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Historical giant near-circumferential osteochondroma of the proximal humerus
J Shoulder Elbow Surg (2010) 19, e12-e15
www.elsevier.com/locate/ymse
Historical giant near-circumferential osteochondroma of the proximal humerus M. Allagui, MD, K. Amara, MD*, I. Aloui, MD, M.F. Hamdi, MD, M. Koubaa, MD, A. Abid, MD Department of Orthopedic Surgery, CHU Fattouma Bourguiba Monastir, Monastir, Tunisia Exostosis is the most common benign tumor that represents a cartilage-capped osseous protrusion on the external surface of a bone. It is derived from cartilaginous tissue of the physis, which is separated from the periphery of the growth plate during augmentation. Osteochondroma commonly occurs in the metaphyseal region of the long bones. Osteochondroma is preferentially located at the distal metaphysis of the femur and the proximal metaphysis of the tibia, which together represent 40% of the cases9; the proximal metaphysis of the humerus represents the second site of predilection.5 The majority of osteochondromas are asymptomatic, but limitations of joint motion and local compression of neurovascular structures are described. We report a case of a giant osteochondroma of the proximal humerus with major limitation of adduction and rotational motion of the arm. Extensive resection was followed by humeral intramedullary nailing.
Case report An 8-year-old girl presented with a large swollen mass in the proximal humerus. She reported a 5-year history of an increasing swollen mass in the proximal metaphyseal region of the left humerus. In the year before consultation, this swollen mass had been growing more rapidly. Moreover, esthetics and severe limitation of shoulder range of motion prevented childhood activities. Notably, the patient did not report any history of trauma. Physical examination showed a large lobulated hard mass of the proximal region of the arm. The patient had spontaneous abduction at 80 , and limitations of internal rotation (0 ) and abduction (100 )
were noted (Figure 1). The skin was supple without any overlying lesions or pigmentation. The radial pulse was present and normal in character. The digits showing instant capillary refill. No neurologic deficit or venous stasis was observed. Radiographs showed a giant exophytic sessile osseous lesion involving the proximal portion of the humerus. The mass measured 17 cm at its greatest dimension and extended circumferentially (Figure 2). Computed tomography confirmed the presence of a large bone tumor with medullar and cortical continuity; the cartilage cap was thick (<1 cm) (Figure 3). The diagnosis was either an osteochondroma or a juxtacortical chondrosarcoma. A biopsy was performed, and histologic examination confirmed a benign osteochondroma (Figure 4). Approximately 3 weeks after the initial biopsy, the patient underwent complete excision of the mass. The giant osteochondroma was accessed through a lateral incision. The first step consisted of individualizing the vascular and nerve structures of the arm. There were no signs of compression or irritation of the vascular or nerve structures. Then, the deltoid and brachial muscles were transected in part to allow clear exposure to the base of the tumor. A reactive bursa formation was discovered and removed. The deltoid muscle and tendons of the rotator cuff presented signs of impingement and tendinitis. By use of an osteotome, the tumor was freed and extensive resection of the tumor was performed. After tumor removal, the humerus was treated with intramedullary nailing with 2 K-wires (Figure 5). The arm was immobilized with a thoraco-brachial sling. No surgical complication occurred. Passive and active motion was started 6 weeks later. At 6 months postoperatively, radiographs showed bone remodeling (Figure 6) and the patient had normal and complete range of motion of the shoulder (Figure 7).
Discussion *Reprint requests: Amara Karim, MD, Department of Orthopedic Surgery, CHU Fattouma Bourguiba Monastir, Rue 1 Juin, 5000 Monastir, Tunisia. E-mail address: [email protected] (K. Amara).
Osteochondromas are generally asymptomatic and only discovered when symptoms occur. Symptoms consist of
1058-2746/$ - see front matter Ó 2010 Journal of Shoulder and Elbow Surgery Board of Trustees. doi:10.1016/j.jse.2010.04.004
Giant osteochondroma of proximal humerus
e13
Figure 1
Figure 2
Clinical aspect of tumor.
Anteroposterior radiographs show a well-circumscribed ossifying lesion, originating from the proximal humerus.
limitation of joint motion, edema, or paresthesia due to neurovascular compression. Painful osteochondromas may be explained by the presence of bursitis that covers the cup of the tumor.1 Osteochondromas of the proximal humerus may become painful and may cause mechanical impairment of the
glenohumeral joint. Despite the fact that osteochondromas are usually asymptomatic, the osteochondroma’s initial size in this case could explain the symptoms: it was impinging with the rotator cuff structures, being underneath the deltoid. Pain could be also explained by mechanical irritation of the deltoid
e14
M. Allagui et al.
Figure 5 After resection, intramedullary nailing of the humerus was performed.
