Gen Thorac Cardiovasc Surg DOI 10.1007/s11748-014-0387-9

ORIGINAL ARTICLE

Rib tumors: a 15-year experience Timothy Sakellaridis • Stylianos Gaitanakis Anastasios Piyis



Received: 11 November 2013 / Accepted: 27 February 2014 Ó The Japanese Association for Thoracic Surgery 2014

Abstract Objective A retrospective study of rib tumors was conducted to review their clinical, radiological, and pathological features, the difficulties in differentiating benign from malignant tumors, as well as the early and long-term results of surgical management. Methods All patients with rib lesions evaluated by the Thoracic Surgery Department from 1998 to 2012 were studied. The patient’s age, sex, symptoms, radiologic evaluation, surgical procedure, pathologic diagnosis and follow-up were assessed. Results Ninety-one patients (81 male, 10 female, age range 16–80) with rib tumors underwent surgery in a period of 15 years (1998–2012). 64 patients (70.33 %) had benign lesions and 27 patients (29.67 %) had malignant tumors. In the group with malignant tumors, the main symptom was pain, and in the group with benign tumors the main symptom was swelling. Ten patients with benign rib tumor and two with malignant tumor were detected during routine chest radiograph. All patients were treated surgically with wide excision of the tumor and the diagnosis was established histologically. In the benign cohort, osteochondromas, fibrous dysplasia, enchondroma, eosinophilic granuloma and posttraumatic fibro-osseous lesion/ dysplasia were among the most customary diagnoses. In

The above manuscript was presented as oral presentation in the 27th EACTS Annual Meeting, 5–9 October 2013, Vienna, Austria. T. Sakellaridis  S. Gaitanakis  A. Piyis Thoracic Surgery Department, 401 General Army Hospital, Athens, Greece T. Sakellaridis (&) 91-95 Antoniou Tritsi, 15238 Halandri, Greece e-mail: [email protected]

the malignant cohort, 13 patients (48.15 %) had metastatic lesions, with the remaining 14 patients having primary malignant rib tumor. Conclusions Although radiographic imaging has evolved, all rib lesions must be considered as potentially malignant until proven otherwise. Prompt intervention is necessary and surgery must consist of wide resection with tumor-free margins to provide the best chance for cure in both benign and malignant lesions. Keywords Rib tumors  Thoracic wall  Benign lesions  Primary tumors  Metastatic lesions

Introduction Primary chest wall tumors are uncommon, accounting for only 0.04 % of all new cancers diagnosed and 5 % of all thoracic neoplasms [1–3]. Primary tumors of the rib comprise 5–7 % of all primary bone neoplasms, but make up 50 % of bony malignant tumors and the majority of benign bony tumors of the chest wall [2, 4]. Most of the rib lesions are malignant tumors, with metastatic involvement or direct invasions from adjacent malignancies such as breast cancer, lung cancer, mesothelioma and mediastinal tumor being the commonest. When metastases are excluded, tumors arising from the rib are as likely to be benign as malignant [2]. Fibrous dysplasia and chondroma are the most frequently detected benign rib lesions. Of malignant tumors, chondrosarcoma and osteosarcoma are most prominent in adults, whereas in children Ewing’s sarcoma is the most common tumor [2, 4–7]. Usually patients with rib lesions present with a palpable mass, pain or both. Less commonly, asymptomatic patients are diagnosed incidentally after

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routine imaging as part of a screening or investigation of an unrelated condition [1, 2, 5]. Although rib abnormalities are often initially identified on chest radiography, computed tomography (CT) and magnetic resonance imaging (MRI) are valuable in determining the boundary of tumor and detecting invasion to the adjacent organs, nerves and vessels [4]. Management consists of wide resection in cases of malignant rib lesions and possible reconstruction of the thoracic wall, and resection of only the affected rib in cases of benign rib lesions [8]. Because of their rarity and often benign presentation, rib tumors can present both a diagnostic and therapeutic dilemma. A retrospective study of rib tumors that were managed in our department was conducted to review their clinical, radiological and pathological features, the difficulties in differentiating benign from malignant tumors, as well as the early and long-term results of surgical management.

