HAND/PERIPHERAL NERVE Hand Tumors: II. Benign and Malignant Bone Tumors of the Hand Megan Henderson, M.D. Michael W. Neumeister, M.D. Reuben A. Bueno, Jr., M.D. Springfield, Ill.
Summary: The incidence of both benign and malignant bone tumors arising in the hand is relatively low in comparison with other locations. Although the overwhelming majority of these tumors are benign, even benign tumors can be locally destructive and compromise hand function. Intralesional tumor excision is the most appropriate surgical intervention for many benign bone tumors of the hand; however, destructive or malignant tumors may require wide local excision or even amputation to achieve complete tumor eradication. The purpose of this review article is to provide an overview of the pertinent benign and malignant bone tumors that may be encountered by hand surgeons. Clinical presentation, radiographic features, recommended workup, and available treatment options are all reviewed. (Plast. Reconstr. Surg. 133: 814e, 2014.)
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f all bone tumors, only 6 percent occur in the hand.1 The overwhelming majority of these tumors are benign. In general, benign bone tumors of the hand are more locally aggressive than their counterparts in other locations, whereas malignant bony hand tumors tend to behave less aggressively. The conspicuous nature of the hand and its utility in everyday function often serve to highlight problematic lesions and to facilitate early patient evaluation. A thorough working knowledge of the wide range of benign and malignant diseases affecting the hand is necessary for hand surgeons. The purpose of this article is to review the underlying disease, presentation, and treatment options for the most common benign and malignant bone tumors arising in the hand. The most frequent presenting complaints of bone tumors are pain and localized swelling. Symptoms may be new onset or chronic. Any unexplained fracture with a history of minor or no trauma should raise suspicion for pathologic fracture from a bony tumor. Workup of all patients should first include a thorough history and physical examination from which a differential diagnosis can be formulated. Hand examination should include inspection of the dorsal and volar surfaces for obvious deformity, skin changes, or localized swelling. Full neurologic examination From the Institute for Plastic and Reconstructive Surgery, Southern Illinois University School of Medicine. Received for publication April 25, 2013; accepted December 3, 2013. Copyright © 2014 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000000178
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of the median, ulnar, and radial nerves should be undertaken, and assessment of range of motion, joint stability, and tendon function should be performed. Diagnostic imaging often begins with plain radiographs. Benign bone tumors exhibit cortical expansion within well-defined borders; conversely, malignancy is often illustrated by cortical destruction, poorly defined borders, and soft-tissue extension. Computed tomographic scanning helps to further delineate location, size, and bony architecture. Magnetic resonance imaging is indicated when significant soft-tissue extension is present. The establishment of a definitive diagnosis is completed through biopsy. Biopsies should be carefully planned to avoid contamination of unaffected tissues. Optimal treatment of bone tumors requires a careful balance of local tumor control and preservation of hand function. Treatment plans are influenced by factors such as tumor size, risk of recurrence, proximity to joint surfaces, and overall predicted function. Benign bone tumors can be successfully treated with curettage excision. Cytotoxic adjuvant agents such as liquid nitrogen, phenol, and hydrogen peroxide can be used in conjunction with curettage to enhance the area of tumor kill, although their efficacy is still controversial.2–4 The addition of bone graft or synthetic bone enhances stability and assists in preventing pathologic fracture. When insufficient bone Disclosure: The authors have no financial interest to declare in relation to the content of this article. No external funding was received.
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Volume 133, Number 6 • Bone Tumors of the Hand stock is available for salvage, en bloc excision with reconstruction is needed. Reconstructive options include osteoarticular allograft, vascularized or nonvascularized bone graft from both local (i.e., distal radius) or distant sites (i.e., fibula or iliac crest), and arthrodesis.5,6 Most malignant bone tumors require a multidisciplinary approach to treatment, including chemotherapy or irradiation in addition to wide local excision or amputation.
BENIGN BONE TUMORS Enchondromas Enchondromas account for nearly 90 percent of all primary bone tumors encountered in the hand.7,8 Thought to arise from aberrant cartilaginous foci, these lesions are most commonly located in the proximal phalanx, metacarpals, and the middle phalanx. Several reports indicate a predilection for ulnar digits.9–11 Although benign, enchondromas are expansile and locally destructive such that pathologic fractures are a common presenting complaint. Typical radiographic findings include a well-circumscribed, radiolucent lesion with a characteristic stippling or popcorn-like appearance from increased calcification4 (Fig. 1). Potential for malignant transformation increases with multiple endochondromatosis (Fig. 2), as in the case of both Ollier disease and Maffuci syndrome. Ollier disease is a nonhereditary form of multiple enchondromatosis. Lesions typically occur in a unilateral fashion in the hands and feet. Similarly, Maffucci syndrome is marked by multiple enchondromas in addition to hemangiomas. Hand involvement in this condition is a common finding.12,13 Malignant transformation to osteosarcoma or chondrosarcoma can occur in
up to 30 percent of enchondromatosis patients, warranting meticulous longitudinal observation, with biopsy of all suspicious lesions.7,14,15 Small or asymptomatic lesions may be simply observed.7 Larger, symptomatic, or suspicious tumors are treated with intralesional surgical excision with or without bone grafting. The addition of autogenous, allogenous, or synthetic bone to the curetted cavity is advocated by most authors7,9 (Fig. 3). In a review of 102 cases, Sassoon et al. found no difference in healing time, recurrence, complications, range of motion, or malignant transformation when comparing various types of autogenous and allogenous grafting materials.16 Alternatively, some authors refute the need for bone grafting to achieve adequate bony healing, citing decreased surgical times, infection risk, and surgical costs as potential benefits.17,18 Pathologic fracture at the time of presentation may complicate treatment. Theoretical advantages of single-stage tumor excision and fracture reduction include a timely definitive diagnosis and the avoidance of prolonged immobilization. Many studies have shown no difference in complication rates between early and delayed treatment groups.16 However, in 2000, Ablove et al. noted a significantly higher rate of complications—namely, residual extensor lag—in patients undergoing tumor excision before pathologic fracture healing.10,19 Calcium phosphate bone cement20 and injectable calcium sulfate cement have been recently used in single-stage procedures with encouraging results, leading to early mobilization and minimal joint stiffness.19 Osteoid Osteomas Five to 15 percent of all osteoid osteomas arise in the hand, constituting nearly 10 percent of all
Fig. 1. Isolated enchondroma of the proximal phalanx of the long finger. (Left) Radiographic appearance. (Right) Intraoperative appearance.