Figure 3 Axial computed tomography scan shows a nearcircumferential tumor with medullar and cortical continuity.
Figure 6
Figure 4 Microscopic appearance consists of cartilage cap covering trabecular bone (hematoxylin-eosin stain, original magnification 40).
and biceps muscles.7 A painful osteochondroma should give rise to a high suspicion for sarcomatous degeneration. This event is seen in approximately 1% to 2% of patients with solitary osteochondroma,5 but the rate of malignant degeneration increases to 5% to 25% with hereditary multiple osteochondromas.8,10 The initial image can be easily mistaken for a chondroma or chondrosarcoma.4,6 In our case, the findings of histologic examination were consistent with a benign osteochondroma. The lesion had a regular cartilage cap, and the chondrocytes did not have a disorganized appearance. In the literature, the method of surgical treatment consists of combined resection and reconstruction with fibular or tibial nonvascularized graft with safe margins.2,9 Grafting procedures could be considered in cases of extensive involvement of the diaphyseal cortical wall, which leads to weakness. The
Final follow-up shows bone restoration.
wide involvement of the cortical wall sometimes makes the grafting procedure necessary. Resection alone is advocated in cases of a stalked bony protuberance of modest size. Intramedullary nailing (‘‘elastic stable intramedullary nailing’’ or ‘‘embrochage centro- me´dullaire elastique stable’’) has shown superb results in the treatment of nonpathologic fractures of the long bones in childhood. This method is rapidly gaining popularity for the treatment of spontaneous or pathologic fractures.3 Our goal is to stabilize an eventual pathologic fracture and the biomechanically weakened humerus. Accordingly, the intramedullary nailing could be described as a safe method: it avoids a second incision, provides good stability, and maintains normal growth for the humerus without impairment of shoulder function.
Conclusion A rare case of giant osteochondroma of the proximal humerus is presented. The tumor was entirely resected and stabilized by intramedullary nailing. This method was not reported previously in the literature. In fact, it is a relatively simple procedure that provides good
Giant osteochondroma of proximal humerus
Figure 7
e15
Clinical aspect at final follow-up.
stabilization and avoids bone grafting. As a result, at 6 months postoperatively, the patient recovered without complications and with normal functional results.
Disclaimer The authors, their immediate families, and any research foundations with which they are affiliated have not received any financial payments or other benefits from any commercial entity related to the subject of this article and have no potential conflicts of interest related to this manuscript.
References 1. D’Ambrosia R, Ferguson AB Jr. The formation of osteochondroma by epiphyseal cartilage transplantation. Clin Orthop Relat Res 1968;61: 103-15.
2. Gebhart M, Vadoud Seyedi J, Lejeune F. Giant solitary osteochondroma of the proximal humerus treated by resection and fibular autograft reconstruction. Acta Orthop Belg 1991;57:447-51. 3. Huber RI, Keller HW, Huber PM, Rehm KE. Flexible intramedullary nailing as fracture treatment in children. J Pediatr Orthop 1996;16:602-5. 4. Lange RH, Lange TA, Rao BK. Correlative radiographic, scintigraphic, and histological evaluation of exostoses. J Bone Joint Surg Am 1984;66:1454-9. 5. Mavrogenis AF, Papagelopoulos PJ, Soucacos PN. Skeletal osteochondromas revisited. Orthopedics 2008 Oct;31(10). pii: orthosupersite.com/view.asp?rID=32071 6. Milgram JW. The origins of osteochondromas and enchondromas. A histopathologic study. Clin Orthop Relat Res 1983:264-84. 7. Ogawa K, Yoshida A, Ui M. Symptomatic osteochondroma of the clavicle. A report of two cases. J Bone Joint Surg Am 1999;81:404-8. 8. Ozaki T, Hillmann A, Blasius S, Link T, Winkelmann W. Multicentric malignant transformation of multiple exostoses. Skeletal Radiol 1998; 27:233-6. 9. Taminiau AH, Hooning van Duyvenbode JF, Slooff TJ. Reconstruction of the proximal humerus with tibial autografts after resection of osteochondroma in adolescents. Clin Orthop Relat Res 1985:173-8. 10. Willms R, Hartwig CH, Bohm P, Sell S. Malignant transformation of a multiple cartilaginous exostosisda case report. Int Orthop 1997;21: 133-6.