between 16 and 80 years (mean age: 31.2 years). 64 patients (70.33 %) had benign lesions (59 males and 5 females with their age ranging between 18 and 68 years; mean age 25.81 years) and 27 patients (29.67 %) had malignant tumors (22 males and 5 females with their age ranging between 16 and 80 years; mean age 44.1 years). The pathologic features of all rib tumors are demonstrated in Table 1. The symptoms of the patients with benign rib tumors varied, with the main symptom in 36 patients (56.25 %) being palpable swelling. In the remaining group, 15 patients (23.44 %) presented with localized pain, 2 patients (3.12 %) with tenderness and 1 patient (1.56 %) with numbness of the left upper arm. Tumors were detected in ten patients (15.63 %) during routine chest radiograph taken for their recruitment in the army. In the group with malignant tumors, 18 patients (66.67 %) presented with localized pain, 6 (22.22 %) with palpable swelling, one (3.7 %) with pathologic fracture after exercise and in two

Materials and methods

Table 1 Pathologic features of rib tumors

The medical records of all patients who underwent surgery with rib lesions in our department during the period 1998–2012 were studied retrospectively. The diagnosis of rib lesion was established through clinical and radiologic studies. Patients with lung, pleural, breast and skin carcinomas invading the chest wall were excluded from this study. Patients with known rib fracture and radiologic findings of callus formation were also excluded from the study. Finally, ninety-one (91) patients were included in this study and analyzed. Diagnostic workup consisted of a thorough medical history and physical examination. Plain chest radiographs as well as CT of the chest were obtained for all patients. MRI and radionuclide bone scan were conducted in 12 and 35 patients, respectively, to rule out bone metastasis and distinguish whether the lesion was primary rib tumor or not. Positron emission tomography (PET/CT) was routinely conducted in four patients, preoperatively in staging due to prior malignant tumor elsewhere and in one patient whose lesion was reported to be potentially malignant after the operation. Fine-needle aspiration biopsy was performed on 5 patients who had three or more rib lesions and infiltration sites through the lung parenchyma, to distinguish the origin of the tumor.

Results 91 patients were included in the study: 81 male patients (89.01 %) and 10 female patients (10.99 %), ranging in age

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Pathologic tumor type

n = 91 (%)

Benign rib tumors

64 (70.33 %)

Posttraumatic fibro-osseous lesion/dysplasia

20

Osteochondroma

16

Enchondroma

11

Fibrous dysplasia

5

Langerhans cell histiocytosis/eosinophilic granuloma (EG)

3

Sclerosing (ossifying) xanthoma

2

Cavernous hemangioma

2

Osteoblastoma

2

Neurilemmoma (schwannoma)

1

Aneurysmal bone cyst

1

Intraosseous lipoma Malignant rib tumors Primary malignant rib tumors

1 27 (29.67 %) 14 (15.38 %)

Chondrosarcoma

6

Osteosarcoma

3

Solitary plasmacytoma

2

Ewing’s sarcoma

2

Primitive neuroectodermal tumor/Askin tumor Metastatic rib tumors

1 13 (14.29 %)

Renal cell carcinoma

5

Breast carcinoma

2

Colon adenocarcinoma

2

Esophageal adenocarcinoma

1

Gastric adenocarcinoma

1

Melanoma

1

Lung adenosquamous carcinoma

1

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all patients with malignant rib tumors and in 49 patients (76.56 %) with benign lesions. The rest of the patients with benign lesion had left the army service in the meantime and contact with them became impossible. The survival rate in the group of patients with benign rib tumors was 100 %, with no recurrence and follow-up of 13–177 months. The overall survival rate in the patient group with malignant rib tumors was 55.56 %, with 15 patients dying during followup from generalized carcinomatosis. The survival curve of primary malignant rib tumors is depicted in Fig. 1. The numbers of patients, the outcome and the mean survival or follow-up according to the outcome of each histological feature of metastatic malignant rib tumor are recorded in Table 3.

patients (7.41 %) the lesions were detected during routine chest radiograph (Table 2). CT scan was performed in all patients found with rib tumor. MRI was conducted in 12 patients and radionuclide bone scan in 35. 26 patients had positive radionuclide bone scan. Preoperative biopsy with fine-needle aspiration was performed in five patients revealing metastatic rib tumor in three patients and primary malignant tumor in two. All patients underwent operation, with different surgical policy in each group of benign, primary malignant and metastatic rib lesion. Intraoperative frozen section analysis determined the pathology of the rib lesion and designated the surgical policy. In metastatic rib lesions, the affected ribs were removed with free margins, including the adjacent muscles and the pleura. If the lesion was benign only the affected ribs were removed with free margins. If primary malignancy was proven, we proceeded to a wide resection including the affected ribs with at least a 4–5 cm free margin proximal and distal to the tumor and also resection of portions of the ribs immediately above and below the tumor, the adjacent muscles and the pleura. Chest wall closure was achieved without difficulty in all the patients. Reconstruction to cover large defects, when the resection consisted of three or more ribs, was performed in 13 patients (14.28 %), 2 patients from the benign group cohort and 11 from the malignant group, with synthetic polypropylene mesh and local muscle flaps (latissimus dorsi muscles or pectoralis major muscles). The mortality rate was 0 %. The morbidity rate was 7.69 %, with 2 patients presenting postoperative atelectasis and 5 patients presenting seroma, which resolved with physiotherapy and antibiotics administration, respectively. All patients with malignant rib tumors were referred to an oncologist for chemotherapy and/or radiotherapy. None of our patients underwent salvage surgery after the final pathology report, even though in 5 patients (5.49 %) the distance of the tumor from the free margins was found to be less than expected, but did not alter the policy of chemotherapy and/or radiotherapy. Follow-up was obtained in Table 2 Clinical presentation of rib tumors

