Aneurysmal Bone Cyst of the Sternum Youichi Ishinada, M.D., Hiroo Yabe, M.D., Eiji Ogoshi, M.D., Kuni Nishikawa, M.D., and Hisami Iri, M.D. gram showed an osteolytic disease destroying and markedly expanding the body of the sternum with a paper-thin shell of new bone formation both anteriorly and posteriorly. The lowest part of the body appeared to be normal (Fig lA, 1B). Selective internal mammary angiography revealed some radiopacity corresponding to the lesion (Fig 1C). Contrast medium injected directly into the lesion colPrimary tumors of the sternum are rare, and lected in the cystic cavity, clearly delineating most are malignant [lo, 14, 151. Benign tumors the extent of involvement. Draining veins were of the sternum are extremely rare. A few cases also demonstrated (Fig 2). Biopsy of the lesion revealed an extremely have been reported as chondroma, giant cell tumor [8, 181, eosinophilic granuloma [13, 17, vascular tumor with a very thin shell of bone. 191, and chondromyxoid fibroma [l, 191. Only Profuse bleeding from the cystic cavity was entwo cases of aneurysmal bone cyst in the ster- countered. The specimen was diagnosed as an num have been described. This report docu- aneurysmal bone cyst. At operation on October 12, 1976, the tumor ments another such cyst. was exposed through a T-shaped skin incision A 26-year-old housewife was seen on August and the pectoralis major muscles were dissected 11, 1976, because of pains and swelling in the over the respective sternocostal region. The sternal region. For approximately three years internal mammary arteries were ligated in the she had experienced slight pain when elevating second and fifth interspaces, and the intercostal her arm, coughing, or sneezing. In May, 1976, arteries were also ligated and severed at the latthe pain was aggravated after she was struck on eral border of the sternum on both sides. The the anterior chest wall by the head of her child. tumor was thus isolated from the major blood At that time the swelling, which had been supply. The second to the sixth costal cartilages minimal, increased markedly but the overlying were excised bilaterally at the sternal border, and the retrosternal wall was separated from the skin remained normal in color. Physical examination revealed a tender and tumor carefully so as not to break the pleura or nonpulsatile mass approximately 9 by 5 cm over pericardium. At the level of the sixth costosterthe body of the sternum. On palpation, the nal joint, the body of the sternum was cut mass appeared to be covered only by a thin transversely where normal bone tissue was found. The manubriosternal joint was disshell of bone. Laboratory studies were all within normal sected, and the whole tumor was removed. The limits, including serum alkaline phosphatase. defect thus created, measuring 10.5 by 5 cm, Roentgenograms of the sternum and a tomo- was reconstructed with 11 cm of autogenous fibula divided lengthwise. The graft spanning From the Departments of Orthopaedic Surgery, Cardiovas- the manubrium and the rest of the body was cular Surgery, and Clinical Pathology, School of Medicine, fixed with wire sutures and was fixed to the Keio University, Tokyo, Japan. stumps of the second to fourth costal cartilage Accepted for publication Mar 31, 1978. with nylon sutures (Fig 3). The retrosternal Address reprint requests to Dr. Ishinada, Department of space was drained by continuous suction tubes. Orthopaedic Surgery, School of Medicine, Keio University, The pectoralis major muscles were sutured to35-Shinanomachi, Shinjuku-ku, 160 Tokyo, Japan. ABSTRACT A very rare case of aneurysmal bone cyst of the sternum is described. Aneurysmal bone cyst is a tumorlike bone lesion occumng commonly in the metaphysis of long bones and in the vertebrae. This report is one of the very few cases in the literature of aneurysmal bone cyst of the sternum. Total resection of the tumor and reconstruction of the sternal defect using autogenous fibula are presented.
