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Calcaneal Chondroblastoma with Pathologic Fracture and Recurrence
The Journal of Foot & Ankle Surgery 54 (2015) 258–267
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Calcaneal Chondroblastoma with Pathologic Fracture and Recurrence Laksha Dutt, DPM 1, Valerie L. Schade, DPM, FACFAS 2, Mark W. Manoso, MD 3 1
Postgraduate Year 2 Resident, Podiatric Medicine Surgical Residency, Madigan Army Medical Center, Tacoma, WA Chief, Limb Preservation Service, Madigan Army Medical Center, Tacoma, WA 3 Chief, Orthopedic Surgery Service, Madigan Army Medical Center, Tacoma, WA 2
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4
Chondroblastomas account for <2% of all bone tumors. The calcaneus is the fifth most common location of occurrence. Males in their second decade of life are most often affected, presenting with an insidious onset of localized pain, swelling, and tenderness. The finding of associated pathologic fracture has been rare. Imaging studies can aid in the formulation of the differential diagnosis and surgical plan. The definitive diagnosis requires histologic examination. Curettage and bone grafting is curative in >80% of cases. Local recurrence rates of 38% have been reported, most often because of inadequate resection, and have been associated with malignant conversion and metastasis. Adjuvant therapies can help minimize the incidence of local recurrence. Long-term follow-up examinations are recommended, given the protracted interval that can exist between recurrence and the potential for malignant conversion and metastasis. We present the case of a young, healthy, active male with a calcaneal chondroblastoma and associated pathologic fracture whose initial treatment consisted of curettage, hydrogen peroxide lavage, and allogeneic bone grafting. Recurrence developed at 15 months postoperatively and was treated with repeat curettage, high-speed burring, and reconstruction with steel Steinman pins and polymethylmethacrylate, resulting in no pain or recurrence at the 5-month follow-up point. Ó 2015 by the American College of Foot and Ankle Surgeons. All rights reserved.
Keywords: cartilaginous cryosurgery high-speed burring
Chondroblastoma is as a rare, benign, cartilaginous bone tumor derived from epiphyseal chondrocytes, accounting for 1% to 2% of all bone tumors (1–12). Kolodny first described the tumor in 1927 as a variant of a giant cell tumor (GCT) (4,5,13). Ewing described the tumor as a calcifying GCT in 1928, followed by Codman in 1931, who reported that the tumor only occurred in the humerus (2–8,13–16). Jaffe and Lichtenstein differentiated it from a GCT variant in 1942, coining the term “benign chondroblastoma,” and determined that the neoplasm arose in bones other than the humerus (2–8,13,14,16). Geschicket and Copeland later determined that both benign and malignant forms exist (5,13). Chondroblastomas typically present in males in the second decade of life, with a reported male/female ratio ranging from 2:1 to 5:1 (1–5,7–10,14,15,17,18). The area of occurrence has most frequently been in the epiphysis of the long bones, in particular, the femur, Financial Disclosure: None reported. Conflict of Interest: None reported. Disclaimer: The views expressed are those of the authors and do not reflect the official policy of the Department of the Army, the Department of Defense, or the US Government. Address correspondence to: Valerie L. Schade, DPM, FACFAS, Limb Preservation Service, Madigan Army Medical Center, 9040 Jackson Drive, MCHJ-CLS-V, Tacoma, WA 98431. E-mail address: [email protected] (V.L. Schade).
humerus, and proximal tibia, but it can also occur in the small bones of the hands and feet, which undergo endochondral ossification (1,2,5,7,9–11,13,17,18). The incidence in the foot has ranged from 3% to 23%, making it the second most common location of occurrence, with reports of occurrence in the navicular, medial cuneiform, cuboid, metatarsals, and phalanges (7,8,11,18–24). However, the tarsal bones most often affected are the talus and calcaneus, with a reported occurrence rate of 45% and 7% to 34%, respectively (7,8,20,21,25–32). The calcaneus has been reported to be the fifth most common location of occurrence (21). The clinical presentation most often involves the insidious onset of localized pain, soft tissue swelling, and tenderness (4–6,9,13–18). Radiographs will reveal an osteolytic lesion with a slow-growing appearance (3–9,11,16–18,33–35). Pathologic fracture is rare (7,17,18,36). Advanced imaging studies aid in formulating the differential diagnosis and surgical treatment plan (34). A histologic examination is essential to determine the definitive diagnosis (14,37). Surgical treatment ranges from simple curettage to en bloc resection (3,6,17). Although these procedures will most often be effective, recurrence has been reported to range from 6% to 38% and has been reported to occur more frequently in the tubular bones than in the long bones (29% versus 11%, respectively) (3,9,12,17,28,32,34,38–41). Curettage and adjuvant therapies have been reported to result in resolution in cases of recurrence (3,16,42). Malignant conversion and metastasis have been associated with local recurrence (38,40,43–47).
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was noted. Mild soft tissue edema at the medial subtalar joint and mild bone marrow edema within the calcaneus from the fracture were seen (Fig. 3). The radiographic work-up revealed no other primary malignancy or metastatic spread. The computed tomography (CT) scan revealed a well-defined osteolytic lesion abutting the posterior inferior aspect of the calcaneus, with no associated periosteal reaction. Cortical discontinuity with minimal diastasis was noted at the fracture site and involved the posterior facet of the subtalar joint (Fig. 2). Operative Technique Fig. 1. Index anteroposterior and calcaneal radiographs of the left foot.
Long-term monitoring is recommended for these reasons (43–45). We present a case of a calcaneal chondroblastoma with an associated pathologic fracture that occurred in a young, healthy, active-duty male soldier. Recurrence developed at 15 months postoperatively. The findings from our case signify the importance of an adequate initial resection and consideration of adjuvant techniques, the need to rule out metastasis in patients with local recurrence before surgical intervention, and the need for long-term monitoring.
Case Report A 21-year-old active-duty male soldier presented to a primary care provider in December 2007 with an 8-month history of left heel pain that began after he had jumped down an approximately 10 500 height from a vehicle while deployed. The patient had initially been diagnosed with Achilles tendinitis and prescribed a nonsteroidal antiinflammatory drug. He was also instructed to limit his activities; to use ice, to use an elastic bandage (for compression), to elevate, and to perform range of motion exercises. No radiographs were taken. In June 2008, the patient was referred for physical therapy because of continuing pain. The initial radiographs were taken in August 2008, revealing an osteolytic lesion in the posterior inferior calcaneus (Fig. 1). The patient was instructed to obtain a magnetic resonance imaging (MRI) study and was referred to the podiatry department. En route to obtaining the MRI scan, the patient stepped off a curb and noted immediate sharp pain and an inability to bear weight on his left foot. The patient proceeded to the emergency department for additional evaluation. Repeat radiographs revealed a pathologic fracture of the left calcaneus, with possible extension into the posterior facet of the subtalar joint (Fig. 2). The patient reported no history of fever, chills, or unexplained weight loss. The patient had no medical or surgical history and denied the use of any medication, apart from the previously prescribed nonsteroidal anti-inflammatory drug. The patient reported smoking 5 cigarettes daily for the previous 2 years. The patient related an allergy to sulfa medications and denied a family history of cancer. Focused examination of the lower extremity revealed the left foot to have an intact neurovascular status and pitting edema localized to the hind foot. Mondor’s sign and ecchymosis were present along the lateral heel rim. No local erythema or regional lymphadenopathy was appreciated. His muscle strength and range of motion of the ankle and subtalar joint were normal, apart from some guarding secondary to pain. The results from a complete blood count, basic metabolic panel, and serum albumin plus prealbumin, C-reactive protein, erythrocyte sedimentation rate, and alkaline phosphatase tests were all within normal limits. The MRI scan revealed a 3.9-cm 4.7-cm 3.2-cm solid lesion with no definite areas of calcification or cystic portions. A fracture running longitudinally, adjacent to the cyst, with involvement of the lateral calcaneal cortex and the posterior facet of the subtalar joint,
The operative technique is shown in Fig. 4. In September 2008, the patient was taken for his initial surgical procedure. The patient underwent general anesthesia and was placed in a right lateral decubitus position to allow access to the lateral aspect of the right foot. A thigh tourniquet was used. A 4-cm linear incision was placed over the lateral aspect of the calcaneus, angled from posterosuperiorly to anteroinferiorly, starting halfway between the Achilles tendon and the posterior aspect of the lateral malleolus and extending to the lateral bisection of the calcaneal tuberosity. Sharp and blunt dissection were performed to expose the calcaneus. A cortical window was created to expose the cavity of the tumor. The contents were thin, friable, and hemorrhagic. A portion of the lining and aspirated contents of the cavity underwent fresh frozen analysis, with no occult malignancy seen. Aggressive curettage was performed to remove the remaining contents and lining. The osseous void was then lavaged with copious amounts of hydrogen peroxide, followed by normal saline infused with bacitracin. Intraoperative cultures were obtained. The osseous void was filled with allogeneic bone graft mixed with bone marrow aspirate obtained from the proximal tibia to fill the void and restore the bone stock. A 4-hole locking plate was placed for fracture fixation and stabilization of the cortical window that had been created. Intraoperative Culture Results and Tissue Specimen Examination The intraoperative culture results were negative for aerobic and anaerobic growth (Fig. 5). The initial histologic examination revealed proliferation of multinucleated giant cells with cellular spindled stroma. Woven bony trabeculae were present, some with and some without osteoblastic rimming. New bone formation was evident, as well as areas of necrosis. Mitotic activity was rare. Although associated hemorrhage was present, the salient features of an aneurysmal bone cyst (ABC) were not. The differential diagnosis was GCT with an associated fracture callus, giant cell reparative granuloma, and other neoplasm. The specimen was sent to the Department of Orthopedic Pathology of the Armed Forces Institute of Pathology for additional evaluation. The final histologic examination revealed small mononuclear cells with multinucleated giant cells admixed. A suggestion of focal eosinophilic cartilage matrix production and extensive areas of infarction and necrosis was found. Focal staining of the tumor cells with S-100 was positive and focal staining with an aldehyde fuchsin stain at pH 1.0 was equivocal. Sulfated matrix production in a delicate “chicken-wire” distribution was seen around the individual tumor cells. The diagnosis was chondroblastoma. Postoperative Course The patient was immobilized in a short leg cast and instructed to be non-weightbearing for 8 weeks. The patient reported falling soon after the first postoperative visit and missed the second postoperative visit, and he had continued smoking. This resulted in superficial dehiscence of the incision site, which ultimately healed with nonoperative wound care and a 20-day course of oral antibiotics. A CT
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Fig. 2. Preoperative lateral radiographs and coronal and lateral computed tomography views after the patient had sustained the pathologic fracture.