Discussion Rib tumors are rare, with an incidence of less than 1 % in the population [1, 2]. Malignancy among them is significantly more common than benign lesions. Approximately 60 % are malignant, although Hsu et al. [1–3, 5, 8] reported a ratio of benign to malignant primary chest wall tumors to be 1:1 in their study. However, among the patients evaluated in our study, those with benign tumors were more common. Reviewing the literature, it is reported that rib tumors occur with equal gender distribution [8]. In our study there was a significant male predominance with a male to female ratio 8:1 (81 males and 10 females). However, it has to be taken into consideration that our hospital was initially designed to provide health care mainly to military personnel of our country and their families, mostly composed of male personnel. Fibrous dysplasia, chondrosarcoma and enchondroma are the most common benign lesions, with Langerhans cell histiocytosis or eosinophilic granuloma, aneurysmal bone cyst, osteoblastoma, intraosseous lipomas and rib hemangiomas considered as rare benign rib tumors (Fig. 2) [1, 2, 4, 5].

Benign (n = 64) (%)

Malignant (n = 27) (%)

Total (n = 91) (%)

Palpable

36 (56.25)

6 (22.22)

42 (46.15)

Pain

15 (23.44)

18 (66.67)

33 (36.26)

Tenderness

2 (3.12)

0

2 (2.2)

Numbness of the upper arm

1 (1.56)

0

1 (1.1)

Pathologic fracture

0

1 (3.7)

1 (1.1)

Asymptomatic

10 (15.63)

2 (7.41)

12 (13.19)

Right/left side

30/34 (46.87/53.13)

15/12 (55.56/44.44)

45/46 (49.45/50.55)

Solitary/multiple

53/11 (82.81/17.19)

11/16 (40.74/59.26)

64/27 (70.33/29.67)

Symptoms

Clinical features

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Gen Thorac Cardiovasc Surg Fig. 1 Survival curve for primary malignant rib tumors following surgery

Table 3 Outcome of metastatic rib tumors and mean value of survival or follow-up according to the outcome Type of malignancy

n

Outcome

Mean value of survival/ follow-up

Renal cell carcinoma

5

Dead

32 (19–47)

Breast carcinoma

2

Dead

47 (15–79)

Colon adenocarcinoma

2

Alive

19.5 (3–36)

Esophageal adenocarcinoma

1

Dead

9

Gastric adenocarcinoma

1

Dead

23

Melanoma

1

Alive

9

Lung adenosquamous carcinoma

1

Alive

8

Chondrosarcoma is the most prominent malignant primary tumor of the chest wall in adults, followed by osteosarcoma. Ewing’s sarcoma is a relatively rare malignant bone neoplasm that typically occurs in children and young adults (Fig. 3a) [1, 2, 5]. Solitary and isolated rib metastases are rare with the most common sites of primary origin being renal, breast, lung and gastrointestinal cancer (Fig. 3b) [9]. The clinical presentation of rib tumors vary. Asymptomatic patients diagnosed incidentally after routine examination as part of a screening or investigation of an unrelated condition are generally few [1, 2]. Mostly, patients present with palpable enlarging mass. Pain is the second most common symptom for which patients complain. Almost two-thirds of benign tumors present with pain, and nearly all malignant tumors will eventually cause pain, which can be a result of periosteal or neural invasion.