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0003-4975/79/030254-06$01.25 @ 1978 by Youichi Ishinada
255
A
Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
B
Fig I . ( A )Lateral roentgenogram of the sternum showing an osteolytic disease of the body with a paper-thin shell of bone. ( B ) Lateral tomogram of the sternum showing a thin bony shell on most of the body anteroposteriorly while the lowest portion remains normal (arrow). (Retouched for clarity.) (C) Lateral projection of a selective internal mammary angiogram revealing diffuse retention of opaque medium in the involved area and slight hypervascularity .
gether in the midline, and the wound was closed in layers. The patient lost about 1,000 ml of blood during the operation. After operation, no paradoxical movement of the chest wall was noted. Suction tubes were removed seventy-two hours after the operation and yielded about 300 ml. The patient’s recovery was uneventful. She was discharged wearing a brace on the fifty-seventh postoperative
C
day. When last seen, twelve months after operation, she was well. A roentgenogram made at that time is shown in Figure 4. The resected specimen, shaped somewhat like the back of a turtle, measured about 9 by 5 by 3 cm and was covered by a very thin shell of bone, which was easily scissored. The cross section of the tumor had a single large cystic cavity in which the bone marrow was replaced by reddish-brown, soft, friable tissue. Microscopically, the tumor consisted of many large blood spaces separated by fibrous trabeculae containing multinucleated giant cells. Newly formed fine bones rimmed by osteoblasts and a few osteoclasts were found at the periphery of the tumor adjacent to the periosteum. These findings were typical of an aneurysmal bone cyst (Fig 5).
256 The Annals of Thoracic Surgery Vol 27 No 3 March 1979
A B Fig 2 . (A) Lateral and (B)anteroposterior views of the bone cyst of the sternum showing the extent of involvement and draining veins (arrows).
1 M
3rd
Fig 3 . The reconstruction. The anterior view is on the
left and the lateral view on the right. (M = manubrium; 2nd and 3rd = costal cartilage; BF = bivalved 'fibula; RB = rest of the body; S = step formation.)
'
RB
257 Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
Fig 4. Lateral roentgenogram made twelve months postoperatively shows bony union between the graft and the sternum both superiorly and inferiorly.
Comment One hundred seven instances of aneurysmal bone cyst were recorded in the Bone Tumor Registry [ l l ] of the Japanese Orthopaedic Association during the period 1964 through 1975. The lesion has been found in various parts of the skeleton but is most common in the metaphysis of long bones and in the vertebrae (Table 1). During the same period, forty-one instances of primary bone tumors of the sternum were reported, but none were aneurysmal bone cysts (Table 2). Only two cases of aneurysmal bone cyst in the sternum have been reported previously in the literature [5, 61. One was not described in
B
Fig 5. (A) Photomicrograph demonstrating typical histological changes of aneurysmal bone cyst. Large blood spaces are separated by fibrous trabeculae containing multinucleated giant cells. (B)Photomicrograph of another part of the tumor showing newly formed bones rimmed by osteoblasts. Multinucleated giant cells are also visible. (HDE; original magnification: A X100, B X200.)
258 The Annals of Thoracic Surgery Vol 27 No 3 March 1979
Table I. Aneurysmal Bone Cyst”
Table 2 . Primary Tumors of the Sternum” No. of cysts
Site Femur Tibia Vertebra Cervical Thoracic Lumbar Sacrum Humerus Fibula Rib Patella Clavicle Ilium Pubis Hand Foot Radius Ischium Mandible
28 20 19 5 4 6 4 10 7 4 4 3 3 2 2 2 1 1 1
Total
107 ~
~
aFrom the Bone Tumor Registry in Japan (1964-1975).
No. of Tumors
Cartilaginous Cartilaginous exostosis Enchondroma Chondrosarcoma Osseous Osteoma Osteoid osteoma Benign osteoblastoma Osteosarcoma Fibrous Fibrous dysplasia Uncertain origin Giant cell tumor Marrow origin Letter-Swie Myeloma Hodgkin Vascular Hemangioma Other Bone cyst Unclassified Total
7 9 4
1 3 1 3 2 1 1 2 41
aFrom the Bone Tumor Registry in Japan (1964-1975).