scan obtained in November 2008 revealed incorporation of the allogeneic bone graft, stable hardware, and no subtalar joint arthrosis. At 3 months postoperatively, the patient was allowed to gradually progress back to normal shoe gear and full activity. At 5 months postoperatively, the patient reported complete resolution of the pain, had returned to full activity, and was preparing for an upcoming
deployment. Radiographs at that point revealed some persistent cystic changes at the most inferior and medial aspect of the calcaneus (Fig. 6). The patient was instructed on routine surveillance because of the potential for recurrence. In June 2009, approximately, 4 months into a 12-month deployment, the patient complained of pain aggravated by high impact
Fig. 3. Preoperative coronal and lateral magnetic resonance images.
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Fig. 4. Intraoperative photographs of the index procedure showing incision placement, intralesional curettage, back filling with allograft or bone marrow aspirate mixture, and final fixation.
activities and walking on uneven terrain. Radiographs were performed. The findings prompted referral for additional evaluation. Repeat MRI revealed a residual cyst in the posterior calcaneus with multiple fluidfluid levels (Fig. 7). The patient was diagnosed with recurrent chondroblastoma in December 2009, 15 months after the index procedure, and was medically evacuated for definitive treatment. Given recurrence of the lesion, the patient underwent evaluation to rule out metastasis. A whole body scan and contrast-enhanced CT scan of the chest revealed no evidence of metastasis (Fig. 8). The complete blood count with differential revealed a slightly elevated white blood cell count (10.9 109/L) and elevated monocytes (1.2 109/L) and neutrophils (6.7 109/L). The alkaline phosphatase and highsensitivity C-reactive protein levels were normal. The patient underwent repeat aggressive curettage with adjuvant high-speed burring, cryosurgery, and reconstruction with polymethylmethacrylate (PMMA) and steel Steinman pins (Fig. 9). The patient was allowed toe
touch weightbearing for 8 weeks, followed by transition to full weightbearing in a removable walking boot for the next 6 weeks. The patient was instructed to obtain repeat foot and chest radiographs every 3 to 6 months for the next 2 years and then annually to monitor for recurrence. The patient reported no pain, and recurrence was not seen on radiographs in June 2010, 5 months after the second surgery. Because the patient was ending his service with the military, he was instructed to follow-up with an orthopedic oncologist at the closest teaching hospital to his home and was subsequently lost to follow-up. Discussion Chondroblastomas are recognized as an uncommon bone tumor, accounting for <2% of all primary bone tumors (1–12). The condition arises more often in males than females, with a mean age of occurrence of 16 years when the tumor is located in the long bones,
Fig. 5. Histologic slides at (Left) 10 and (Right) 20 magnification, with hematoxylin and eosin staining.
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Fig. 6. Coronal and lateral radiographs and computed tomography images after the index procedure denoting healing, with cystic areas seen at the posteroinferior calcaneus on the computed tomography scan.
compared with a mean age of 28 years when the tumor is located in flat or tubular bones (1–5,7–11,14,15,17,18,21). A review of the published data of the occurrence in the foot revealed a consistent gravitation toward involvement of the calcaneus and talus (7,8,20,21,26–32). Kricun et al (25) reported 14 cases of calcaneal chondroblastoma, with 9 occurring adjacent to the talocalcaneal articulation and 4 occurring in the posterior calcaneus. These
calcaneal locations were also noted by Fink et al (21). This has been postulated to occur secondary to the tumor arising in the apophysis, similar to their development in the epiphysis of the long bones (25). This location is consistent with the present case in which the tumor arose in the inferoposterior aspect of the calcaneus. The most common presenting symptom is insidious onset of localized pain, swelling, and tenderness. Patients might also complain
Fig. 7. Coronal and lateral radiographs and magnetic resonance images showing recurrence of calcaneal chondroblastoma.
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Fig. 8. Images showing whole body scan was performed to rule out metastasis.
of limited range of motion if the tumor is located next to a joint, or it could be asymptomatic and initially acknowledged when radiographs of the foot are obtained (4–6,9,13–18). The characteristic radiographic findings include a round or ovoid osteolytic lesion with a thin sclerotic border that is centrally or eccentrically located near an epiphysis or apophysis. A central or peripheral, radiopaque, stippled or fluffy calcification (i.e., “ground-glass,” fuzzy, or mottled appearance) might be seen (4–6,9,11,13,17,18). Turcotte et al (16) performed a radiographic review of 46 chondroblastomas and reported the following incidence for each of these characteristic findings: 89% for cortical erosion or destruction, 74% for well-defined borders, 52% for an epiphyseal-metaphyseal location, 48% for purely epiphyseal, 37% for a sclerotic rim, 35% for matrix calcifications, 24% for sclerosis of the surrounding bone, and 22% for a periosteal reaction. A peritonitis reaction has been reported to be an uncommon radiographic finding (16,35). However, Brower et al (35) found that peritonitis is a common finding, with a reported 75% incidence in calcaneal chondroblastomas. Previous reports of peritonitis being an uncommon finding were thought to have resulted from peritonitis occurring distant from the lesion. The etiology of peritonitis has been hypothesized to be an inflammatory response to the tumor (35). The tumor size has been reported to be 5 cm (7). Advanced imaging studies have been advocated to assess the tumor further. CT scan has been reported to aid in identifying intraarticular fragments and surgical planning (34). Quint et al (48) reported on the CT evaluation of 3 cases of chondroblastoma and found CT to be superior to plain film radiographs in defining accurate tumor borders, finding cortical destruction, determining the spatial relationships to the adjacent structures, determining the presence of loose bodies and articular involvement, and aiding in distinguishing the lesion from a sarcoma by visualization of the characteristic sclerotic rim and internal calcification. Visualization of this characteristic well-circumscribed lesion with internal calcification and no internal trabeculation or fluid-fluid levels in the apophyseal location of the calcaneus narrows the differential diagnosis, specifically making calcaneal GCT, ABC, and a simple bone cyst less likely (20,49,50).
On MRI, chondroblastomas will appear with a low to intermediate heterogeneous intense signal with a lobular internal structure and fine lobular margins on both T1- and T2-weighted images (49–52). A hypointense peripheral rim of the tumor can be seen on both T1- and T2-weighted images, corresponding to the thin sclerotic margin. On contrast-enhanced imaging, internal lobular and septal enhancement will be seen (52). A periosteal reaction and adjacent bone marrow and soft tissue edema will also enhance owing to the hyperemia and inflammatory reactions to the tumor. All these findings will disappear when the tumor has been removed (50). MRI scanning will further assist in distinguishing chondroblastomas from other neoplasms. The presence of foci with a very low signal within the tumor on T1- and T2-weighted images is consistent with matrix calcification. Areas of low-signal intensity on T1-weighted images and high-signal intensity on T2-weighted images within the tumor are consistent with cartilage within the tumor and might or might not be encircled by a hypointense rim on both T1- and T2-weighted images owing to the surrounding calcification. This hypointense rim is a relatively specific finding for well-differentiated cartilage matrix tumors and will not been seen with eosinophilic granuloma, GCT, or osteoblastoma (49–52). The presence of a surrounding periosteal reaction, joint effusions, and bone marrow and soft tissue edema will decrease the likelihood of enchondroma, chondromyxoid fibroma, and GCT in the differential diagnosis. When an associated ABC is present, multiple cystic areas of increased signal and fluid-fluid levels will be seen on both T1- and T2-weighted images (49–52). Davila et al (8) found that 80% of the time when fluid-fluid levels are present and 40% of the time when surrounding edema is present that an associated ABC was present. The differential diagnosis for chondroblastoma includes ABC, GCT, epiphyseal GCT, unicameral bone cyst, degenerative cyst, chondrosarcoma, osteosarcoma, fibrosarcoma, osteoblastoma, enchondromas, benign epiphyseal enchondroma, tuberculosis, chondromyxoid fibroma, eosinophilic granuloma, nonosteogenic fibroma of the bone, and fibrous dysplasia (4,5,11,13,15,20,33). The diagnosis of some of these tumors will be less likely according to their radiographic and advanced
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Fig. 9. Lateral intraoperative and final postoperative radiographs after the second procedure.