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Pain is often vague and diffuse, affecting a particular region of the chest, and it is often attributed to a musculoskeletal cause, such as arthritis or recent trauma [1]. Neurological signs and symptoms such as brachial plexus neuropathy and muscle weakness are also reported. Other rare reported symptoms are spinal cord compression and dyspnea due to pleural effusion [1, 8, 10, 11]. The diagnosis of a rib tumor includes a careful history and physical examination followed by a plain chest radiographs and a CT scan. A history of trauma to that specific area may suggest that the lesion is benign and probably a posttraumatic fibro-osseous lesion. 20 patients from our study were found to have posttraumatic fibroosseous lesion, but none could recollect a history of trauma in the specific area. Nevertheless, some patients may attribute a painful mass to a recent injury or pain due to rib fracture when it is in fact a malignant lesion. A history of prior malignancy may suggest an etiology of rib tumor. Physical examination might or might not reveal a mass or tenderness and the examination should include inspection for enlarged lymph nodes. Systemic symptoms of fever, malaise, fatigue and weight loss are suggestive of infection or malignant tumor [1, 2, 8, 10]. Rapid increase of tumor size, involvement of surrounding tissues and cortical destruction, although not pathognomonic, suggest malignancy [2]. Plain chest radiographs (CR) are the first imaging studies used in the diagnosis of rib tumors. Additionally, some tumors are incidentally found on CR. Although inadequate as the sole imaging modality in the current era of advanced imaging techniques, CR can still provide useful information about the tumor: the size, location,

Gen Thorac Cardiovasc Surg Fig. 2 Benign rib tumors. a CT 3D-reconstruction of patient with osteochondroma of the 7th left rib. b. Coronal view of the 3rd left rib osteoblastoma in T1 signal MRI. c CT of patient with intraosseous lipoma of the 7th left rib. d CT of a hemangioma in the 5th left rib

Fig. 3 Malignant rib tumors. a CT of primitive neuroectodermal tumor arising from the 5th right rib. b Radionuclide bone scintigraphy of solitary rib metastasis of the 5th right rib from renal cell carcinoma

calcification, ossification, bony involvement, presence of enlarged mediastinal lymph nodes and, if any, of large pulmonary metastases can all be ascertained [2, 12, 13]. CT is far more sensitive than CR in visualizing calcification and cortical destruction. It provides valuable information on the size and location of rib tumor; the extent of the tumor; rib involvement; the presence of infiltration to

pleura, lung or soft tissues; and the presence or not of pulmonary metastases. Tumor vascularity can also be ascertained when intravenous contrast is used, providing some typical features that can be linked to certain types of neoplasms, such as the presence of phleboliths and vascular enhancement in hemangioma [1, 2, 4, 8, 12–15]. CT is useful for operative planning, monitoring response to

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chemotherapy or radiation and follow-up evaluation of patients with chest wall tumors [1, 2, 8]. However, imaging features of chest wall tumors are nonspecific. Soft tissue mass, bone destruction, calcification and bone deformity are frequently described, but are not diagnostic of malignancy [9, 14]. MRI in some cases may be the preferred method to image rib tumors. It provides detailed information on soft tissue involvement and possible extension to the spine or thoracic inlet, on infiltration of brachial plexus, subclavian artery and subclavian vein [1, 2]. There are reports that indicate the superiority of MRI in the detection of rib metastases in patients with lung cancer compared with CT and radionuclide scan [16–18]. Radionucleotide bone scan is recommended to evaluate for metastases and rule out another primary bony tumor [1, 2]. PET with F-18-fluoro-2-deoxy-D-glucose is another imaging modality that can be used to evaluate patients with rib tumors, especially in staging [19–21]. Specific data for rib tumors are limited; however, application of PET or PET combined with CT (PET/CT) to characterize and stage other tumors is promising. With regard to sarcomas, PET has been shown to be more sensitive in detecting primary tumors (100 %), lymph node involvement (95 %) and bony manifestations (89 %) compared with conventional imaging techniques, including CT and MRI [1, 19, 20]. Until now, compared with CT, PET has: improved ability to distinguish benign from malignant rib tumors and to detect metastases, higher accuracy in identifying local recurrence and greater accuracy in evaluating response to treatment for sarcomas [1, 19–22]. The need of preoperative biopsy remains controversial. Fine-needle aspiration or non-excisional biopsy of the rib tumor should be performed cautiously. Some authors consider that fine-needle biopsy must be discouraged since diagnosis by cytologic specimen is sometimes unsatisfactory. Incorrect diagnosis due to inadequate tissue specimen may even lead to less than optimum treatment and worse prognosis. The risk of implantation of tumor tissue along the needle track after aspiration biopsy is noteworthy and makes it an unpopular preoperative diagnostic tool [1, 8, 23]. Excisional biopsy is the preferred method for diagnosing small tumors, with margins of at least 1–2 cm needed, whereas those with high suspicion of malignancy should receive wide radical resection or subsequent resection for safe margin [1, 8, 23]. Intraoperative frozen sections analysis of the tumor and the margins, wherever possible, will define the extent of excision. Non-excision biopsy should be reserved for those with suspicion of metastatic or hematologic disease, in whom aggressive surgical resection is less beneficial [8]. Rib tumors should be considered malignant until proven otherwise, due to difficulty in differentiating benign from