www.elsevier.com/locate/ymse
Historical giant near-circumferential osteochondroma of the proximal humerus M. Allagui, MD, K. Amara, MD*, I. Aloui, MD, M.F. Hamdi, MD, M. Koubaa, MD, A. Abid, MD Department of Orthopedic Surgery, CHU Fattouma Bourguiba Monastir, Monastir, Tunisia Exostosis is the most common benign tumor that represents a cartilage-capped osseous protrusion on the external surface of a bone. It is derived from cartilaginous tissue of the physis, which is separated from the periphery of the growth plate during augmentation. Osteochondroma commonly occurs in the metaphyseal region of the long bones. Osteochondroma is preferentially located at the distal metaphysis of the femur and the proximal metaphysis of the tibia, which together represent 40% of the cases9; the proximal metaphysis of the humerus represents the second site of predilection.5 The majority of osteochondromas are asymptomatic, but limitations of joint motion and local compression of neurovascular structures are described. We report a case of a giant osteochondroma of the proximal humerus with major limitation of adduction and rotational motion of the arm. Extensive resection was followed by humeral intramedullary nailing.
Case report An 8-year-old girl presented with a large swollen mass in the proximal humerus. She reported a 5-year history of an increasing swollen mass in the proximal metaphyseal region of the left humerus. In the year before consultation, this swollen mass had been growing more rapidly. Moreover, esthetics and severe limitation of shoulder range of motion prevented childhood activities. Notably, the patient did not report any history of trauma. Physical examination showed a large lobulated hard mass of the proximal region of the arm. The patient had spontaneous abduction at 80 , and limitations of internal rotation (0 ) and abduction (100 )
were noted (Figure 1). The skin was supple without any overlying lesions or pigmentation. The radial pulse was present and normal in character. The digits showing instant capillary refill. No neurologic deficit or venous stasis was observed. Radiographs showed a giant exophytic sessile osseous lesion involving the proximal portion of the humerus. The mass measured 17 cm at its greatest dimension and extended circumferentially (Figure 2). Computed tomography confirmed the presence of a large bone tumor with medullar and cortical continuity; the cartilage cap was thick (<1 cm) (Figure 3). The diagnosis was either an osteochondroma or a juxtacortical chondrosarcoma. A biopsy was performed, and histologic examination confirmed a benign osteochondroma (Figure 4). Approximately 3 weeks after the initial biopsy, the patient underwent complete excision of the mass. The giant osteochondroma was accessed through a lateral incision. The first step consisted of individualizing the vascular and nerve structures of the arm. There were no signs of compression or irritation of the vascular or nerve structures. Then, the deltoid and brachial muscles were transected in part to allow clear exposure to the base of the tumor. A reactive bursa formation was discovered and removed. The deltoid muscle and tendons of the rotator cuff presented signs of impingement and tendinitis. By use of an osteotome, the tumor was freed and extensive resection of the tumor was performed. After tumor removal, the humerus was treated with intramedullary nailing with 2 K-wires (Figure 5). The arm was immobilized with a thoraco-brachial sling. No surgical complication occurred. Passive and active motion was started 6 weeks later. At 6 months postoperatively, radiographs showed bone remodeling (Figure 6) and the patient had normal and complete range of motion of the shoulder (Figure 7).
Discussion *Reprint requests: Amara Karim, MD, Department of Orthopedic Surgery, CHU Fattouma Bourguiba Monastir, Rue 1 Juin, 5000 Monastir, Tunisia. E-mail address: [email protected] (K. Amara).
Osteochondromas are generally asymptomatic and only discovered when symptoms occur. Symptoms consist of
1058-2746/$ - see front matter Ó 2010 Journal of Shoulder and Elbow Surgery Board of Trustees. doi:10.1016/j.jse.2010.04.004
Giant osteochondroma of proximal humerus
e13
Figure 1
Figure 2
Clinical aspect of tumor.
Anteroposterior radiographs show a well-circumscribed ossifying lesion, originating from the proximal humerus.
limitation of joint motion, edema, or paresthesia due to neurovascular compression. Painful osteochondromas may be explained by the presence of bursitis that covers the cup of the tumor.1 Osteochondromas of the proximal humerus may become painful and may cause mechanical impairment of the
glenohumeral joint. Despite the fact that osteochondromas are usually asymptomatic, the osteochondroma’s initial size in this case could explain the symptoms: it was impinging with the rotator cuff structures, being underneath the deltoid. Pain could be also explained by mechanical irritation of the deltoid
e14
M. Allagui et al.
Figure 5 After resection, intramedullary nailing of the humerus was performed.