imaging presentation; however, histologic examination is necessary for a definitive diagnosis. Definitive treatment should not proceed until histologic confirmation of the diagnosis has been obtained. Gross anatomic examination will typically reveal a soft, dark-blue to gray-yellow, gray-pink, or gray-brown friable mass. Grayish pink or hemorrhagic soft fragments with alternating gritty yellowish calcific flecks and cystic areas will be seen. The presence of bluish gray nodules will correspond with chondroid tissue (4–6,9,10,17). The microscopic appearance can vary. The common histologic features include a monocellular lesion composed of chondroblasts with a sharply demarcated boarder that are densely packed, uniform, oval, round, or polyhedral, with a polymorphic nucleus that is often indented. Giant cells and small or distinct, large, multinucleated cells can be seen, making differentiation from GCT difficult (1,4–6,10,13,17,18). Chondroblast mitosis will only be occasionally seen (5). The matrix is chondroid in nature and could become calcified, giving chondroblastomas their characteristic lace-like or “chicken-wire” appearance (7). Degenerating cartilage, fibrous tissue, small cysts areas of necrosis, hemorrhage, and vascular channels can be seen. The presence of these blood-filled spaces accompanied by areas of necrosis contributes to the difficulty in differentiating a chondroblastoma from an ABC. Reactive bone and osteoid are infrequent (1,4–6,10,13,17,18). Positive staining with SD-100 and CD68 will aid in the diagnosis of chondroblastoma (53).
Treatment of symptomatic chondroblastoma necessitates surgical intervention (3,22). The surgical techniques have included curettage alone, endoscopic curettage with PMMA or fat implantation, resection with allogeneic bone grafting, marginal resection with radiofrequency ablation, and osteochondral autograft transfer (3,5,6,54). Curettage and bone grafting will be curative in 80% to 90% of cases (17). Local recurrence has been reported to occur at a rate of 6% to 38%, with the greatest incidence seen after simple curettage alone. The use of adjuvant therapies has been reported to minimize this potential (3,9,12,18,32,34,38–42,55). However, adjuvant chemotherapy is not indicated, and adjuvant radiotherapy is contraindicated owing to its association with malignant transformation (11,17,18,22). High-speeding burring, local lavage, and cryosurgery after aggressive curettage are adjuvant techniques that have been reported to minimize the potential for local recurrence. High-speed burring will result in tissue necrosis of 2 mm in depth. Hsu et al (55) reported 3 cases in which aggressive curettage was followed by high-speed burring. They noted no recurrence in all 3 cases at a follow-up point of 62 months (55). Quint et al (56) found that a solution of 5% phenol at room temperature caused coagulation necrosis 1.5 mm in depth, making it an option for adjuvant therapy after curettage. Intralesional alcoholic lavage after aggressive curettage has also been reported to be useful for local control in the treatment of recurrent chondroblastoma with aggressive behavior (57). Schreuder
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et al (58) reported on using cryosurgery after curettage. No tourniquet is used during the procedure. An oval window is created to access the tumor, and aggressive curettage is performed. Thermocouplers are then placed in and around the lesion. Cryosurgery is performed using liquid nitrogen through a cryoprobe. The duration of use is determined by achieving an intralesional temperature of 50 C followed by spontaneous thawing to 20 C for a total of 3 cycles. Lavage with a sodium hyponitrite solution is then used to prevent tumor cell seeding. If temperature monitoring is performed, the surgeon can control the extent of freezing, minimizing the risk of damage to the surrounding tissues (41). Back filling of the tumor cavity with allogeneic or autogenic bone or PMMA has also been found to decrease the potential for local recurrence (16,22,24,28–31,46,55). The addition of bone grafting after curettage has been reported to decrease the incidence of local recurrence compared with phenol irrigation alone (1% versus 21%, respectively) (16). Ramappa et al (46) performed a retrospective study in which 7 of 47 chondroblastomas (w15%) recurred during an average follow-up period of 8.8 6.0 years (range 2.7 to 20) after the index procedure. Owing to the small number of subjects, the stratification of local recurrence secondary to age, gender, tumor size, or follow-up duration, could not be performed. The 1 salient feature noted was that recurrence did not develop in any patient treated with curettage and back filling with PMMA. The heat produced from the process of hardening was hypothesized to destroy any residual tumor cells that might be present (46). Atalar et al (42) reported on 28 patients with chondroblastoma treated with curettage, high-speed burring, and electrocauterization (n ¼ 25; 23 of which were back filled with bone graft and 2 with PMMA), marginal resection (n ¼ 1), en bloc resection with back filling with a fibular allograft (n ¼ 1), en bloc resection with placement of a modular tumor prosthesis (n ¼ 1), en bloc resection with a knee arthrodesis (n ¼ 1), and en bloc resection with hip arthrodesis (n ¼ 1). All 28 patients were followed up for an average of approximately 5 years (range 0.75 to 14). Of these patients, 6 (21.4%) had developed local recurrence at an average of 10 months (range 4 to 24) postoperatively. Four of these recurrences developed after surgery had been performed at another center before the patient was seen by the authors of the study. These recurrences were treated with curettage and bone grafting (n ¼ 4), en bloc resection and autografting (n ¼ 1), and en bloc resection and hip arthrodesis (n ¼ 1). The patients were followed up for an average of 91 months (range 42 to 155). No subsequent recurrence was reported. The results of their study led the investigators to conclude that their preferred method of treatment of chondroblastoma was curettage and bone grafting (42). Long-term follow-up was also recommended because of the period observed between the initial surgical intervention and local recurrence (42). Several other investigators have also recommended long-term follow-up, because the mean reported interval to local recurrence has ranged from 5 months to 8 years (3,16,40,41). Recurrence has primarily been attributed to the tumor location, younger age, the presence of open epiphyseal plates, the presence of an associated ABC, tumor aggressiveness, and inadequate resection. The potential for local recurrence with the first 3 factors has been debated and thought to be more related to inadequate resection secondary to the fear of damaging the adjacent joints, growth plates, and vascular supply to the bone (3,9,32,39,40,42,46,47). The presence of an ABC associated with a chondroblastoma has been reported to occur in 10% to 15% of the cases. Recurrence in such cases has been reported to be dramatically increased, with 1 study reporting a 100% recurrence rate (18,32). This finding has been widely disputed by subsequent studies that found no effect on local recurrence when chondroblastoma was associated with an ABC (3,11,23,59–62). Springfield et al (3) reported only 1 case (2.5%) of
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local recurrence in 50 cases of chondroblastoma with an associated ABC in patients followed up for 2 years after surgical intervention. Otsuka et al (59) reported on a calcaneal chondroblastoma with an associated ABC treated with endoscopic curettage only that had no recurrence at 2 years postoperatively. Guedes et al (62) also reported on a calcaneal chondroblastoma with an associated ABC treated with a total calcanectomy that had no recurrence at 12 months. Sessions et al (60) reported on a cuboidal chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by phenol and saline lavage, with no recurrence at 10 months. Sepah et al (61) also reported a cuboidal chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by bone grafting, with no recurrence at 6 months. Fang and Chen (23) reported on a navicular chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by phenol lavage and allogeneic bone grafting, with no recurrence at 3 years. The weakness of these studies was that they were all case reports, and the follow-up duration can be considered short for determining the true recurrence rate given the extended period during which recurrence can develop (3,16,40–42,46). Lin et al (47) reported on 48 patients with surgically treated chondroblastoma who were followed up for a minimum of 12 months. Local recurrence was found to be dependent on 2 factors, tumor aggressiveness and inadequate resection. Recurrent chondroblastomas appear to have a high growth activity. Testing for the presence of proliferating cell nuclear antigen might be useful in determining the tumor aggressiveness and, thus, whether the potential for recurrence is increased. Inadequate resection has been reported to