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Japanese Journal of Clinical Oncology 30:354-357 (2000)
© 2000 Foundation for Promotion of Cancer Research

Hemangioma of the Rib: a Case Report

Takehiro Okumura1, Hisao Asamura1, Haruhiko Kondo1, Yoshihiro Matsuno2 and Ryosuke Tsuchiya1

,+ 1Thoracic Surgery and 2Clinical Laboratory Divisions, National Cancer Center Hospital, Tokyo


    ABSTRACT
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
A case of hemangioma of the left seventh rib is presented. In January 1999, a 59-year-old woman presented with an enlarged costal mass which had been followed up for 4 years. ­Preoperative examination suggested chondrosarcoma because of tumor growth beyond the disrupted bony cortex. She underwent resection of the left seventh rib along with the sixth and seventh intercostal muscles and reconstruction of the chest wall defect. The pathological ­diagnosis of the lesion was hemangioma. She was discharged after an uneventful postoperative course. There has been no evidence of recurrence after a 14-month follow-up. Tumor growth beyond the disrupted bony cortex was a characteristic feature by both imagery and pathological examination in this case. This case represents a difficulty of a preoperative definite diagnosis of the chest wall tumors by imagery alone.


    INTRODUCTION
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
Primary chest wall tumors, including bony and soft tissue tumors, account for ~2% of all primary tumors. Malignant fibrous histiocytoma, chondrosarcoma and rhabdomyosarcoma are common primary malignant tumors. Among primary benign tumors, cartilaginous tumor, desmoid and fibrous dysplasia are common (1). Previous reports on chest wall tumors have demonstrated a high incidence of malignancy, which varies from 30 to 80% (2–6). Since chest wall tumors include various entities, their treatment depends on the condition of the lesion, such as histological type and location. Careful preoperative evaluation is indispensable for planning the treatment.

Hemangioma arising from a rib is a rare benign entity. In previous reports on rib hemangioma (7,8), the radiographic findings were compatible with benign characteristics. We report a case of hemangioma of the rib with radiographic findings which suggested malignant characteristics.


    CASE REPORT
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
A 59-year-old woman was referred to the National Cancer Center Hospital, Tokyo, because enlargement of an asymptomatic costal mass was detected on a chest radiograph in January 1999. Four years before this presentation, the mass had been incidentally found by a chest radiograph in a health survey and had been followed up as a fracture of the left seventh rib until this reference. The past histories of the patient and her family were unremarkable. All laboratory studies including serum tumor markers were normal. On physical examination, the lesion could be recognized as a dilatation of the left seventh rib. A chest radiograph taken on admission showed a mass shadow measuring 3.8 x 2.5 cm with extra­pleural signs in the left middle lung field. A rib radiograph showed a mass shadow expanding beyond the irregular cortex of the left seventh rib. The mass had some calcifications, which presented a so-called honeycomb appearance (Fig. 1). Chest computed tomography (CT) showed localized expansive growth of the tumor projecting toward the thoracic cavity and disrupted bony cortex without any lesions in the bilateral lung fields or mediastinum (Fig. 2). Magnetic resonance imaging of this lesion revealed a low signal on T1-weighted images, a slightly high signal on T2-weighted images and enhancement by contrast media (Fig. 3). A bone scan did not detect any other lesions. Low-grade malignant tumor of the rib, such as chon­drosarcoma, was suspected as a preoperative diagnosis because of its growth beyond the bony cortex and relatively long-term clinical course.



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Figure 1. Rib radiography. A mass (arrow) is located in the left seventh rib and expands beyond the irregular cortex. Calcification inside the mass presents a so-called honeycomb appearance.

 


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Figure 2. Chest CT shows localized expansive growth of the tumor (arrow) projecting toward the thoracic cavity.

 


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Figure 3. Magnetic resonance imaging of this lesion shows a low signal on T1-weighted images (a), a slightly high signal on T2-weighted images (b) and enhancement by contrast media (c).