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Plastic and Reconstructive Surgery • June 2014 pain relieved by nonsteroidal antiinflammatory drugs. Radiographs characteristically reveal a central radiolucent nidus with surrounding sclerosis (Fig. 4). Computed tomographic scan may be indicated to confirm the diagnosis if not apparent on radiography.15 When the diagnosis is certain, symptomatic management with nonsteroidal antiinflammatory drugs is an accepted treatment option because of the nonprogressive nature of this tumor.21 When conservative measures fail to relieve symptoms, curettage with bone grafting or en bloc excision can yield recurrence rates as low as 6 percent with removal of the central nidus.4,22–24 Recently, percutaneous radiofrequency ablation in the proximal forearm and spine has been implemented with promising results; however, potential thermal damage to adjacent neurovascular structures merits further investigation regarding its use in the hand.25–27
bone tumors in the hand.7 Osteoid osteomas are benign, osteoblastic tumors frequently located in the metacarpals, proximal phalanges, and carpal bones of younger populations. Typical presentation includes localized swelling with nighttime
Periosteal Chondromas Periosteal chondromas are rare, benign, cartilaginous tumors predominately affecting male patients during the second and third decades.7 Radiographic findings show a subperiosteal, lytic lesion with “scalloping” of the underlying cortex. Histologically, these lesions can resemble chondrosarcomas. Accurate diagnosis is thus necessary to avoid excessive resection. Treatment consists of marginal excision with inclusion of the
Fig. 3. Enchondroma of the proximal phalanx of the thumb. (Left) Preoperative radiographic appearance. (Right) Postoperative appearance after treatment with curettage and iliac crest bone graft.
Fig. 4. Radiograph of osteoid osteoma at the head of the proximal phalanx. Note nidus and halo lucency.
Fig. 2. Radiograph of enchondromatosis with lesions at multiple locations, including the third metacarpal and proximal phalanges of the index and middle fingers.
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Volume 133, Number 6 • Bone Tumors of the Hand overlying periosteum.7,28 Additional removal of the underlying cortex has been reported to prevent recurrence.4 Osteochondromas Osteochondromas are benign, bony tumors infrequently encountered in the hand, except in cases of multiple hereditary exostosis.7 Lesions frequently arise in the distal aspect of the proximal phalanx, but involvement of the scaphoid, hamate, and lunate have all been reported.4,29 The characteristic appearance is marked by an osseous growth capped with cartilage, extending beyond the cortex on a narrow stalk (Fig. 5). Secondary to their protuberant shape, symptoms of osteochondromas include compression of superficial nerves, limited range of motion, a snapping wrist, tendon rupture, and/or gross cosmetic deformity.29,30 Asymptomatic lesions may be observed, but marginal excision is warranted for symptomatic relief. No reports of malignant transformation for lesions involving the hand have been reported.7 Giant Cell Tumors of the Bone Giant cell tumors of the bone are benign but locally aggressive bone tumors that uniquely possess the potential for pulmonary metastasis. Only 2 to 5 percent of all giant cell tumors of the bone arise from the hand, with locations in the metacarpals, phalanges, and all carpal bones described.7,15,31 The distal radius is the third most common site of origination. Hand and distal radius lesions have higher incidences of recurrence and metastasis than other locations.15,31,32
Presentation is typically during the third decade, and radiographs frequently reveal juxta-articular, lytic lesions with cortical expansion. The Campanacci grading system is implemented to describe radiographic features15,32 (Table 1). Bone scans and computed tomographic imaging are recommended to identify multicentric disease and bony architecture. Computed tomographic scanning of the chest to evaluate for pulmonary metastasis should be included in the workup of these lesions. The destructive nature and predilection for recurrence of giant cell tumors of the bone have led many authors to advocate more radical surgical interventions such as wide local excision or amputation. For Campannaci grade I and II tumors, curettage with bone grafting or cement is generally recommended.32 Additional adjuvant treatments such as phenol, liquid nitrogen, and hydrogen peroxide have been advocated by some to increase tumor kill through their cytotoxic chemical and thermal properties. Recurrence rates of less than 20 percent have been cited in the literature following the addition of adjuvant therapies to curettage.2,31,33–35 However, reports on the efficacy of adjuvants remain divided. A recent review of 349 cases of giant cell tumors of the bone revealed no statistically significant decrease in recurrence rates for tumors treated with adjuvant therapy compared with curettage alone.3 A multicenter study in 2002 comparing multiple adjuvant modalities in 189 cases showed no significant association between
Fig. 5. Osteochondroma of the proximal phalanx of the ring finger. (Left) Radiographic appearance. (Right) Intraoperative appearance. Note the proximity to the proximal interphalangeal joint.