be the only parameter for local recurrence and has been, by far, the primary reported cause (3,9,32,39,40,42,46,47). The recurrence in the present patient was postulated to be secondary to inadequate resection. The postoperative radiographs after the index procedure and, later, the advanced imaging studies revealed areas of the tumor still present at the most inferoposterior aspect of the calcaneus, with subsequent expansion. Repeated aggressive curettage, high-speed burring, cryosurgery, back filling with PMMA, and reinforcement with Steinman pins to restore the bone stock were used to prevent additional recurrence and the potential for malignant conversion and metastasis. Although the risk of malignant conversion and metastasis is low, it does exist. In the past, the potential for these was found to be increased after adjuvant radiotherapy, making its use in the treatment of chondroblastomas contraindicated (11,17,18,22). Local recurrence is the current primary factor for malignant conversion and metastasis (40,46,47). Rodgers and Mankin (38) reported on 80 patients with chondroblastoma treated surgically and followed up for >25 years. During that period, 6 patients (7.5%) developed local recurrence, 2 (2.5%) progressed to distant metastasis, and 1 (1.3%) died, with the death attributed to malignant conversion. Malignant conversion and metastasis after previous surgical treatment has been postulated to be secondary to the accidental surgical implantation of tumor cells within the lymphatic or vascular system (63–69). Metastasis occurs primarily through the pulmonary system and has been reported to occur 4 months to 33 years after the index surgical procedure (43–45,65–67). The treatment of pulmonary metastasis with chemotherapy, radiotherapy, or surgical excision has been reported to result in survival (43,44). The evaluation of locally recurrent lesions should include the exclusion of metastasis, including a chest radiography, chest CT, bone scintigraphy, and MRI, at a minimum (44,45). Although chondroblastomas are rare, they do occur in the foot, with the calcaneus the fifth most common location of occurrence in the body (7,8,11,18–32). The present case revealed the characteristic findings associated with chondroblastoma: occurrence in a male in
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the second decade of life, located in the region of the calcaneal apophysis, imaging study findings consistent with chondroblastoma, and specific histologic testing that confirmed the diagnosis. The present case had the added difficulty of treating an associated pathologic fracture, which has been reported to be rare in association with chondroblastoma (7,17,18,36). The local recurrence in our patient was most likely secondary to inadequate resection, which has been reported to be the most common cause (3,9,32,39,40,42,46,47). An evaluation to rule out metastasis was performed, just as should be done in all cases of local recurrence. Repeat surgical intervention with curettage, high-speed burring, cryosurgery, back filling of the void with PMMA, and Steinman pins for reinforcement has been reported to minimize the potential for recurrence and were used in our patient (41,46,55,58). The present patient was instructed to follow-up with an orthopedic oncologist at the closest teaching hospital to his home and to obtain repeat foot and chest radiographs every 3 to 6 months for the next 2 years and annually thereafter for lifelong monitoring because of the potential for malignant conversion and metastasis. References 1. Smith DA, Graham WC, Smith FR. Benign chondroblastoma of bone: report of an unusual case. J Bone Joint Surg Am 44-A:571–577, 1962. 2. Hull MT, Gonzalez–Crussi F, DeRosa GP, Graul RS. Aggressive chondroblastoma: report of a case with multiple bone and soft tissue involvement. Clin Orthop Relat Res 126:261–265, 1977. 3. Springfield DS, Capanna R, Gherlinzoni F, Picci P, Campanacci M. Chondroblastoma: a review of seventy cases. J Bone Joint Surg Am 67:748–755, 1985. 4. Sundaram TK. Benign chondroblastoma. J Bone Joint Surg Br 48:92–104, 1966. 5. McBryde A Jr, Goldner JL. Chondroblastoma of bone. Am Surg 36:94–108, 1970. 6. Kunkel MG, Dahlin DC, Young HH. Benign chondroblastoma. J Bone Joint Surg Am 38-A:817–826, 1956. 7. Kurt AM, Unni KK, Sim FH, McLeod RA. Chondroblastoma of bone. Hum Pathol 20:965–976, 1989. 8. Davila JA, Amrami KK, Sundaram M, Adkins MC, Unni KK. Chondroblastoma of the hands and feet. Skeletal Radiol 33:582–587, 2004. 9. Schajowicz F, Gallardo H. Epiphyseal chondroblastoma of bone: a clinicopathological study of sixty-nine cases. J Bone Joint Surg Br 52:205–226, 1970. 10. Raina V. Benign chondroblastomada clinico-pathological study of ten cases. Indian J Cancer 16:22–25, 1979. 11. Bloem JL, Mulder JD. Chondroblastoma: a clinical and radiological study of 104 cases. Skeletal Radiol 14:1–9, 1985. 12. Iwai T, Abe S, Miki Y, Tokizaki T, Matsuda K, Wakimoto N, Nakamura S, Imamura T, Matsushita T. A trapdoor procedure for chondroblastoma of the femoral head: a case report. Arch Orthop Trauma Surg 128:763–767, 2008. 13. Valls J, Ottolenghi CE, Schajowicz F. Epiphyseal chondroblastoma of bone. J Bone Joint Surg Am 33-A:997–1009, 1951. 14. Treasure ER. Benign chondroblastoma of bone: report of a case. J Bone Joint Surg Br 37-B:462–465, 1955. 15. Sherman RS, Uzel AR. Benign chondroblastoma of bone: its roentgen diagnosis. Am J Roentgenol Radium Ther Nucl Med 76:1132–1140, 1956. 16. Turcotte RE, Kurt AM, Sim FH, Unni KK, McLeod RA. Chondroblastoma. Hum Pathol 24:944–949, 1993. 17. Dahlin DC, Ivins JC. Benign chondroblastoma: a study of 125 cases. Cancer 30:401– 413, 1972. 18. Huvos AG, Marcove RC. Chondroblastoma of bone: a critical review. Clin Orthop Relat Res 95:300–312, 1973. 19. Katz HB, Hunt DW. Chondroblastoma of the calcaneus: a case report. J Am Podiatry Assoc 65:184–187, 1975. 20. Kahmann R, Gold RH, Eckardt JJ, Mirra JM. Case report 337: cystic chondroblastoma of calcaneus. Skeletal Radiol 14:301–304, 1985. 21. Fink BR, Temple HT, Chiricosta FM, Mizel MS, Murphey MD. Chondroblastoma of the foot. Foot Ankle Int 18:236–242, 1997. 22. Dhatt SS, Bhagwat KR, Kumar V, Dhillon MS. Chondroblastoma in a metatarsal treated with autogenous fibular graft: a case report. J Foot Ankle Surg 51:356–361, 2012. 23. Fang Z, Chen M. Chondroblastoma associated with aneurysmal cyst of the navicular bone: a case report. World J Surg Oncol 11:50, 2013. ral G, Yıldırım A, Kandemir O. Chondroblastoma of the 24. Arıkan M, Aktas E, Tog medial cuneiform bone in a 32-year-old woman. Am J Case Rep 28:317–321, 2014. 25. Kricun ME, Kricun R, Haskin ME. Chondroblastoma of the calcaneus: radiographic features with emphasis on location. AJR Am J Roentgenol 128:613–616, 1977. 26. Barbera C, Pinotti N, Klein MJ, Lewis MM. An unusual case of cystic chondroblastoma of the calcaneus: a case report. Bull Hosp Jt Dis Orthop Inst 48:88–92, 1988.
27. Ochsner PE, von Hochstetter AR, Hilfiker B. Chondroblastoma of the talus: natural development over 9.5 years. Case report. Arch Orthop Trauma Surg 107:122–125, 1988. 28. Blitch E, Mendicino RW. Chondroblastoma of the calcaneus: literature review and case presentation. J Foot Ankle Surg 35:250–254, 1996. 29. Yu GV, Sellers CS. Chondroblastoma of the talus. J Foot Ankle Surg 35:72–77, 1996. 30. Sterling G, Wilson A. Chondroblastoma of the talus: a case report. J Foot Ankle Surg 41:178–182, 2002. 31. Anderson AF, Ramsey JR. Chondroblastoma of the talus treated with osteochondral autograft transfer from the lateral femoral condyle. Foot Ankle Int 24:283–287, 2003. 32. Ningegowda RV, Subramanian K, Suresh I. Chondroblastoma of the talus. J Foot Ankle Surg 52:673–677, 2013. 33. Plum GE, Pugh DG. Roentgenologic aspects of benign chondroblastoma of bone. Am J Roentgenol Radium Ther Nucl Med 79:584–591, 1958. 34. Hudson TM, Hawkins IF Jr. Radiological evaluation of chondroblastoma. Radiology 139:1–10, 1981. 35. Brower AC, Moser RP, Kransdorf MJ. The frequency and diagnostic significance of periostitis in chondroblastoma. AJR Am J Roentgenol 154:309–314, 1990. 36. Prohaska DJ, Kneidel TW. Chondroblastoma in a metatarsal. J Foot Ankle Surg 37:63–65, 1998. 37. Ly JQ, LaGatta LM, Beall DP. Calcaneal chondroblastoma with secondary aneurysmal bone cyst. AJR Am J Roentgenol 182:130, 2004. 38. Rodgers WB, Mankin HJ. Metastatic malignant chondroblastoma. Am J Orthop (Belle Mead NJ) 25:846–849, 1996. 39. de Silva MV, Reid R. Chondroblastoma: varied histologic appearance, potential diagnostic pitfalls, and clinicopathologic features associated with local recurrence. Ann Diagn Pathol 7:205–213, 2003. 40. Suneja R, Grimer RJ, Belthur M, Jeys L, Carter SR, Tillman RM, Davies AM. Chondroblastoma of bone: long-term results and functional outcome after intralesional curettage. J Bone Joint Surg Br 87:974–978, 2005. 41. van der Geest IC, van Noort MP, Schreuder HW, Pruszczynski M, de Rooy JW, Veth RP. The cryosurgical treatment of chondroblastoma of bone: long-term oncologic and functional results. J Surg Oncol 96:230–234, 2007. 