 
She underwent resection of the left seventh rib along with the sixth and seventh intercostal muscles. Reconstruction of the chest wall defect was performed with a Gore-Tex® (polytetrafluoroethylene) patch.

Macroscopically, the tumor measuring 3.5 x 2.5 x 1.5 cm had a firm surface and bulged from the inner part of the seventh rib toward the thoracic cavity. It was covered with smooth pleura. The cut surface of the tumor had a red and multilocular appearance. Disrupted bony cortex was found inside the tumor (Fig. 4). Microscopically, the tumor was composed of a homogeneous conglomerate of thin-walled blood vessels with dilating channels. Most of the channels contained red blood cells in the cavity. A single layer of flat endothelial cells, which did not show significant atypia or mitosis, lined these channels (Fig. 5). There was no necrosis in the tumor. The pathological diagnosis of this lesion was hemangioma of the bone. The bony cortex inside the tumor was disrupted partially with reactive bone formation and was not accompanied by periosteum. On the other hand, there was periosteum between the tumor surface and the pleura, which confirmed expansive growth of the tumor as shown by radiography. No tumor tissue was detected at the surgical margin. She was discharged after an uneventful postoperative course. There has been no evidence of recurrence after a 14-month follow-up.




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Figure 4. Macroscopic appearance of the resected specimen and the cut surface of the tumor. (a) The tumor is covered by smooth pleura. (b) There is disrupted bony cortex (arrow) inside the tumor.

 



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Figure 5. Microscopic findings of the tumor. (a) The boundary (arrow) between the tumor (left side) and the surrounding normal tissue (hematoxylin–eosin, original magnification x40). (b) The tumor is composed of thin-walled blood vessels with dilating channels. A single layer of flat endothelial cells line these channels, which contained red blood cells (hematoxylin–eosin, original magnification x100).

 

    DISCUSSION
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
Hemangioma of the rib is very rare as both bone hemangioma and primary rib tumor. Hemangioma is defined as a neoplastic entity which arises from blood vessels. Although skin is by far the most common primary site of hemangioma, any organ including the bone can be affected. Hemangioma of the bone accounts for ~1% of all bone tumors in some series in the literature (9). While most of these cases occur in the skull or vertebral bodies, some have been reported to originate from the rib (7,8). On the other hand, primary tumors of the rib are uncommon, accounting for only 6–10% of primary bone tumors and approximately half of them are malignant (2,3,10). Ala-Kulju et al. reported that cartilaginous tumors were the most common of his 34 cases of primary rib tumor (6). According to the Japanese Bone Tumor Registration Database, from 1972 to 1994 there were only eight cases of hemangioma of the rib, accounting for 0.018% of all bone tumors (11).

Radiographically, bony hemangioma usually presents as lucent, well-demarcated defects. It expands and thins the cortex in flat bones, but complete cortical disruption is not present (12). Most cases of bone hemangioma can be diagnosed by characteristic radiographic findings which reflect the formation of reactive spicula produced by the lesion. These findings are generally called ‘sunburst appearance’ for skull hemangioma, ‘corduroy-like appearance’ for vertebra hemangioma and ‘honeycomb appearance’ for rib hemangioma (12). Although the present case presented a ‘honeycomb appearance’ on rib radiography, the tumor showed expansive growth beyond the disrupted bony cortex on the rib radiograph and chest CT. These findings suggested malignant characteristics. To our knowledge, there has been no previous report of rib hemangioma with such radiological findings.