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Plastic and Reconstructive Surgery • June 2014 Table 1. Campanacci Grading System* Grade I II III
Description Well-marginated, intact cortex Well-marginated, expanded cortex Ill-defined, soft-tissue extension, cortical destruction
*Data from Trumble TE, Budoff JE. Hand Surgery Update IV. Chicago, Ill: American Society for Surgery of the Hand; 2007; and Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am. 1987;69:106–114.
the risk of recurrence, the adjuvant method, or filling material.3 Grade III lesions treated with intralesional excision have shown recurrence rates as high as 100 percent.4 As a result, extralesional treatment with en bloc excision and bony reconstruction is advocated.4,31,32,36 Carpal bone involvement requires en bloc excision.37 Both vascularized and nonvascularized fibular reconstruction for distal radius tumors have been described.6,32 Following comparison of vascularized free fibular transfer versus nonvascularized iliac crest transfer in 27 patients with giant cell tumors of the bone of the distal radius, Clarkson et al. recommended nonvascularized iliac crest transfer because of decreased surgical times and reoperation rates. The exception is in cases of significant soft-tissue defects or osseous defects larger than 10 cm, where vascularized free fibular transfer proved the optimal reconstructive option.38 Aneurysmal Bone Cysts Aneurysmal bone cysts are benign, locally aggressive bone tumors that are rare in the hand. Metacarpal involvement is common. Lesions may arise de novo or secondary to other benign lesions such as giant cell tumors. There is no associated risk of metastasis.32 Radiographic findings may be indistinguishable from giant cell tumors of the bone or enchondromas7; magnetic resonance imaging, however, can reveal fluid levels consistent with the diagnosis of this tumor. Risk of local recurrence may be as high as 60 percent when treated with curettage and bone grafting alone.7,39 Adjuvant treatments such as liquid nitrogen, argon laser, cement, burring, and liquid nitrogen may decrease recurrence rates to less than 20 percent15,39,40 and should be considered in the presence of articular surface involvement, complete bony involvement, or recurrent lesions.4,41 Liquid nitrogen should be used with caution in skeletally immature patient because of the risk of premature physeal closure.42 Wide local excision or amputation is reserved for aggressive lesions
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where insufficient bone stock following resection precludes salvage.
MALIGNANT BONE TUMORS Chondrosarcomas Chondrosarcomas are malignant, cartilaginous lesions that, despite their rare occurrence, are the most common malignant bone tumor in the hand. Most arise de novo, but secondary development in the setting of enchondromas or osteochondromas has been observed. The metacarpals and proximal phalanges are frequent sites of occurrence. Pain and swelling may be present for years before treatment is sought, reflecting their slow-growing nature. Radiographs reveal poor margination, cortical expansion, and frequent soft-tissue extension.7 Although hand involvement is generally associated with locally aggressive and high-grade lesions, metastasis is rare with lesions of the phalanges.43–47 Recurrence rates with intralesional excision are historically high, such that wide excision or amputation had previously been the mainstay of surgical treatment.7,15,43,45 Recent literature, however, advocates intralesional excision with close follow-up for low-grade lesions or phalangeal tumors in circumstances where amputation will result in significant functional loss.44,46 No role for irradiation or chemotherapy has been described.15 Osteosarcomas Osteosarcomas are malignant tumors derived from osteoblastic cells. Hand osteosarcomas tend to have a later onset compared with other locations in the body.48 The reported incidence is as low as 0.9 percent.49 Presentation includes an acute onset of pain and localized swelling of the proximal phalanx or metacarpals. Radiographs reveal an expansile, lytic lesion located centrally, juxtacortically or extraosseously. Soft-tissue extension may be present. Surgical intervention involves wide en bloc excision or amputation of the digit or ray to achieve negative surgical margins.7,15 Neoadjuvant or adjuvant chemotherapy is recommended for high-grade lesions to increase survival rates and lower rates of local recurrence.7,48,50 Five-year overall survival rates with the use of multidrug chemotherapy range from 71 to 87 percent.51–54 One large study conducted by Ferrani et al. of 157 patients receiving both neoadjuvant and adjuvant chemotherapy in addition to surgical resection reported an incidence of local recurrence as low as 4 percent.51 Therefore, a theoretical reduction of tumor size, localized
Volume 133, Number 6 • Bone Tumors of the Hand edema precluding easy surgical resection, and elimination of micrometastasis through the use of chemotherapy may potentiate more conservative surgical measures. Ewing Sarcoma Ewing sarcoma is a common pediatric bone tumor, although only 1.4 percent of all lesions occur in the upper extremity.55 The predilection is for male patients during the second decade, and the presentation may be insidious—including complaints of fevers, pain, swelling, and malaise—and can lead to the inaccurate diagnosis of infection.1,7 Radiographs and magnetic resonance imaging aid in making a correct diagnosis by demonstrating local bony destruction, periosteal reaction, and soft-tissue extension. Metastasis is identified in 25 percent of presenting cases.56 Surgical excision involves wide local resection or amputation. Neoadjuvant or adjuvant chemotherapy has been shown to improve overall survival and is recommended in all cases of Ewing sarcoma.7,55,56 Irradiation has been advocated for inoperable tumors or for cases where negative margins cannot be successfully achieved, yielding rates of local control as high as 89 percent in some studies.1,55,57 Metastatic Tumors Metastatic tumors to the hand are rare, constituting only 0.1 percent of all skeletal metastases.15 However, almost half of these cases occur as the initial presenting patient complaint.58–60 The predilection is for the distal phalanx, where they occur as lytic bony lesions (Fig. 6). Common primary tumor sites include the lung, kidney, and breast.1 When metastasis to the hand occurs, the overall prognosis is usually poor, with surgical intervention aimed at palliative treatment. The exception is isolated renal cell carcinoma, where excision of both the distal extremity lesion and the primary tumor have shown increased rates of survival.1,7
Fig. 6. Metastatic squamous cell carcinoma of the lung involving the proximal phalanx of the thumb. (Left) Clinical appearance. (Right) Radiographic appearance of a metastatic lesion. Note erosion and loss of bone from the metastatic tumor.