42. Atalar H, Basarir K, Yildiz Y, Erekul S, Saglik Y. Management of chondroblastoma: retrospective review of 28 patients. J Orthop Sci 12:334–340, 2007. 43. Kyriakos M, Land VJ, Penning HL, Parker SG. Metastatic chondroblastoma: report of a fatal case with a review of the literature on atypical, aggressive, and malignant chondroblastoma. Cancer 55:1770–1789, 1985. 44. Elek EM, Grimer RJ, Mangham DC, Davies AM, Carter SR, Tillman RM. Malignant chondroblastoma of the os calcis. Sarcoma 2:45–48, 1998. 45. Jambhekar NA, Desai PB, Chitale DA, Patil P, Arya S. Benign metastasizing chondroblastoma: a case report. Cancer 82:675–678, 1998. 46. Ramappa AJ, Lee FY, Tang P, Carlson JR, Gebhardt MC, Mankin HJ. Chondroblastoma of bone. J Bone Joint Surg Am 82-A:1140–1145, 2000. 47. Lin PP, Thenappan A, Deavers MT, Lewis VO, Yasko AW. Treatment and prognosis of chondroblastoma. Clin Orthop Relat Res 438:103–109, 2005. 48. Quint LE, Gross BH, Glazer GM, Braunstein EM, White SJ. CT evaluation of chondroblastoma. J Comput Assist Tomogr 8:907–910, 1984. 49. Weatherall PT, Maale GE, Mendelsohn DB, Sherry CS, Erdman WE, Pascoe HR. Chondroblastoma: classic and confusing appearance at MR imaging. Radiology 190:467–474, 1994. 50. Azouz EM. Magnetic resonance imaging of benign bone lesions: cysts and tumors. Top Magn Reson Imaging 13:219–229, 2002. 51. Oxtoby JW, Davies AM. MRI characteristics of chondroblastoma. Clin Radiol 51:22– 26, 1996. 52. Jee WH, Park YK, McCauley TR, Choi KH, Ryu KN, Suh JS, Suh KJ, Cho JH, Lee JH, Park JM, Lee YS, Ok IY, Kim JM. Chondroblastoma: MR characteristics with pathologic correlation. J Comput Assist Tomogr 23:721–726, 1999. 53. Zhang K, Gao Y, Dai H, Zhang S, Li G, Yu B. Chondroblastoma of the talus: a case report and literature review. J Foot Ankle Surg 51:262–265, 2012. 54. Erickson JK, Rosenthal DI, Zaleske DJ, Gebhardt MC, Cates JM. Primary treatment of chondroblastoma with percutaneous radio-frequency heat ablation: report of three cases. Radiology 221:463–468, 2001. 55. Hsu CC, Wang JW, Chen CE, Lin JW. Results of curettage and high-speed burring for chondroblastoma of the bone. Chang Gung Med J 26:761–767, 2003. € ller RT, Mu € ller G. Characteristics of phenol: instillation in intralesional 56. Quint U, Mu tumor excision of chondroblastoma, osteoclastoma and enchondroma. Arch Orthop Trauma Surg 117:43–46, 1998. 57. Masui F, Ushigome S, Kamitani K, Asanuma K, Fujii K. Chondroblastoma: a study of 11 cases. Eur J Surg Oncol 28:869–874, 2002. 58. Schreuder HW, Pruszczynski M, Veth RP, Lemmens JA. Treatment of benign and low-grade malignant intramedullary chondroid tumours with curettage and cryosurgery. Eur J Surg Oncol 24:120–126, 1998. 59. Otsuka T, Kobayashi M, Yonezawa M, Kamiyama F, Matsushita Y, Matsui N. Treatment of chondroblastoma of the calcaneus with a secondary aneurysmal bone cyst using endoscopic curettage without bone grafting. Arthroscopy 18:430–435, 2002. 60. Sessions W, Siegel HJ, Thomas J, Pitt M, Said-Al-Naief N, Casillas MA. Chondroblastoma with associated aneurysmal bone cyst of the cuboid. J Foot Ankle Surg 44:64–67, 2005. 61. Sepah YJ, Umer M, Minhas K, Hafeez K. Chondroblastoma of the cuboid with an associated aneurysmal bone cyst: a case report. J Med Case Rep 14:135, 2007.
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Calcaneal Chondroblastoma with Pathologic Fracture and Recurrence Laksha Dutt, DPM 1, Valerie L. Schade, DPM, FACFAS 2, Mark W. Manoso, MD 3 1
Postgraduate Year 2 Resident, Podiatric Medicine Surgical Residency, Madigan Army Medical Center, Tacoma, WA Chief, Limb Preservation Service, Madigan Army Medical Center, Tacoma, WA 3 Chief, Orthopedic Surgery Service, Madigan Army Medical Center, Tacoma, WA 2
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4
Chondroblastomas account for <2% of all bone tumors. The calcaneus is the fifth most common location of occurrence. Males in their second decade of life are most often affected, presenting with an insidious onset of localized pain, swelling, and tenderness. The finding of associated pathologic fracture has been rare. Imaging studies can aid in the formulation of the differential diagnosis and surgical plan. The definitive diagnosis requires histologic examination. Curettage and bone grafting is curative in >80% of cases. Local recurrence rates of 38% have been reported, most often because of inadequate resection, and have been associated with malignant conversion and metastasis. Adjuvant therapies can help minimize the incidence of local recurrence. Long-term follow-up examinations are recommended, given the protracted interval that can exist between recurrence and the potential for malignant conversion and metastasis. We present the case of a young, healthy, active male with a calcaneal chondroblastoma and associated pathologic fracture whose initial treatment consisted of curettage, hydrogen peroxide lavage, and allogeneic bone grafting. Recurrence developed at 15 months postoperatively and was treated with repeat curettage, high-speed burring, and reconstruction with steel Steinman pins and polymethylmethacrylate, resulting in no pain or recurrence at the 5-month follow-up point. Ó 2015 by the American College of Foot and Ankle Surgeons. All rights reserved.
Keywords: cartilaginous cryosurgery high-speed burring
Chondroblastoma is as a rare, benign, cartilaginous bone tumor derived from epiphyseal chondrocytes, accounting for 1% to 2% of all bone tumors (1–12). Kolodny first described the tumor in 1927 as a variant of a giant cell tumor (GCT) (4,5,13). Ewing described the tumor as a calcifying GCT in 1928, followed by Codman in 1931, who reported that the tumor only occurred in the humerus (2–8,13–16). Jaffe and Lichtenstein differentiated it from a GCT variant in 1942, coining the term “benign chondroblastoma,” and determined that the neoplasm arose in bones other than the humerus (2–8,13,14,16). Geschicket and Copeland later determined that both benign and malignant forms exist (5,13). Chondroblastomas typically present in males in the second decade of life, with a reported male/female ratio ranging from 2:1 to 5:1 (1–5,7–10,14,15,17,18). The area of occurrence has most frequently been in the epiphysis of the long bones, in particular, the femur, Financial Disclosure: None reported. Conflict of Interest: None reported. Disclaimer: The views expressed are those of the authors and do not reflect the official policy of the Department of the Army, the Department of Defense, or the US Government. Address correspondence to: Valerie L. Schade, DPM, FACFAS, Limb Preservation Service, Madigan Army Medical Center, 9040 Jackson Drive, MCHJ-CLS-V, Tacoma, WA 98431. E-mail address: [email protected] (V.L. Schade).
humerus, and proximal tibia, but it can also occur in the small bones of the hands and feet, which undergo endochondral ossification (1,2,5,7,9–11,13,17,18). The incidence in the foot has ranged from 3% to 23%, making it the second most common location of occurrence, with reports of occurrence in the navicular, medial cuneiform, cuboid, metatarsals, and phalanges (7,8,11,18–24). However, the tarsal bones most often affected are the talus and calcaneus, with a reported occurrence rate of 45% and 7% to 34%, respectively (7,8,20,21,25–32). The calcaneus has been reported to be the fifth most common location of occurrence (21). The clinical presentation most often involves the insidious onset of localized pain, soft tissue swelling, and tenderness (4–6,9,13–18). Radiographs will reveal an osteolytic lesion with a slow-growing appearance (3–9,11,16–18,33–35). Pathologic fracture is rare (7,17,18,36). Advanced imaging studies aid in formulating the differential diagnosis and surgical treatment plan (34). A histologic examination is essential to determine the definitive diagnosis (14,37). Surgical treatment ranges from simple curettage to en bloc resection (3,6,17). Although these procedures will most often be effective, recurrence has been reported to range from 6% to 38% and has been reported to occur more frequently in the tubular bones than in the long bones (29% versus 11%, respectively) (3,9,12,17,28,32,34,38–41). Curettage and adjuvant therapies have been reported to result in resolution in cases of recurrence (3,16,42). Malignant conversion and metastasis have been associated with local recurrence (38,40,43–47).
1067-2516/$ - see front matter Ó 2015 by the American College of Foot and Ankle Surgeons. All rights reserved. http://dx.doi.org/10.1053/j.jfas.2014.12.006
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was noted. Mild soft tissue edema at the medial subtalar joint and mild bone marrow edema within the calcaneus from the fracture were seen (Fig. 3). The radiographic work-up revealed no other primary malignancy or metastatic spread. The computed tomography (CT) scan revealed a well-defined osteolytic lesion abutting the posterior inferior aspect of the calcaneus, with no associated periosteal reaction. Cortical discontinuity with minimal diastasis was noted at the fracture site and involved the posterior facet of the subtalar joint (Fig. 2). Operative Technique Fig. 1. Index anteroposterior and calcaneal radiographs of the left foot.