Figure 3 Axial computed tomography scan shows a nearcircumferential tumor with medullar and cortical continuity.
Figure 6
Figure 4 Microscopic appearance consists of cartilage cap covering trabecular bone (hematoxylin-eosin stain, original magnification 40).
and biceps muscles.7 A painful osteochondroma should give rise to a high suspicion for sarcomatous degeneration. This event is seen in approximately 1% to 2% of patients with solitary osteochondroma,5 but the rate of malignant degeneration increases to 5% to 25% with hereditary multiple osteochondromas.8,10 The initial image can be easily mistaken for a chondroma or chondrosarcoma.4,6 In our case, the findings of histologic examination were consistent with a benign osteochondroma. The lesion had a regular cartilage cap, and the chondrocytes did not have a disorganized appearance. In the literature, the method of surgical treatment consists of combined resection and reconstruction with fibular or tibial nonvascularized graft with safe margins.2,9 Grafting procedures could be considered in cases of extensive involvement of the diaphyseal cortical wall, which leads to weakness. The
Final follow-up shows bone restoration.
wide involvement of the cortical wall sometimes makes the grafting procedure necessary. Resection alone is advocated in cases of a stalked bony protuberance of modest size. Intramedullary nailing (‘‘elastic stable intramedullary nailing’’ or ‘‘embrochage centro- me´dullaire elastique stable’’) has shown superb results in the treatment of nonpathologic fractures of the long bones in childhood. This method is rapidly gaining popularity for the treatment of spontaneous or pathologic fractures.3 Our goal is to stabilize an eventual pathologic fracture and the biomechanically weakened humerus. Accordingly, the intramedullary nailing could be described as a safe method: it avoids a second incision, provides good stability, and maintains normal growth for the humerus without impairment of shoulder function.
Conclusion A rare case of giant osteochondroma of the proximal humerus is presented. The tumor was entirely resected and stabilized by intramedullary nailing. This method was not reported previously in the literature. In fact, it is a relatively simple procedure that provides good
Giant osteochondroma of proximal humerus
Figure 7
e15
Clinical aspect at final follow-up.
stabilization and avoids bone grafting. As a result, at 6 months postoperatively, the patient recovered without complications and with normal functional results.
Disclaimer The authors, their immediate families, and any research foundations with which they are affiliated have not received any financial payments or other benefits from any commercial entity related to the subject of this article and have no potential conflicts of interest related to this manuscript.
References 1. D’Ambrosia R, Ferguson AB Jr. The formation of osteochondroma by epiphyseal cartilage transplantation. Clin Orthop Relat Res 1968;61: 103-15.
2. Gebhart M, Vadoud Seyedi J, Lejeune F. Giant solitary osteochondroma of the proximal humerus treated by resection and fibular autograft reconstruction. Acta Orthop Belg 1991;57:447-51. 3. Huber RI, Keller HW, Huber PM, Rehm KE. Flexible intramedullary nailing as fracture treatment in children. J Pediatr Orthop 1996;16:602-5. 4. Lange RH, Lange TA, Rao BK. Correlative radiographic, scintigraphic, and histological evaluation of exostoses. J Bone Joint Surg Am 1984;66:1454-9. 5. Mavrogenis AF, Papagelopoulos PJ, Soucacos PN. Skeletal osteochondromas revisited. Orthopedics 2008 Oct;31(10). pii: orthosupersite.com/view.asp?rID=32071 6. Milgram JW. The origins of osteochondromas and enchondromas. A histopathologic study. Clin Orthop Relat Res 1983:264-84. 7. Ogawa K, Yoshida A, Ui M. Symptomatic osteochondroma of the clavicle. A report of two cases. J Bone Joint Surg Am 1999;81:404-8. 8. Ozaki T, Hillmann A, Blasius S, Link T, Winkelmann W. Multicentric malignant transformation of multiple exostoses. Skeletal Radiol 1998; 27:233-6. 9. Taminiau AH, Hooning van Duyvenbode JF, Slooff TJ. Reconstruction of the proximal humerus with tibial autografts after resection of osteochondroma in adolescents. Clin Orthop Relat Res 1985:173-8. 10. Willms R, Hartwig CH, Bohm P, Sell S. Malignant transformation of a multiple cartilaginous exostosisda case report. Int Orthop 1997;21: 133-6.