benign and malignant bone tumors is made by tissue biopsy. Benign tumors are commonly managed with intralesional surgical excision through curettage, with or without the addition of adjuvant therapies. Locally destructive lesions may require wide local excision with reconstruction or amputation if function cannot be preserved. Complete tumor resection is the primary goal of surgical treatment, with secondary goals of reconstruction of stability, movement, and appearance to restore form and function. Irradiation and chemotherapy play an integral role in the current management of many types of malignant bone tumors of the hand, contributing to both the increasing rate of salvage surgical procedures and overall increased survival rates. Reuben A. Bueno, Jr., M.D. Division of Plastic Surgery Southern Illinois University School of Medicine P.O. Box 19653 Springfield, Ill. 62794-9653 [email protected]
SUMMARY A wide variety of bone tumors can affect the hand, wrist, and proximal forearm. Fortunately, the incidence of occurrence is rare in comparison with alternative locations, and the majority of these tumors are benign. Each patient should be approached with a thorough hand and forearm examination, including inspection of skin, subcutaneous tissue, bony architecture, and tendon and nerve function. Definitive diagnosis for both
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28. Nosanchuk JS, Kaufer H. Recurrent periosteal chondroma: Report of two cases and a review of the literature. J Bone Joint Surg Am. 1969;51:375–380. 29. Katayama T, Ono H, Furuta K. Osteochondroma of the lunate with extensor tendons rupture of the index finger: A case report. Hand Surg. 2011;16:181–184. 30. Shah NR, Wilczynski M, Gelberman R. Osteochondroma of the capitate causing rupture of the extensor digiti minimi: Case report. J Hand Surg Am. 2009;34:46–48. 31. Athanasian EA, Wold LE, Amadio PC. Giant cell tumors of the bones of the hand. J Hand Surg Am. 1997;22:91–98. 32. Athanasian EA. Aneurysmal bone cyst and giant cell tumor of bone of the hand and distal radius. Hand Clin. 2004;20:269–281, vi. 33. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am. 1987;69:106–114. 34. Rock MG. Curettage of giant-cell tumor of bone: Factor influencing local recurrences and metastasis. Chir Organi Mov. 1990;75(Suppl 1):204–205. 35. Capanna R, Fabbri N, Bettelli G. Curettage of giant cell tumor of bone: The effect of surgical technique and adjuvants on local recurrence rate. Chir Organi Mov. 1990;75(1 Suppl):206. 36. Athanasian EA, McCormack RR. Recurrent aneurysmal bone cyst of the proximal phalanx treated with cryosurgery: A case report. J Hand Surg Am. 1999;24:405–412. 37. Shigematsu K, Kobata Y, Yajima H, Kawamura K, Maegawa N, Takakura Y. Giant-cell tumors of the carpus. J Hand Surg Am. 2006;31:1214–1219. 38. Clarkson PW, Sandford K, Phillips AE, et al. Functional results following vascularized versus nonvascularized bone grafts for wrist arthrodesis following excision of giant cell tumors. J Hand Surg Am. 2013;38:935–940.e1. 39. Marcove RC, Sheth DS, Takemoto S, Healy JH. The treatment of aneurysmal bone cyst. Clin Orthop Relat Res. 1995;311:157–163. 40. Jafari D, Jamshidi K, Najdmazhar F, Shariatzade H, Liaghat O. Expansile aneurysmal bone cyst in the tubular bones of the hand treated with en bloc excision and autograft reconstruction: A report of 12 cases. J Hand Surg Eur Vol. 2011;36:648–655. 41. Ropars M, Kaila R, Briggs T, Cannon S. Aneurysmal bone cysts of the metacarpals and phalanges of the hand: A 6 case series and literature review. Chir Main 2007;26:214–217. 42. Campanacci M, Giunti A, Olmi R. Giant cell tumors of the bone: A study of 209 cases with long-term follow-up in 130. Ital J Orthop Traumatol. 1975;1:24–27. 43. Patil S, de Silva MV, Crossan J, Reid R. Chondrosarcoma of small bones of the hand. J Hand Surg Br. 2003;28:602–608. 44. Mittmayer F, Dominkus M, Krepler P, et al. Chondrosarcoma of the hand: Is a wide surgical resection necessary? Clin Orthop Rel Res. 2004;24:211–215. 45. Ogose A, Unni KK, Swee RG, May GK, Rowland CM, Sim FH. Chondrosarcoma of small bones of the hands and feet. Cancer 1997;80:50–59. 46. Bovée JV, van der Heul RO, Taminiau AH, Hogendoorn PC. Chondrosarcoma of the phalanx: A locally aggressive lesion with minimal metastatic potential: A report of 35 cases and a review of the literature. Cancer 1999;86:1724–1732. 47. Mankin HJ. Chondrosarcomas of digits: Are they really malignant? Cancer 1999;86:1635–1637. 48. Anninga JK, Picci P, Fiocco M, et al. Osteosarcoma of the hands and feet: A distinct clinico-pathological subgroup. Virchows Arch. 2013;462:109–120. 49. Fowble VA, Pae R, Vitale A, Bryk E, Vigorita VJ. Case Reports: Osteosarcoma of the hand: One case and a literature review. Clin Orthop Relat Res. 2005;440:255–261.