Long-term monitoring is recommended for these reasons (43–45). We present a case of a calcaneal chondroblastoma with an associated pathologic fracture that occurred in a young, healthy, active-duty male soldier. Recurrence developed at 15 months postoperatively. The findings from our case signify the importance of an adequate initial resection and consideration of adjuvant techniques, the need to rule out metastasis in patients with local recurrence before surgical intervention, and the need for long-term monitoring.
Case Report A 21-year-old active-duty male soldier presented to a primary care provider in December 2007 with an 8-month history of left heel pain that began after he had jumped down an approximately 10 500 height from a vehicle while deployed. The patient had initially been diagnosed with Achilles tendinitis and prescribed a nonsteroidal antiinflammatory drug. He was also instructed to limit his activities; to use ice, to use an elastic bandage (for compression), to elevate, and to perform range of motion exercises. No radiographs were taken. In June 2008, the patient was referred for physical therapy because of continuing pain. The initial radiographs were taken in August 2008, revealing an osteolytic lesion in the posterior inferior calcaneus (Fig. 1). The patient was instructed to obtain a magnetic resonance imaging (MRI) study and was referred to the podiatry department. En route to obtaining the MRI scan, the patient stepped off a curb and noted immediate sharp pain and an inability to bear weight on his left foot. The patient proceeded to the emergency department for additional evaluation. Repeat radiographs revealed a pathologic fracture of the left calcaneus, with possible extension into the posterior facet of the subtalar joint (Fig. 2). The patient reported no history of fever, chills, or unexplained weight loss. The patient had no medical or surgical history and denied the use of any medication, apart from the previously prescribed nonsteroidal anti-inflammatory drug. The patient reported smoking 5 cigarettes daily for the previous 2 years. The patient related an allergy to sulfa medications and denied a family history of cancer. Focused examination of the lower extremity revealed the left foot to have an intact neurovascular status and pitting edema localized to the hind foot. Mondor’s sign and ecchymosis were present along the lateral heel rim. No local erythema or regional lymphadenopathy was appreciated. His muscle strength and range of motion of the ankle and subtalar joint were normal, apart from some guarding secondary to pain. The results from a complete blood count, basic metabolic panel, and serum albumin plus prealbumin, C-reactive protein, erythrocyte sedimentation rate, and alkaline phosphatase tests were all within normal limits. The MRI scan revealed a 3.9-cm 4.7-cm 3.2-cm solid lesion with no definite areas of calcification or cystic portions. A fracture running longitudinally, adjacent to the cyst, with involvement of the lateral calcaneal cortex and the posterior facet of the subtalar joint,
The operative technique is shown in Fig. 4. In September 2008, the patient was taken for his initial surgical procedure. The patient underwent general anesthesia and was placed in a right lateral decubitus position to allow access to the lateral aspect of the right foot. A thigh tourniquet was used. A 4-cm linear incision was placed over the lateral aspect of the calcaneus, angled from posterosuperiorly to anteroinferiorly, starting halfway between the Achilles tendon and the posterior aspect of the lateral malleolus and extending to the lateral bisection of the calcaneal tuberosity. Sharp and blunt dissection were performed to expose the calcaneus. A cortical window was created to expose the cavity of the tumor. The contents were thin, friable, and hemorrhagic. A portion of the lining and aspirated contents of the cavity underwent fresh frozen analysis, with no occult malignancy seen. Aggressive curettage was performed to remove the remaining contents and lining. The osseous void was then lavaged with copious amounts of hydrogen peroxide, followed by normal saline infused with bacitracin. Intraoperative cultures were obtained. The osseous void was filled with allogeneic bone graft mixed with bone marrow aspirate obtained from the proximal tibia to fill the void and restore the bone stock. A 4-hole locking plate was placed for fracture fixation and stabilization of the cortical window that had been created. Intraoperative Culture Results and Tissue Specimen Examination The intraoperative culture results were negative for aerobic and anaerobic growth (Fig. 5). The initial histologic examination revealed proliferation of multinucleated giant cells with cellular spindled stroma. Woven bony trabeculae were present, some with and some without osteoblastic rimming. New bone formation was evident, as well as areas of necrosis. Mitotic activity was rare. Although associated hemorrhage was present, the salient features of an aneurysmal bone cyst (ABC) were not. The differential diagnosis was GCT with an associated fracture callus, giant cell reparative granuloma, and other neoplasm. The specimen was sent to the Department of Orthopedic Pathology of the Armed Forces Institute of Pathology for additional evaluation. The final histologic examination revealed small mononuclear cells with multinucleated giant cells admixed. A suggestion of focal eosinophilic cartilage matrix production and extensive areas of infarction and necrosis was found. Focal staining of the tumor cells with S-100 was positive and focal staining with an aldehyde fuchsin stain at pH 1.0 was equivocal. Sulfated matrix production in a delicate “chicken-wire” distribution was seen around the individual tumor cells. The diagnosis was chondroblastoma. Postoperative Course The patient was immobilized in a short leg cast and instructed to be non-weightbearing for 8 weeks. The patient reported falling soon after the first postoperative visit and missed the second postoperative visit, and he had continued smoking. This resulted in superficial dehiscence of the incision site, which ultimately healed with nonoperative wound care and a 20-day course of oral antibiotics. A CT
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Fig. 2. Preoperative lateral radiographs and coronal and lateral computed tomography views after the patient had sustained the pathologic fracture.
scan obtained in November 2008 revealed incorporation of the allogeneic bone graft, stable hardware, and no subtalar joint arthrosis. At 3 months postoperatively, the patient was allowed to gradually progress back to normal shoe gear and full activity. At 5 months postoperatively, the patient reported complete resolution of the pain, had returned to full activity, and was preparing for an upcoming
deployment. Radiographs at that point revealed some persistent cystic changes at the most inferior and medial aspect of the calcaneus (Fig. 6). The patient was instructed on routine surveillance because of the potential for recurrence. In June 2009, approximately, 4 months into a 12-month deployment, the patient complained of pain aggravated by high impact
Fig. 3. Preoperative coronal and lateral magnetic resonance images.
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Fig. 4. Intraoperative photographs of the index procedure showing incision placement, intralesional curettage, back filling with allograft or bone marrow aspirate mixture, and final fixation.
activities and walking on uneven terrain. Radiographs were performed. The findings prompted referral for additional evaluation. Repeat MRI revealed a residual cyst in the posterior calcaneus with multiple fluidfluid levels (Fig. 7). The patient was diagnosed with recurrent chondroblastoma in December 2009, 15 months after the index procedure, and was medically evacuated for definitive treatment. Given recurrence of the lesion, the patient underwent evaluation to rule out metastasis. A whole body scan and contrast-enhanced CT scan of the chest revealed no evidence of metastasis (Fig. 8). The complete blood count with differential revealed a slightly elevated white blood cell count (10.9 109/L) and elevated monocytes (1.2 109/L) and neutrophils (6.7 109/L). The alkaline phosphatase and highsensitivity C-reactive protein levels were normal. The patient underwent repeat aggressive curettage with adjuvant high-speed burring, cryosurgery, and reconstruction with polymethylmethacrylate (PMMA) and steel Steinman pins (Fig. 9). The patient was allowed toe
touch weightbearing for 8 weeks, followed by transition to full weightbearing in a removable walking boot for the next 6 weeks. The patient was instructed to obtain repeat foot and chest radiographs every 3 to 6 months for the next 2 years and then annually to monitor for recurrence. The patient reported no pain, and recurrence was not seen on radiographs in June 2010, 5 months after the second surgery. Because the patient was ending his service with the military, he was instructed to follow-up with an orthopedic oncologist at the closest teaching hospital to his home and was subsequently lost to follow-up. Discussion Chondroblastomas are recognized as an uncommon bone tumor, accounting for <2% of all primary bone tumors (1–12). The condition arises more often in males than females, with a mean age of occurrence of 16 years when the tumor is located in the long bones,
Fig. 5. Histologic slides at (Left) 10 and (Right) 20 magnification, with hematoxylin and eosin staining.
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Fig. 6. Coronal and lateral radiographs and computed tomography images after the index procedure denoting healing, with cystic areas seen at the posteroinferior calcaneus on the computed tomography scan.
compared with a mean age of 28 years when the tumor is located in flat or tubular bones (1–5,7–11,14,15,17,18,21). A review of the published data of the occurrence in the foot revealed a consistent gravitation toward involvement of the calcaneus and talus (7,8,20,21,26–32). Kricun et al (25) reported 14 cases of calcaneal chondroblastoma, with 9 occurring adjacent to the talocalcaneal articulation and 4 occurring in the posterior calcaneus. These
calcaneal locations were also noted by Fink et al (21). This has been postulated to occur secondary to the tumor arising in the apophysis, similar to their development in the epiphysis of the long bones (25). This location is consistent with the present case in which the tumor arose in the inferoposterior aspect of the calcaneus. The most common presenting symptom is insidious onset of localized pain, swelling, and tenderness. Patients might also complain
Fig. 7. Coronal and lateral radiographs and magnetic resonance images showing recurrence of calcaneal chondroblastoma.