detail [61. I n t he other, t he cyst developed in the manubrium of the sternum of a 46-year-old m a n a n d w a s removed totally. Curettage and bone graft 191 is the common choice for t h e treatment of aneurysmal bone cyst. Occasionally more radical approaches o r irradiation 1121 ma y be needed. I n our patient, however, because of the large size of the tumor, t h e thin bony shell of the cyst anteroposteriorly, a n d the possible bleeding during curettage, en bloc resection of the lesion and reconstruction of the chest wall were considered necessary. To minimize bleeding during the procedure, major blood vessels were ligated before dissection. Several methods were described in the literature for t h e repair a n d reconstruction of sternal a n d chest wall defects [2, 71. Iliac [4, 161, tibia1 [ 8 ] , o r costal [3] grafts have been employed b y several authors with satisfactory results. W e chose t h e autogenous fibula to reconstruct a firm, well-supported, and stable chest wall. The
transplanted bone consolidated with the host and apparently served its expected purpose twelve months after operation.
References 1. Alonso-Lej F, de Linera FA: Resection of the entire sternum and replacement with acrylic resin. J Thorac Cardiovasc Surg 62:271, 1971 2. Baue AE: Total resection of the sternum. J Thorac Cardiovasc Surg 45:559, 1963 3. Bisgard JD, Swenson SA Jr: Tumors of the sternum: report of a case with special operative technic. Arch Surg 56:570, 1948 4. Brodin H, Linden K: Resection of the whole of the sternum and the cartilaginous parts of costae 1-4: a case report. Acta Chir Scand 118:13, 1959 5. Dabska M, Buraczewski J: Aneurysmal bone cyst: pathology, clinical course and radiologic appearances. Cancer 23:371, 1969 6. Dahlin DC, Besse BE Jr, Pugh DG, et al: Aneurysmal bone cysts. Radiology 64:56, 1955 7. Ishikawa S, Itaya H, Shiratsu F, et al: The surgical treatment for primary tumors of the sternum. Jpn J Thorac Surg 2 7 9 3 , 1974
259 Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
8. Kinsella TJ, White SM, Koucky RW: Two unusual tumors of the sternum. J Thorac Surg 16:640,1947 9. Koskinen EVS, Visuri TI, Holstrom T, et al: Aneurysmal bone cyst-evaluation of resection and of curettage in 20 cases. Clin Orthop 118:136, 1976 10. Martini N, Huvos AG, Smith J, et al: Primary malignant tumors of the sternum. Surg Gynecol Obstet 138:391, 1974 11. National Cancer Center: Bone Tumor Registration in Japan. Tokyo, National Cancer Center, 1974, 1975 12. Nobler MP, Higinbotham NL, Phillips RF: The cure of aneurysmal bone cyst-irradiation superior to surgery in an analysis of 33 cases. Radiology 90:1185, 1968 13. Ochsner A Jr, Lucas GL, McFarlan GB Jr: Tumors of the thoracic skeleton: review of 134 cases. J Thorac Cardiovasc Surg 52:311, 1966
14. O’Neal LW, Ackerman LV: Cartilaginous tumors of ribs and sternum. J Thorac Surg 21:71,1951 15. Pascuzzi CA, Dahlin DC, Clagett OT: Primary tumors of the ribs and sternum. Surg Gynecol Obstet 104:390, 1957 16. Pradhan DJ, Musikabhumma S, Michelson E, et al: Total sternal resection and reconstruction using bone grafts and fascia lata. Am Surg 39:180, 1973 17. Sayama M, Okamoto J, Watabe T, et al: Eosinophilic granuloma of the sternum: a case report. Orthop Surg (in Japanese) 12:673, 1961 18. Takemitsu Y, Fukuma H, Kai T, et al: Resection and reconstruction operation of the sternum: a case report of giant cell tumor. J West Jpn Orthop Trauma 20:23, 1971 19. Teitelbaum SL: Twenty years’ experience with intrinsic tumors of the bony thorax at a large institution. J Thorac Cardiovasc Surg 63:776, 1972
254
0003-4975/79/030254-06$01.25 @ 1978 by Youichi Ishinada
255
A
Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
B
Fig I . ( A )Lateral roentgenogram of the sternum showing an osteolytic disease of the body with a paper-thin shell of bone. ( B ) Lateral tomogram of the sternum showing a thin bony shell on most of the body anteroposteriorly while the lowest portion remains normal (arrow). (Retouched for clarity.) (C) Lateral projection of a selective internal mammary angiogram revealing diffuse retention of opaque medium in the involved area and slight hypervascularity .