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Summary: The incidence of both benign and malignant bone tumors arising in the hand is relatively low in comparison with other locations. Although the overwhelming majority of these tumors are benign, even benign tumors can be locally destructive and compromise hand function. Intralesional tumor excision is the most appropriate surgical intervention for many benign bone tumors of the hand; however, destructive or malignant tumors may require wide local excision or even amputation to achieve complete tumor eradication. The purpose of this review article is to provide an overview of the pertinent benign and malignant bone tumors that may be encountered by hand surgeons. Clinical presentation, radiographic features, recommended workup, and available treatment options are all reviewed. (Plast. Reconstr. Surg. 133: 814e, 2014.)
O
f all bone tumors, only 6 percent occur in the hand.1 The overwhelming majority of these tumors are benign. In general, benign bone tumors of the hand are more locally aggressive than their counterparts in other locations, whereas malignant bony hand tumors tend to behave less aggressively. The conspicuous nature of the hand and its utility in everyday function often serve to highlight problematic lesions and to facilitate early patient evaluation. A thorough working knowledge of the wide range of benign and malignant diseases affecting the hand is necessary for hand surgeons. The purpose of this article is to review the underlying disease, presentation, and treatment options for the most common benign and malignant bone tumors arising in the hand. The most frequent presenting complaints of bone tumors are pain and localized swelling. Symptoms may be new onset or chronic. Any unexplained fracture with a history of minor or no trauma should raise suspicion for pathologic fracture from a bony tumor. Workup of all patients should first include a thorough history and physical examination from which a differential diagnosis can be formulated. Hand examination should include inspection of the dorsal and volar surfaces for obvious deformity, skin changes, or localized swelling. Full neurologic examination From the Institute for Plastic and Reconstructive Surgery, Southern Illinois University School of Medicine. Received for publication April 25, 2013; accepted December 3, 2013. Copyright © 2014 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000000178
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of the median, ulnar, and radial nerves should be undertaken, and assessment of range of motion, joint stability, and tendon function should be performed. Diagnostic imaging often begins with plain radiographs. Benign bone tumors exhibit cortical expansion within well-defined borders; conversely, malignancy is often illustrated by cortical destruction, poorly defined borders, and soft-tissue extension. Computed tomographic scanning helps to further delineate location, size, and bony architecture. Magnetic resonance imaging is indicated when significant soft-tissue extension is present. The establishment of a definitive diagnosis is completed through biopsy. Biopsies should be carefully planned to avoid contamination of unaffected tissues. Optimal treatment of bone tumors requires a careful balance of local tumor control and preservation of hand function. Treatment plans are influenced by factors such as tumor size, risk of recurrence, proximity to joint surfaces, and overall predicted function. Benign bone tumors can be successfully treated with curettage excision. Cytotoxic adjuvant agents such as liquid nitrogen, phenol, and hydrogen peroxide can be used in conjunction with curettage to enhance the area of tumor kill, although their efficacy is still controversial.2–4 The addition of bone graft or synthetic bone enhances stability and assists in preventing pathologic fracture. When insufficient bone Disclosure: The authors have no financial interest to declare in relation to the content of this article. No external funding was received.
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Volume 133, Number 6 • Bone Tumors of the Hand stock is available for salvage, en bloc excision with reconstruction is needed. Reconstructive options include osteoarticular allograft, vascularized or nonvascularized bone graft from both local (i.e., distal radius) or distant sites (i.e., fibula or iliac crest), and arthrodesis.5,6 Most malignant bone tumors require a multidisciplinary approach to treatment, including chemotherapy or irradiation in addition to wide local excision or amputation.