A definite histological diagnosis is essential for treating chest wall tumors. There are three types of biopsy methods for chest wall tumors: needle aspiration biopsy, excisional biopsy and incisional biopsy. Ayala and Zornosa demonstrated that the diagnostic rate of needle biopsy for primary bone tumors was 83% in malignant tumors and 64% in benign tumors (13). However, in general, needle biopsy is not recommended unless there is a strong suspicion of myeloma or metastatic disease (14). Excisional biopsy can be generally applied in lesions less than 2 cm in diameter. Open biopsy has some problems that may interfere with a definitive therapy. For example, infection of the biopsy wound may delay initiating the appropriate therapy and the initial resection may interfere with identification of the area which should be removed in a later radical resection (15). Clinically, our preoperative diagnosis of the present case was low-grade primary malignant tumor of the chest wall. We did not perform any type of preoperative biopsy considering the indications and problems mentioned above. Therefore, resection was performed for both a definitive diagnosis and treatment. This case represents a difficulty of a preoperative definite diagnosis of the chest wall tumors by imagery alone.


    Acknowledgment
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
This work was supported in part by a Grant-in-Aid for Cancer Research (11–19) from the Ministry of Health and Welfare, Japan.


    FOOTNOTES
 
+ For reprints and all correspondence: Hisao Asamura, Thoracic Surgery Division, National Cancer Center Hospital, 1–1, Tsukiji 5-chome, Chuo-ku, Tokyo 104-0045, Japan. E-mail: hasamura@ncc.go.jp Back


    REFERENCES
 TOP
 ABSTRACT
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 Acknowledgment
 REFERENCES
 
1 Pairolero PC. Chest wall tumors. In: Shields TW, LoCicero J 3rd, Ponn RB, editors. General Thoracic Surgery, 5th ed. Baltimore: Williams and Wilkins 2000;589–98.

2 Graeber GM, Snyder RJ, Fleming AW, Head HD, Lough FC, Parker JS, et al. Initial and long-term results in the management of primary chest wall neoplasms. Ann Thorac Surg 1982;34:664–73.[Abstract]

3 Sabanathan S, Salama FD, Morgan WE, Harvey JA. Primary chest tumors. Ann Thorac Surg 1985;39:4–15.[Abstract]

4 McAfee MK, Pairolero PC, Bergstrahl EJ, Piehler JM, Unni KK, McLeod RA, et al. Chondrosarcoma of the chest wall: factors affecting survival. Ann Thorac Surg 1985;40:535–40.[Abstract]

5 King RM, Pairolero PC, Trastek VF, Piehler JM, Payne WS, Bernatz PE. Primary chest wall tumors: factors affecting survival. Ann Thorac Surg 1986;41:597–601.[Abstract]

6 Ala-kulju K, Ketonen P, Järvinen A, Salo J, Luosto R. Primary tumors of the ribs. Scand J Thorac Cardiovasc Surg 1988;22:97–100.

7 Clements RD, Turnage RB, Tyndal EC. Hemangioma of the rib: a rare diagnosis. Am Surg 1998;64:1027–9.[ISI][Medline]

8 Filosso PL, Oliaro A, Ruffini E, Abbona GC, Casadio C, Sapino A, et al. Hemangioma of the rib: a case report. J Cardiovasc Surg 1995;36:97–8.[Medline]

9 Dorfman HD, Steiner GC, Jaffe HL. Vascular tumors of bone. Hum Pathol 1971;2:349–76.[ISI][Medline]

10 Schmidt FE, Trummer MJ. Primary tumor of ribs. Ann Thorac Surg 1972;13:251–7.[Medline]

11 Bone Tumor Registry in Japan. Musculoskeletal Tumor Committee of the Japanese Orthopaedic Association, Tokyo 1994.

12 Dorfman HD, Czerniak B. Vascular lesions. In: Bone Tumors. St. Louis: Mosby 1997;729–814.

13 Ayala AG, Zornosa J. Primary bone tumors: percutaneous needle biopsy. Radiologic–pathologic study of 222 biopsies. Radiology 1983;149:675–9.[Abstract/Free Full Text]

14 Stelzer P, Gay WA. Tumor of the chest wall. Surg Clin North Am 1980;60:779–91.[ISI][Medline]

15 Ryan MB, McMurtrey MJ, Roth JA. Current management of chest-wall tumors. Surg Clin North Am 1989;69:1061–80.[ISI][Medline]

Received April 7, 2000; accepted May 29, 2000.


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