gether in the midline, and the wound was closed in layers. The patient lost about 1,000 ml of blood during the operation. After operation, no paradoxical movement of the chest wall was noted. Suction tubes were removed seventy-two hours after the operation and yielded about 300 ml. The patient’s recovery was uneventful. She was discharged wearing a brace on the fifty-seventh postoperative
C
day. When last seen, twelve months after operation, she was well. A roentgenogram made at that time is shown in Figure 4. The resected specimen, shaped somewhat like the back of a turtle, measured about 9 by 5 by 3 cm and was covered by a very thin shell of bone, which was easily scissored. The cross section of the tumor had a single large cystic cavity in which the bone marrow was replaced by reddish-brown, soft, friable tissue. Microscopically, the tumor consisted of many large blood spaces separated by fibrous trabeculae containing multinucleated giant cells. Newly formed fine bones rimmed by osteoblasts and a few osteoclasts were found at the periphery of the tumor adjacent to the periosteum. These findings were typical of an aneurysmal bone cyst (Fig 5).
256 The Annals of Thoracic Surgery Vol 27 No 3 March 1979
A B Fig 2 . (A) Lateral and (B)anteroposterior views of the bone cyst of the sternum showing the extent of involvement and draining veins (arrows).
1 M
3rd
Fig 3 . The reconstruction. The anterior view is on the
left and the lateral view on the right. (M = manubrium; 2nd and 3rd = costal cartilage; BF = bivalved 'fibula; RB = rest of the body; S = step formation.)
'
RB
257 Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
Fig 4. Lateral roentgenogram made twelve months postoperatively shows bony union between the graft and the sternum both superiorly and inferiorly.
Comment One hundred seven instances of aneurysmal bone cyst were recorded in the Bone Tumor Registry [ l l ] of the Japanese Orthopaedic Association during the period 1964 through 1975. The lesion has been found in various parts of the skeleton but is most common in the metaphysis of long bones and in the vertebrae (Table 1). During the same period, forty-one instances of primary bone tumors of the sternum were reported, but none were aneurysmal bone cysts (Table 2). Only two cases of aneurysmal bone cyst in the sternum have been reported previously in the literature [5, 61. One was not described in
B
Fig 5. (A) Photomicrograph demonstrating typical histological changes of aneurysmal bone cyst. Large blood spaces are separated by fibrous trabeculae containing multinucleated giant cells. (B)Photomicrograph of another part of the tumor showing newly formed bones rimmed by osteoblasts. Multinucleated giant cells are also visible. (HDE; original magnification: A X100, B X200.)
258 The Annals of Thoracic Surgery Vol 27 No 3 March 1979
Table I. Aneurysmal Bone Cyst”
Table 2 . Primary Tumors of the Sternum” No. of cysts
Site Femur Tibia Vertebra Cervical Thoracic Lumbar Sacrum Humerus Fibula Rib Patella Clavicle Ilium Pubis Hand Foot Radius Ischium Mandible
28 20 19 5 4 6 4 10 7 4 4 3 3 2 2 2 1 1 1
Total
107 ~
~
aFrom the Bone Tumor Registry in Japan (1964-1975).