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malignant tumor preoperatively. Radical en bloc surgical excision with immediate reconstruction is the key to success in the management of malignant primary rib tumors. Generally with malignant rib tumors, to prevent recurrence, the resection should include a 4–5 cm margin of normal tissue on all sides, which includes resection of portions of the ribs immediately above and below the tumor, the adjacent muscles and the pleura. If there is infiltration of the lung parenchyma or diaphragm, one should not hesitate to excise the involved portions [1, 2, 5, 23]. For benign or metastatic rib tumors, surgery must consist of wide resection of the involved ribs with 2–3 cm free margins to be considered as sufficient. When we treated patients with metastatic rib tumors, we followed the criteria for treating metastases and in particular: (1) the primary tumor was controlled, (2) no extrathoracic lesions were present, (3) the metastases were technically resectable and (4) the general and functional risks were tolerable. All patients with malignant rib tumors must consult an oncologist to define the appropriate postoperative chemotherapy or/and radiotherapy [5]. The size of the tumor and the extent of the resection should not be limited by the size of the resulting defect. For small defects, less than 5 cm in diameter or those located posteriorly under the scapula above the fourth rib, the skeletal component can be ignored and the defect closed with only soft tissue [1, 23, 24]. The goals of chest wall reconstruction are twofold. Restoration of functional and structural integrity is the main priority; however, the ultimate esthetics of the reconstruction is an integral component. Other objectives of chest wall reconstruction are: to avoid a lung hernia from the chest wall defect; to counteract substantial shrinking of the operated side of the thorax, leading to a thoracoplasty-like effect; to prevent impaction of the scapula (intrathoracic scapula) in case of posterior chest wall resections, especially when the resection is extended down to the 5th and 6th ribs; and to protect the underlying mediastinal organs against external impact [1, 24]. Loss of more than 4 consecutive ribs or defects of the lateral chest wall larger than 5 cm necessitates skeletal stabilization to avert a flail chest deformity. The existing options include either autogenous or alloplastic reconstruction. Many different types of flaps, either pedicled or free, have been described for autogenous reconstruction of the chest wall deficiency. Pedicled muscle and myocutaneous options include the pectoralis major, latissimus dorsi, abdominal rectus, external oblique and trapezius flaps. Omentum is another option that may be used. If appropriate local options have been compromised by prior surgery, if the defect is particularly large, or if there is extensive local injury from prior radiotherapy, free tissue transfer may be required [1, 5, 23, 24]. The uses of alloplastic materials in chest wall reconstruction are based on convenience, reliability and ease of

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application of the synthetic materials. Alloplastic materials include non-absorbable synthetic woven meshes: polypropylene, polyester and polytetrafluoroethylene soft tissue patches or, more recently, knitted meshes. To strengthen the rigidity of reconstruction, methyl methacrylate is usually used as sandwich between two layers of mesh or in neo-ribs technique. Biologic meshes, as those based on bovine pericardium, have the same tensile strength and elasticity as those of synthetic meshes and proper physiologic properties, which make them more resistant to infection and contamination [1, 23, 24]. The overall 5-year survival after resection of primary chest wall neoplasms is approximately 60 %. Recurrence can occur in up to 50 % of patients, with a resultant 5-year survival of 17 % [1, 25]. The prognosis of benign tumors after surgical excision is uniformly excellent. For malignant neoplasms, the adequacy of surgical excision is an important determinant of the incidence of recurrence and survival [23, 25].

Conclusion Rib tumors are rare and should be considered malignant until proven otherwise. After all the diagnostic workup, which must include plain chest radiographs, CT of the chest and in selected cases MRI, radionuclide bone scan and PET/CT scan, surgery must be offered to patients. Although some rib lesions have radiologic findings suggestive for a diagnosis, frozen sections of the margins and the tumor is the most appropriate method to differentiate benign from malignant tumors and to state a different surgical approach. Surgery consists of wide resection of the tumor with free margins, in order to provide the best chance for cure in both benign and malignant lesions. Conflict of interest interest exists.

The authors have declared that no conflict of

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Rib tumors: a 15-year experience.

A retrospective study of rib tumors was conducted to review their clinical, radiological, and pathological features, the difficulties in differentiati...
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