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Fig. 8. Images showing whole body scan was performed to rule out metastasis.
of limited range of motion if the tumor is located next to a joint, or it could be asymptomatic and initially acknowledged when radiographs of the foot are obtained (4–6,9,13–18). The characteristic radiographic findings include a round or ovoid osteolytic lesion with a thin sclerotic border that is centrally or eccentrically located near an epiphysis or apophysis. A central or peripheral, radiopaque, stippled or fluffy calcification (i.e., “ground-glass,” fuzzy, or mottled appearance) might be seen (4–6,9,11,13,17,18). Turcotte et al (16) performed a radiographic review of 46 chondroblastomas and reported the following incidence for each of these characteristic findings: 89% for cortical erosion or destruction, 74% for well-defined borders, 52% for an epiphyseal-metaphyseal location, 48% for purely epiphyseal, 37% for a sclerotic rim, 35% for matrix calcifications, 24% for sclerosis of the surrounding bone, and 22% for a periosteal reaction. A peritonitis reaction has been reported to be an uncommon radiographic finding (16,35). However, Brower et al (35) found that peritonitis is a common finding, with a reported 75% incidence in calcaneal chondroblastomas. Previous reports of peritonitis being an uncommon finding were thought to have resulted from peritonitis occurring distant from the lesion. The etiology of peritonitis has been hypothesized to be an inflammatory response to the tumor (35). The tumor size has been reported to be 5 cm (7). Advanced imaging studies have been advocated to assess the tumor further. CT scan has been reported to aid in identifying intraarticular fragments and surgical planning (34). Quint et al (48) reported on the CT evaluation of 3 cases of chondroblastoma and found CT to be superior to plain film radiographs in defining accurate tumor borders, finding cortical destruction, determining the spatial relationships to the adjacent structures, determining the presence of loose bodies and articular involvement, and aiding in distinguishing the lesion from a sarcoma by visualization of the characteristic sclerotic rim and internal calcification. Visualization of this characteristic well-circumscribed lesion with internal calcification and no internal trabeculation or fluid-fluid levels in the apophyseal location of the calcaneus narrows the differential diagnosis, specifically making calcaneal GCT, ABC, and a simple bone cyst less likely (20,49,50).
On MRI, chondroblastomas will appear with a low to intermediate heterogeneous intense signal with a lobular internal structure and fine lobular margins on both T1- and T2-weighted images (49–52). A hypointense peripheral rim of the tumor can be seen on both T1- and T2-weighted images, corresponding to the thin sclerotic margin. On contrast-enhanced imaging, internal lobular and septal enhancement will be seen (52). A periosteal reaction and adjacent bone marrow and soft tissue edema will also enhance owing to the hyperemia and inflammatory reactions to the tumor. All these findings will disappear when the tumor has been removed (50). MRI scanning will further assist in distinguishing chondroblastomas from other neoplasms. The presence of foci with a very low signal within the tumor on T1- and T2-weighted images is consistent with matrix calcification. Areas of low-signal intensity on T1-weighted images and high-signal intensity on T2-weighted images within the tumor are consistent with cartilage within the tumor and might or might not be encircled by a hypointense rim on both T1- and T2-weighted images owing to the surrounding calcification. This hypointense rim is a relatively specific finding for well-differentiated cartilage matrix tumors and will not been seen with eosinophilic granuloma, GCT, or osteoblastoma (49–52). The presence of a surrounding periosteal reaction, joint effusions, and bone marrow and soft tissue edema will decrease the likelihood of enchondroma, chondromyxoid fibroma, and GCT in the differential diagnosis. When an associated ABC is present, multiple cystic areas of increased signal and fluid-fluid levels will be seen on both T1- and T2-weighted images (49–52). Davila et al (8) found that 80% of the time when fluid-fluid levels are present and 40% of the time when surrounding edema is present that an associated ABC was present. The differential diagnosis for chondroblastoma includes ABC, GCT, epiphyseal GCT, unicameral bone cyst, degenerative cyst, chondrosarcoma, osteosarcoma, fibrosarcoma, osteoblastoma, enchondromas, benign epiphyseal enchondroma, tuberculosis, chondromyxoid fibroma, eosinophilic granuloma, nonosteogenic fibroma of the bone, and fibrous dysplasia (4,5,11,13,15,20,33). The diagnosis of some of these tumors will be less likely according to their radiographic and advanced
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Fig. 9. Lateral intraoperative and final postoperative radiographs after the second procedure.
imaging presentation; however, histologic examination is necessary for a definitive diagnosis. Definitive treatment should not proceed until histologic confirmation of the diagnosis has been obtained. Gross anatomic examination will typically reveal a soft, dark-blue to gray-yellow, gray-pink, or gray-brown friable mass. Grayish pink or hemorrhagic soft fragments with alternating gritty yellowish calcific flecks and cystic areas will be seen. The presence of bluish gray nodules will correspond with chondroid tissue (4–6,9,10,17). The microscopic appearance can vary. The common histologic features include a monocellular lesion composed of chondroblasts with a sharply demarcated boarder that are densely packed, uniform, oval, round, or polyhedral, with a polymorphic nucleus that is often indented. Giant cells and small or distinct, large, multinucleated cells can be seen, making differentiation from GCT difficult (1,4–6,10,13,17,18). Chondroblast mitosis will only be occasionally seen (5). The matrix is chondroid in nature and could become calcified, giving chondroblastomas their characteristic lace-like or “chicken-wire” appearance (7). Degenerating cartilage, fibrous tissue, small cysts areas of necrosis, hemorrhage, and vascular channels can be seen. The presence of these blood-filled spaces accompanied by areas of necrosis contributes to the difficulty in differentiating a chondroblastoma from an ABC. Reactive bone and osteoid are infrequent (1,4–6,10,13,17,18). Positive staining with SD-100 and CD68 will aid in the diagnosis of chondroblastoma (53).
Treatment of symptomatic chondroblastoma necessitates surgical intervention (3,22). The surgical techniques have included curettage alone, endoscopic curettage with PMMA or fat implantation, resection with allogeneic bone grafting, marginal resection with radiofrequency ablation, and osteochondral autograft transfer (3,5,6,54). Curettage and bone grafting will be curative in 80% to 90% of cases (17). Local recurrence has been reported to occur at a rate of 6% to 38%, with the greatest incidence seen after simple curettage alone. The use of adjuvant therapies has been reported to minimize this potential (3,9,12,18,32,34,38–42,55). However, adjuvant chemotherapy is not indicated, and adjuvant radiotherapy is contraindicated owing to its association with malignant transformation (11,17,18,22). High-speeding burring, local lavage, and cryosurgery after aggressive curettage are adjuvant techniques that have been reported to minimize the potential for local recurrence. High-speed burring will result in tissue necrosis of 2 mm in depth. Hsu et al (55) reported 3 cases in which aggressive curettage was followed by high-speed burring. They noted no recurrence in all 3 cases at a follow-up point of 62 months (55). Quint et al (56) found that a solution of 5% phenol at room temperature caused coagulation necrosis 1.5 mm in depth, making it an option for adjuvant therapy after curettage. Intralesional alcoholic lavage after aggressive curettage has also been reported to be useful for local control in the treatment of recurrent chondroblastoma with aggressive behavior (57). Schreuder
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et al (58) reported on using cryosurgery after curettage. No tourniquet is used during the procedure. An oval window is created to access the tumor, and aggressive curettage is performed. Thermocouplers are then placed in and around the lesion. Cryosurgery is performed using liquid nitrogen through a cryoprobe. The duration of use is determined by achieving an intralesional temperature of 50 C followed by spontaneous thawing to 20 C for a total of 3 cycles. Lavage with a sodium hyponitrite solution is then used to prevent tumor cell seeding. If temperature monitoring is performed, the surgeon can control the extent of freezing, minimizing the risk of damage to the surrounding tissues (41). Back filling of the tumor cavity with allogeneic or autogenic bone or PMMA has also been found to decrease the potential for local recurrence (16,22,24,28–31,46,55). The addition of bone grafting after curettage has been reported to decrease the incidence of local recurrence compared with phenol irrigation alone (1% versus 21%, respectively) (16). Ramappa et al (46) performed a retrospective study in which 7 of 47 chondroblastomas (w15%) recurred during an average follow-up period of 8.8 6.0 years (range 2.7 to 20) after the index procedure. Owing to the small number of subjects, the stratification of local recurrence secondary to age, gender, tumor size, or follow-up duration, could not be performed. The 1 salient feature noted was that recurrence did not develop in any patient treated with curettage and back filling with PMMA. The heat produced from the process of hardening was hypothesized to destroy any residual tumor cells that might be present (46). Atalar et al (42) reported on 28 patients with chondroblastoma treated with curettage, high-speed burring, and electrocauterization (n ¼ 25; 23 