revealed recurrence of the fibroma in the first patient, and a fibrotic mass without evidence of nerve sheath cells in the patient with the previously resected neurilemoma. No re-recurrence has been noted 5.5 and 8 years after reoperation, respectively.

Follow-up of all 23 patients at 4–14 years after surgery revealed one death (the patient with an aneurysmal bone cyst), which was not related to the tumor (drug overdose).

3.2. Malignant CWTs
Eighteen of the 41 patients (44%) had a malignant CWT (Table 1). Nine were male (50%) and nine were female (50%), with an age range of 33–78 years (mean 59 years). The patients with a malignant CWT were significantly older than the patients with a benign CWT (mean age 59 vs. 36 years, respectively, P<0.001). The duration of symptoms (Table 2) was significantly shorter in patients with a malignant CWT compared to the benign CWT group (mean duration 9 vs. 21 months, respectively, P<0.001).

Half (9/18) of the malignant CWTs originated from bone cartilage (five male, four female, mean age 64 years), and the other half originated from soft tissues (five male, four female, mean age 54 years). Most of the bony tumors were located at the ribs (6/9, 66.7%) rather than the sternum (3/9), with equal distribution between anterior and posterior halves of the rib (3:3). Most of the soft tissue tumors were located at the posterior chest wall (6/9). The maximum diameter of a malignant CWT ranged from 2 to 16 cm (mean 6.5 cm).

Surgical management consisted of incisional biopsy (n=2), excisional biopsy (n=2), and wide resection (n=11). In three cases (17%), a Marlex mesh with methylmethacrylate (n=2) or a PTFE patch were required for the chest wall reconstruction. In one case, a wedge resection of the underlying lung invaded by the CWT was performed. There was no perioperative mortality or significant morbidity. Minor wound infection was observed in three cases (none among patients with prostheses) requiring only antibiotics and removal of some stitches. Follow-up at 3–13 years revealed 12 deaths within 5 years after surgery (5-year mortality 66.7%). All deaths were disease-related. All these patients developed metastases and two of them (11.1%) developed recurrence at the tumor site as well. The six survivors are alive and well 6–13 years after surgery (mean 10.2 years). The 5- and 10-year actuarial survival in patients with malignant CWT was 33.3%.

The five patients with solitary plasmacytoma were treated with incisional (n=2) or excisional biopsy (n=1), followed by irradiation of the tumor bed in all three cases, or wide resection (n=2). One of the latter two patients had a sternal solitary plasmacytoma and was submitted to subtotal sternectomy and reconstruction with Marlex mesh and methylmethacrylate. Four patients developed multiple myeloma within 2.5 years after surgery and underwent chemotherapy. The only 5-year survivor was the patient in whom the disease did not convert to multiple myeloma. The two patients with chondrosarcoma underwent radical excision (subtotal sternectomy in one of them). The tumors were low grade chondrosarcomas. Both patients are alive and disease-free 6 and 11 years after surgery.

The two patients with osteosarcoma were treated with wide resection. No detectable metastases were present at the time of surgery. One of them received adjuvant chemotherapy and both developed local recurrence and multiple lung metastases and died within 2 years after surgery.

All but one of the patients with soft tissue tumors were treated with wide resection. The patient with hemangiosarcoma had a local excision and adjuvant radiation therapy. The two patients with the desmoid tumors and the one with the tendon sheath sarcoma are alive and well 6, 9, and 7 years after surgery, respectively. All the others died within 4 years due to metastatic disease.

	4. Discussion

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 
PCWTs are rare. They are estimated to be accounted for only 0.04% of all new cancers diagnosed in the USA [1]. A review of the literature reveals that 60% of PCWTs are malignant [3]. In our series, benign CWTs are the majority (56%). The most common tissue of PCWT origin is bone or cartilage according to a review of 15 reports with 663 cases [3], but the Memorial Sloan–Kettering Cancer Center experience highlights the soft tissues [4]. In our material, bone cartilage and soft tissues contributed equally to malignancy. Ribs are a far more common location of CWTs (85–92%) than the sternum [1,4–7]. Sternal tumors are almost always malignant [7,8]. In our series, two of the five sternal tumors were benign.