No. of Tumors
Cartilaginous Cartilaginous exostosis Enchondroma Chondrosarcoma Osseous Osteoma Osteoid osteoma Benign osteoblastoma Osteosarcoma Fibrous Fibrous dysplasia Uncertain origin Giant cell tumor Marrow origin Letter-Swie Myeloma Hodgkin Vascular Hemangioma Other Bone cyst Unclassified Total
7 9 4
1 3 1 3 2 1 1 2 41
aFrom the Bone Tumor Registry in Japan (1964-1975).
detail [61. I n t he other, t he cyst developed in the manubrium of the sternum of a 46-year-old m a n a n d w a s removed totally. Curettage and bone graft 191 is the common choice for t h e treatment of aneurysmal bone cyst. Occasionally more radical approaches o r irradiation 1121 ma y be needed. I n our patient, however, because of the large size of the tumor, t h e thin bony shell of the cyst anteroposteriorly, a n d the possible bleeding during curettage, en bloc resection of the lesion and reconstruction of the chest wall were considered necessary. To minimize bleeding during the procedure, major blood vessels were ligated before dissection. Several methods were described in the literature for t h e repair a n d reconstruction of sternal a n d chest wall defects [2, 71. Iliac [4, 161, tibia1 [ 8 ] , o r costal [3] grafts have been employed b y several authors with satisfactory results. W e chose t h e autogenous fibula to reconstruct a firm, well-supported, and stable chest wall. The
transplanted bone consolidated with the host and apparently served its expected purpose twelve months after operation.
References 1. Alonso-Lej F, de Linera FA: Resection of the entire sternum and replacement with acrylic resin. J Thorac Cardiovasc Surg 62:271, 1971 2. Baue AE: Total resection of the sternum. J Thorac Cardiovasc Surg 45:559, 1963 3. Bisgard JD, Swenson SA Jr: Tumors of the sternum: report of a case with special operative technic. Arch Surg 56:570, 1948 4. Brodin H, Linden K: Resection of the whole of the sternum and the cartilaginous parts of costae 1-4: a case report. Acta Chir Scand 118:13, 1959 5. Dabska M, Buraczewski J: Aneurysmal bone cyst: pathology, clinical course and radiologic appearances. Cancer 23:371, 1969 6. Dahlin DC, Besse BE Jr, Pugh DG, et al: Aneurysmal bone cysts. Radiology 64:56, 1955 7. Ishikawa S, Itaya H, Shiratsu F, et al: The surgical treatment for primary tumors of the sternum. Jpn J Thorac Surg 2 7 9 3 , 1974
259 Case Report: Ishinada et al: Aneurysmal Bone Cyst of Sternum
8. Kinsella TJ, White SM, Koucky RW: Two unusual tumors of the sternum. J Thorac Surg 16:640,1947 9. Koskinen EVS, Visuri TI, Holstrom T, et al: Aneurysmal bone cyst-evaluation of resection and of curettage in 20 cases. Clin Orthop 118:136, 1976 10. Martini N, Huvos AG, Smith J, et al: Primary malignant tumors of the sternum. Surg Gynecol Obstet 138:391, 1974 11. National Cancer Center: Bone Tumor Registration in Japan. Tokyo, National Cancer Center, 1974, 1975 12. Nobler MP, Higinbotham NL, Phillips RF: The cure of aneurysmal bone cyst-irradiation superior to surgery in an analysis of 33 cases. Radiology 90:1185, 1968 13. Ochsner A Jr, Lucas GL, McFarlan GB Jr: Tumors of the thoracic skeleton: review of 134 cases. J Thorac Cardiovasc Surg 52:311, 1966
14. O’Neal LW, Ackerman LV: Cartilaginous tumors of ribs and sternum. J Thorac Surg 21:71,1951 15. Pascuzzi CA, Dahlin DC, Clagett OT: Primary tumors of the ribs and sternum. Surg Gynecol Obstet 104:390, 1957 16. Pradhan DJ, Musikabhumma S, Michelson E, et al: Total sternal resection and reconstruction using bone grafts and fascia lata. Am Surg 39:180, 1973 17. Sayama M, Okamoto J, Watabe T, et al: Eosinophilic granuloma of the sternum: a case report. Orthop Surg (in Japanese) 12:673, 1961 18. Takemitsu Y, Fukuma H, Kai T, et al: Resection and reconstruction operation of the sternum: a case report of giant cell tumor. J West Jpn Orthop Trauma 20:23, 1971 19. Teitelbaum SL: Twenty years’ experience with intrinsic tumors of the bony thorax at a large institution. J Thorac Cardiovasc Surg 63:776, 1972