BENIGN BONE TUMORS Enchondromas Enchondromas account for nearly 90 percent of all primary bone tumors encountered in the hand.7,8 Thought to arise from aberrant cartilaginous foci, these lesions are most commonly located in the proximal phalanx, metacarpals, and the middle phalanx. Several reports indicate a predilection for ulnar digits.9–11 Although benign, enchondromas are expansile and locally destructive such that pathologic fractures are a common presenting complaint. Typical radiographic findings include a well-circumscribed, radiolucent lesion with a characteristic stippling or popcorn-like appearance from increased calcification4 (Fig. 1). Potential for malignant transformation increases with multiple endochondromatosis (Fig. 2), as in the case of both Ollier disease and Maffuci syndrome. Ollier disease is a nonhereditary form of multiple enchondromatosis. Lesions typically occur in a unilateral fashion in the hands and feet. Similarly, Maffucci syndrome is marked by multiple enchondromas in addition to hemangiomas. Hand involvement in this condition is a common finding.12,13 Malignant transformation to osteosarcoma or chondrosarcoma can occur in
up to 30 percent of enchondromatosis patients, warranting meticulous longitudinal observation, with biopsy of all suspicious lesions.7,14,15 Small or asymptomatic lesions may be simply observed.7 Larger, symptomatic, or suspicious tumors are treated with intralesional surgical excision with or without bone grafting. The addition of autogenous, allogenous, or synthetic bone to the curetted cavity is advocated by most authors7,9 (Fig. 3). In a review of 102 cases, Sassoon et al. found no difference in healing time, recurrence, complications, range of motion, or malignant transformation when comparing various types of autogenous and allogenous grafting materials.16 Alternatively, some authors refute the need for bone grafting to achieve adequate bony healing, citing decreased surgical times, infection risk, and surgical costs as potential benefits.17,18 Pathologic fracture at the time of presentation may complicate treatment. Theoretical advantages of single-stage tumor excision and fracture reduction include a timely definitive diagnosis and the avoidance of prolonged immobilization. Many studies have shown no difference in complication rates between early and delayed treatment groups.16 However, in 2000, Ablove et al. noted a significantly higher rate of complications—namely, residual extensor lag—in patients undergoing tumor excision before pathologic fracture healing.10,19 Calcium phosphate bone cement20 and injectable calcium sulfate cement have been recently used in single-stage procedures with encouraging results, leading to early mobilization and minimal joint stiffness.19 Osteoid Osteomas Five to 15 percent of all osteoid osteomas arise in the hand, constituting nearly 10 percent of all
Fig. 1. Isolated enchondroma of the proximal phalanx of the long finger. (Left) Radiographic appearance. (Right) Intraoperative appearance.
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Plastic and Reconstructive Surgery • June 2014 pain relieved by nonsteroidal antiinflammatory drugs. Radiographs characteristically reveal a central radiolucent nidus with surrounding sclerosis (Fig. 4). Computed tomographic scan may be indicated to confirm the diagnosis if not apparent on radiography.15 When the diagnosis is certain, symptomatic management with nonsteroidal antiinflammatory drugs is an accepted treatment option because of the nonprogressive nature of this tumor.21 When conservative measures fail to relieve symptoms, curettage with bone grafting or en bloc excision can yield recurrence rates as low as 6 percent with removal of the central nidus.4,22–24 Recently, percutaneous radiofrequency ablation in the proximal forearm and spine has been implemented with promising results; however, potential thermal damage to adjacent neurovascular structures merits further investigation regarding its use in the hand.25–27
bone tumors in the hand.7 Osteoid osteomas are benign, osteoblastic tumors frequently located in the metacarpals, proximal phalanges, and carpal bones of younger populations. Typical presentation includes localized swelling with nighttime
Periosteal Chondromas Periosteal chondromas are rare, benign, cartilaginous tumors predominately affecting male patients during the second and third decades.7 Radiographic findings show a subperiosteal, lytic lesion with “scalloping” of the underlying cortex. Histologically, these lesions can resemble chondrosarcomas. Accurate diagnosis is thus necessary to avoid excessive resection. Treatment consists of marginal excision with inclusion of the
Fig. 3. Enchondroma of the proximal phalanx of the thumb. (Left) Preoperative radiographic appearance. (Right) Postoperative appearance after treatment with curettage and iliac crest bone graft.
Fig. 4. Radiograph of osteoid osteoma at the head of the proximal phalanx. Note nidus and halo lucency.
Fig. 2. Radiograph of enchondromatosis with lesions at multiple locations, including the third metacarpal and proximal phalanges of the index and middle fingers.