Differential diagnosis between benign and malignant CWTs is very difficult preoperatively, and sometimes even after surgery under the microscope [6]. There are no reliable clinical features for distinguishing benign from malignant CWTs. A palpable mass and pain were common in both groups in our series. The duration of symptoms was significantly shorter in the malignant CWT group, and the patients were significantly older, but these features are far from being considered diagnostic [9].

Radiographic evaluation with chest radiographs and CT scan is essential [1,3,6–10]. A rapid increase in tumor size, cortical destruction, involvement of the surrounding tissues, and metastases suggest malignancy [1–3,7,8,10]. However, the vast majority of the malignant CWTs in our series lacked these features. A radionuclide bone scan can rule out multiple sites and should be performed before any major resection of a PCWT [1], or at least in the case of a medical chest wall tumor (plasmacytoma, Ewing's sarcoma) [8]. In our series, a bone scan was usually obtained if the CWT was located at the bones (i.e. plasmacytoma, chondrosarcoma, osteosarcoma). Fine needle aspiration yielded no satisfactory results in some of our cases, and has not been included in our diagnostic armamentarium. This is in accordance with the experience of others [1,5,6,10]. Core needle aspiration seems to be much more reliable [1,10], but it was not employed in our series.

Excisional biopsy is the preferred method of treatment of small PCWTs [1,6,7,10,11]. For larger CWTs, wide resection including the site of previous incisional biopsy is essential to successful management [6,7,12,13]. Wide resection means a margin of resection grossly free of tumor by several centimeters (4 cm at least). This wide resection may be adequate for benign or low grade malignant CWTs, like chondrosarcoma; however, in tumors of high malignancy, like osteosarcoma with the potential to spread within the marrow cavity, or along the periosteum or the parietal pleura, such a wide resection may be inadequate [8]. That is an explanation for our good results in chondrosarcomas and our poor results in osteosarcomas, all of which were treated with wide resection by the same surgical team.

For chondrosarcomas, overall 5- and 10-year survival is reported to be about 64 and 48%, respectively [13,14]. Early wide resection is the treatment of choice and can result in 10-year survival of up to 96% [14]. Of the risk factors influencing survival (tumor grade, diameter, and location [14]), we agree that low grade chondrosarcomas have excellent prognosis after early wide resection.

Osteosarcoma is a highly malignant CWT with poor results after radical surgical resection, with or without adjuvant chemotherapy (5-year survival 15%) [1,13]. Two-thirds of the patients present with metastases [1]. The prognosis was also poor in our two patients treated with wide resection and adjuvant chemotherapy.

Solitary plasmacytoma involving the chest wall is rather uncommon. In our series, it was the most common malignant lesion. Prognosis is determined by the conversion or non-conversion to multiple myeloma [1,15], as it was in our cases.

Soft tissue sarcomas have a poor prognosis, with a reported overall 5- and 10-year survival of 50–66 and 34%, respectively [16,17]. Tumor grade and differentiation are the most important factors affecting survival [16,17]. Wide resection is the treatment of choice [16,17], and in low grade tumors it can provide 5-year survival of up to 90% [17]. Adjuvant radio- and/or chemotherapy is considered for high grade sarcomas. The 5- and 10-year survival in our series was 33.3% (3/9). Of the patients who died, six had high grade tumors: fibrosarcoma, two cases; malignant fibrous histiocytoma, one case; leiomyosarcoma, one case; hemangiosarcoma, one case; neurosarcoma, one case.

Among soft tissue sarcomas, desmoid tumors are a special type. They are also best treated with wide resection with evidence of microscopically clear margins, because of their high local recurrence rate (up to 70%) [18]. Radiation therapy has been effective in recurrent desmoid tumors. Our two patients enjoy a disease-free life after wide resection of the tumor.

We conclude that all CWTs should be considered malignant until proven otherwise. Wide resection with tumor-free margins is required in order to provide the best chance for cure in both benign and malignant lesions.

	Footnotes

 
Presented at the 14th Annual Meeting of the European Association for Cardio-thoracic Surgery, Frankfurt, Germany, 7–11 October 2000.

	References

Top Abstract 1. Introduction 2. Material and methods 3. Results 4. Discussion References

 

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