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Volume 133, Number 6 • Bone Tumors of the Hand overlying periosteum.7,28 Additional removal of the underlying cortex has been reported to prevent recurrence.4 Osteochondromas Osteochondromas are benign, bony tumors infrequently encountered in the hand, except in cases of multiple hereditary exostosis.7 Lesions frequently arise in the distal aspect of the proximal phalanx, but involvement of the scaphoid, hamate, and lunate have all been reported.4,29 The characteristic appearance is marked by an osseous growth capped with cartilage, extending beyond the cortex on a narrow stalk (Fig. 5). Secondary to their protuberant shape, symptoms of osteochondromas include compression of superficial nerves, limited range of motion, a snapping wrist, tendon rupture, and/or gross cosmetic deformity.29,30 Asymptomatic lesions may be observed, but marginal excision is warranted for symptomatic relief. No reports of malignant transformation for lesions involving the hand have been reported.7 Giant Cell Tumors of the Bone Giant cell tumors of the bone are benign but locally aggressive bone tumors that uniquely possess the potential for pulmonary metastasis. Only 2 to 5 percent of all giant cell tumors of the bone arise from the hand, with locations in the metacarpals, phalanges, and all carpal bones described.7,15,31 The distal radius is the third most common site of origination. Hand and distal radius lesions have higher incidences of recurrence and metastasis than other locations.15,31,32
Presentation is typically during the third decade, and radiographs frequently reveal juxta-articular, lytic lesions with cortical expansion. The Campanacci grading system is implemented to describe radiographic features15,32 (Table 1). Bone scans and computed tomographic imaging are recommended to identify multicentric disease and bony architecture. Computed tomographic scanning of the chest to evaluate for pulmonary metastasis should be included in the workup of these lesions. The destructive nature and predilection for recurrence of giant cell tumors of the bone have led many authors to advocate more radical surgical interventions such as wide local excision or amputation. For Campannaci grade I and II tumors, curettage with bone grafting or cement is generally recommended.32 Additional adjuvant treatments such as phenol, liquid nitrogen, and hydrogen peroxide have been advocated by some to increase tumor kill through their cytotoxic chemical and thermal properties. Recurrence rates of less than 20 percent have been cited in the literature following the addition of adjuvant therapies to curettage.2,31,33–35 However, reports on the efficacy of adjuvants remain divided. A recent review of 349 cases of giant cell tumors of the bone revealed no statistically significant decrease in recurrence rates for tumors treated with adjuvant therapy compared with curettage alone.3 A multicenter study in 2002 comparing multiple adjuvant modalities in 189 cases showed no significant association between
Fig. 5. Osteochondroma of the proximal phalanx of the ring finger. (Left) Radiographic appearance. (Right) Intraoperative appearance. Note the proximity to the proximal interphalangeal joint.
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Plastic and Reconstructive Surgery • June 2014 Table 1. Campanacci Grading System* Grade I II III
Description Well-marginated, intact cortex Well-marginated, expanded cortex Ill-defined, soft-tissue extension, cortical destruction
*Data from Trumble TE, Budoff JE. Hand Surgery Update IV. Chicago, Ill: American Society for Surgery of the Hand; 2007; and Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am. 1987;69:106–114.
the risk of recurrence, the adjuvant method, or filling material.3 Grade III lesions treated with intralesional excision have shown recurrence rates as high as 100 percent.4 As a result, extralesional treatment with en bloc excision and bony reconstruction is advocated.4,31,32,36 Carpal bone involvement requires en bloc excision.37 Both vascularized and nonvascularized fibular reconstruction for distal radius tumors have been described.6,32 Following comparison of vascularized free fibular transfer versus nonvascularized iliac crest transfer in 27 patients with giant cell tumors of the bone of the distal radius, Clarkson et al. recommended nonvascularized iliac crest transfer because of decreased surgical times and reoperation rates. The exception is in cases of significant soft-tissue defects or osseous defects larger than 10 cm, where vascularized free fibular transfer proved the optimal reconstructive option.38 Aneurysmal Bone Cysts Aneurysmal bone cysts are benign, locally aggressive bone tumors that are rare in the hand. Metacarpal involvement is common. Lesions may arise de novo or secondary to other benign lesions such as giant cell tumors. There is no associated risk of metastasis.32 Radiographic findings may be indistinguishable from giant cell tumors of the bone or enchondromas7; magnetic resonance imaging, however, can reveal fluid levels consistent with the diagnosis of this tumor. Risk of local recurrence may be as high as 60 percent when treated with curettage and bone grafting alone.7,39 Adjuvant treatments such as liquid nitrogen, argon laser, cement, burring, and liquid nitrogen may decrease recurrence rates to less than 20 percent15,39,40 and should be considered in the presence of articular surface involvement, complete bony involvement, or recurrent lesions.4,41 Liquid nitrogen should be used with caution in skeletally immature patient because of the risk of premature physeal closure.42 Wide local excision or amputation is reserved for aggressive lesions
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where insufficient bone stock following resection precludes salvage.
MALIGNANT BONE TUMORS Chondrosarcomas Chondrosarcomas are malignant, cartilaginous lesions that, despite their rare occurrence, are the most common malignant bone tumor in the hand. Most arise de novo, but secondary development in the setting of enchondromas or osteochondromas has been observed. The metacarpals and proximal phalanges are frequent sites of occurrence. Pain and swelling may be present for years before treatment is sought, reflecting their slow-growing nature. Radiographs reveal poor margination, cortical expansion, and frequent soft-tissue extension.7 Although hand involvement is generally associated with locally aggressive and high-grade lesions, metastasis is rare with lesions of the phalanges.43–47 Recurrence rates with intralesional excision are historically high, such that wide excision or amputation had previously been the mainstay of surgical treatment.7,15,43,45 Recent literature, however, advocates intralesional excision with close follow-up for low-grade lesions or phalangeal tumors in circumstances where amputation will result in significant functional loss.44,46 No role for irradiation or chemotherapy has been described.15 Osteosarcomas Osteosarcomas are malignant tumors derived from osteoblastic cells. Hand osteosarcomas tend to have a later onset compared with other locations in the body.48 The reported incidence is as low as 0.9 percent.49 Presentation includes an acute onset of pain and localized swelling of the proximal phalanx or metacarpals. Radiographs reveal an expansile, lytic lesion located centrally, juxtacortically or extraosseously. Soft-tissue extension may be present. Surgical intervention involves wide en bloc excision or amputation of the digit or ray to achieve negative surgical margins.7,15 Neoadjuvant or adjuvant chemotherapy is recommended for high-grade lesions to increase survival rates and lower rates of local recurrence.7,48,50 Five-year overall survival rates with the use of multidrug chemotherapy range from 71 to 87 percent.51–54 One large study conducted by Ferrani et al. of 157 patients receiving both neoadjuvant and adjuvant chemotherapy in addition to surgical resection reported an incidence of local recurrence as low as 4 percent.51 Therefore, a theoretical reduction of tumor size, localized
Volume 133, Number 6 • Bone Tumors of the Hand edema precluding easy surgical resection, and elimination of micrometastasis through the use of chemotherapy may potentiate more conservative surgical measures. Ewing Sarcoma Ewing sarcoma is a common pediatric bone tumor, although only 1.4 percent of all lesions occur in the upper extremity.55 The predilection is for male patients during the second decade, and the presentation may be insidious—including complaints of fevers, pain, swelling, and malaise—and can lead to the inaccurate diagnosis of infection.1,7 Radiographs and magnetic resonance imaging aid in making a correct diagnosis by demonstrating local bony destruction, periosteal reaction, and soft-tissue extension. Metastasis is identified in 25 percent of presenting cases.56 Surgical excision involves wide local resection or amputation. Neoadjuvant or adjuvant chemotherapy has been shown to improve overall survival and is recommended in all cases of Ewing sarcoma.7,55,56 Irradiation has been advocated for inoperable tumors or for cases where negative margins cannot be successfully achieved, yielding rates of local control as high as 89 percent in some studies.1,55,57 Metastatic Tumors Metastatic tumors to the hand are rare, constituting only 0.1 percent of all skeletal metastases.15 However, almost half of these cases occur as the initial presenting patient complaint.58–60 The predilection is for the distal phalanx, where they occur as lytic bony lesions (Fig. 6). Common primary tumor sites include the lung, kidney, and breast.1 When metastasis to the hand occurs, the overall prognosis is usually poor, with surgical intervention aimed at palliative treatment. The exception is isolated renal cell carcinoma, where excision of both the distal extremity lesion and the primary tumor have shown increased rates of survival.1,7
Fig. 6. Metastatic squamous cell carcinoma of the lung involving the proximal phalanx of the thumb. (Left) Clinical appearance. (Right) Radiographic appearance of a metastatic lesion. Note erosion and loss of bone from the metastatic tumor.
benign and malignant bone tumors is made by tissue biopsy. Benign tumors are commonly managed with intralesional surgical excision through curettage, with or without the addition of adjuvant therapies. Locally destructive lesions may require wide local excision with reconstruction or amputation if function cannot be preserved. Complete tumor resection is the primary goal of surgical treatment, with secondary goals of reconstruction of stability, movement, and appearance to restore form and function. Irradiation and chemotherapy play an integral role in the current management of many types of malignant bone tumors of the hand, contributing to both the increasing rate of salvage surgical procedures and overall increased survival rates. Reuben A. Bueno, Jr., M.D. Division of Plastic Surgery Southern Illinois University School of Medicine P.O. Box 19653 Springfield, Ill. 62794-9653 [email protected]
SUMMARY A wide variety of bone tumors can affect the hand, wrist, and proximal forearm. Fortunately, the incidence of occurrence is rare in comparison with alternative locations, and the majority of these tumors are benign. Each patient should be approached with a thorough hand and forearm examination, including inspection of skin, subcutaneous tissue, bony architecture, and tendon and nerve function. Definitive diagnosis for both
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54. Bacci G, Briccoli A, Ferrari S, et al. Neoadjuvant chemotherapy for osteosarcoma of the extremity: Long-term results of the Rizzoli’s 4th protocol. Eur J Cancer 2001;37:2030–2039. 55. Parida L, Fernandez-Pineda I, Uffman J, et al. Clinical management of Ewing sarcoma of the bones of the hands and feet: A retrospective single-institution review. J Pediatr Surg. 2012;47:1806–1810. 56. Gorlick R, Janeway K, Lessnick S, Randall RL, Marina N. Children’s Oncology Groups 2013 blueprint for research: Bone tumors. Pediatr Blood Cancer 2016;60:1009–1015. 57. Krasin MJ, Rodriguez-Galindo C, Davidoff AM, et al. Efficacy of combined surgery and irradiation for localized Ewings sarcoma family of tumors. Pediatr Blood Cancer 2004;43:229–236. 58. Healey JH, Turnbull AD, Miedema B, Lane JM. Acrometastases: A study of twenty-nine patients with osseous involvement of the hands and feet. J Bone Joint Surg Am. 1986;68:743–746. 59. Hicks MC, Kalmon EH Jr, Glasser SM. Metastatic malignancy to phalanges. South Med J. 1964;57:85–88. 60. Patel MR, Sanchez HJ, Silver JW, Pearlman HS. Metastatic carcinoma of hand: Clinical manifestations of occult or overt bronchogenic carcinoma. N Y State J Med. 1978;78:2233–2237.
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