of which were back filled with bone graft and 2 with PMMA), marginal resection (n ¼ 1), en bloc resection with back filling with a fibular allograft (n ¼ 1), en bloc resection with placement of a modular tumor prosthesis (n ¼ 1), en bloc resection with a knee arthrodesis (n ¼ 1), and en bloc resection with hip arthrodesis (n ¼ 1). All 28 patients were followed up for an average of approximately 5 years (range 0.75 to 14). Of these patients, 6 (21.4%) had developed local recurrence at an average of 10 months (range 4 to 24) postoperatively. Four of these recurrences developed after surgery had been performed at another center before the patient was seen by the authors of the study. These recurrences were treated with curettage and bone grafting (n ¼ 4), en bloc resection and autografting (n ¼ 1), and en bloc resection and hip arthrodesis (n ¼ 1). The patients were followed up for an average of 91 months (range 42 to 155). No subsequent recurrence was reported. The results of their study led the investigators to conclude that their preferred method of treatment of chondroblastoma was curettage and bone grafting (42). Long-term follow-up was also recommended because of the period observed between the initial surgical intervention and local recurrence (42). Several other investigators have also recommended long-term follow-up, because the mean reported interval to local recurrence has ranged from 5 months to 8 years (3,16,40,41). Recurrence has primarily been attributed to the tumor location, younger age, the presence of open epiphyseal plates, the presence of an associated ABC, tumor aggressiveness, and inadequate resection. The potential for local recurrence with the first 3 factors has been debated and thought to be more related to inadequate resection secondary to the fear of damaging the adjacent joints, growth plates, and vascular supply to the bone (3,9,32,39,40,42,46,47). The presence of an ABC associated with a chondroblastoma has been reported to occur in 10% to 15% of the cases. Recurrence in such cases has been reported to be dramatically increased, with 1 study reporting a 100% recurrence rate (18,32). This finding has been widely disputed by subsequent studies that found no effect on local recurrence when chondroblastoma was associated with an ABC (3,11,23,59–62). Springfield et al (3) reported only 1 case (2.5%) of
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local recurrence in 50 cases of chondroblastoma with an associated ABC in patients followed up for 2 years after surgical intervention. Otsuka et al (59) reported on a calcaneal chondroblastoma with an associated ABC treated with endoscopic curettage only that had no recurrence at 2 years postoperatively. Guedes et al (62) also reported on a calcaneal chondroblastoma with an associated ABC treated with a total calcanectomy that had no recurrence at 12 months. Sessions et al (60) reported on a cuboidal chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by phenol and saline lavage, with no recurrence at 10 months. Sepah et al (61) also reported a cuboidal chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by bone grafting, with no recurrence at 6 months. Fang and Chen (23) reported on a navicular chondroblastoma with an associated ABC treated with aggressive curettage and high-speed burring followed by phenol lavage and allogeneic bone grafting, with no recurrence at 3 years. The weakness of these studies was that they were all case reports, and the follow-up duration can be considered short for determining the true recurrence rate given the extended period during which recurrence can develop (3,16,40–42,46). Lin et al (47) reported on 48 patients with surgically treated chondroblastoma who were followed up for a minimum of 12 months. Local recurrence was found to be dependent on 2 factors, tumor aggressiveness and inadequate resection. Recurrent chondroblastomas appear to have a high growth activity. Testing for the presence of proliferating cell nuclear antigen might be useful in determining the tumor aggressiveness and, thus, whether the potential for recurrence is increased. Inadequate resection has been reported to be the only parameter for local recurrence and has been, by far, the primary reported cause (3,9,32,39,40,42,46,47). The recurrence in the present patient was postulated to be secondary to inadequate resection. The postoperative radiographs after the index procedure and, later, the advanced imaging studies revealed areas of the tumor still present at the most inferoposterior aspect of the calcaneus, with subsequent expansion. Repeated aggressive curettage, high-speed burring, cryosurgery, back filling with PMMA, and reinforcement with Steinman pins to restore the bone stock were used to prevent additional recurrence and the potential for malignant conversion and metastasis. Although the risk of malignant conversion and metastasis is low, it does exist. In the past, the potential for these was found to be increased after adjuvant radiotherapy, making its use in the treatment of chondroblastomas contraindicated (11,17,18,22). Local recurrence is the current primary factor for malignant conversion and metastasis (40,46,47). Rodgers and Mankin (38) reported on 80 patients with chondroblastoma treated surgically and followed up for >25 years. During that period, 6 patients (7.5%) developed local recurrence, 2 (2.5%) progressed to distant metastasis, and 1 (1.3%) died, with the death attributed to malignant conversion. Malignant conversion and metastasis after previous surgical treatment has been postulated to be secondary to the accidental surgical implantation of tumor cells within the lymphatic or vascular system (63–69). Metastasis occurs primarily through the pulmonary system and has been reported to occur 4 months to 33 years after the index surgical procedure (43–45,65–67). The treatment of pulmonary metastasis with chemotherapy, radiotherapy, or surgical excision has been reported to result in survival (43,44). The evaluation of locally recurrent lesions should include the exclusion of metastasis, including a chest radiography, chest CT, bone scintigraphy, and MRI, at a minimum (44,45). Although chondroblastomas are rare, they do occur in the foot, with the calcaneus the fifth most common location of occurrence in the body (7,8,11,18–32). The present case revealed the characteristic findings associated with chondroblastoma: occurrence in a male in
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the second decade of life, located in the region of the calcaneal apophysis, imaging study findings consistent with chondroblastoma, and specific histologic testing that confirmed the diagnosis. The present case had the added difficulty of treating an associated pathologic fracture, which has been reported to be rare in association with chondroblastoma (7,17,18,36). The local recurrence in our patient was most likely secondary to inadequate resection, which has been reported to be the most common cause (3,9,32,39,40,42,46,47). An evaluation to rule out metastasis was performed, just as should be done in all cases of local recurrence. Repeat surgical intervention with curettage, high-speed burring, cryosurgery, back filling of the void with PMMA, and Steinman pins for reinforcement has been reported to minimize the potential for recurrence and were used in our patient (41,46,55,58). The present patient was instructed to follow-up with an orthopedic oncologist at the closest teaching hospital to his home and to obtain repeat foot and chest radiographs every 3 to 6 months for the next 2 years and annually thereafter for lifelong monitoring because of the potential for malignant conversion and metastasis. References 1. Smith DA, Graham WC, Smith FR. Benign chondroblastoma of bone: report of an unusual case. J Bone Joint Surg Am 44-A:571–577, 1962. 2. Hull MT, Gonzalez–Crussi F, DeRosa GP, Graul RS. Aggressive chondroblastoma: report of a case with multiple bone and soft tissue involvement. Clin Orthop Relat Res 126:261–265, 1977. 3. Springfield DS, Capanna R, Gherlinzoni F, Picci P, Campanacci M. Chondroblastoma: a review of seventy cases. J Bone Joint Surg Am 67:748–755, 1985. 4. Sundaram TK. Benign chondroblastoma. J Bone Joint Surg Br 48:92–104, 1966. 5. McBryde A Jr, Goldner JL. Chondroblastoma of bone. Am Surg 36:94–108, 1970. 6. Kunkel MG, Dahlin DC, Young HH. Benign chondroblastoma. J Bone Joint Surg Am 38-A:817–826, 1956. 7. Kurt AM, Unni KK, Sim FH, McLeod RA. Chondroblastoma of bone. Hum Pathol 20:965–976, 1989. 8. Davila JA, Amrami KK, Sundaram M, Adkins MC, Unni KK. Chondroblastoma of the hands and feet. Skeletal Radiol 33:582–587, 2004. 9. Schajowicz F, Gallardo H. Epiphyseal chondroblastoma of bone: a clinicopathological study of sixty-nine cases. J Bone Joint Surg Br 52:205–226, 1970. 10. Raina V. Benign chondroblastomada clinico-pathological study of ten cases. Indian J Cancer 16:22–25, 1979. 11. Bloem JL, Mulder JD. Chondroblastoma: a clinical and radiological study of 104 cases. Skeletal Radiol 14:1–9, 1985. 12. Iwai T, Abe S, Miki Y, Tokizaki T, Matsuda K, Wakimoto N, Nakamura S, Imamura T, Matsushita T. A trapdoor procedure for chondroblastoma of the femoral head: a case report. Arch Orthop Trauma Surg 128:763–767, 2008. 13. Valls J, Ottolenghi CE, Schajowicz F. Epiphyseal chondroblastoma of bone. J Bone Joint Surg Am 33-A:997–1009, 1951. 14. Treasure ER. Benign chondroblastoma of bone: report of a case. J Bone Joint Surg Br 37-B:462–465, 1955. 15. Sherman RS, Uzel AR. Benign chondroblastoma of bone: its roentgen diagnosis. Am J Roentgenol Radium Ther Nucl Med 76:1132–1140, 1956. 16. Turcotte RE, Kurt AM, Sim FH, Unni KK, McLeod RA. Chondroblastoma. Hum Pathol 24:944–949, 1993. 17. Dahlin DC, Ivins JC. Benign chondroblastoma: a study of 125 cases. Cancer 30:401– 413, 1972. 18. Huvos AG, Marcove RC. Chondroblastoma of bone: a critical review. Clin Orthop Relat Res 95:300–312, 1973. 19. Katz HB, Hunt DW. Chondroblastoma of the calcaneus: a case report. J Am Podiatry Assoc 65:184–187, 1975. 20. Kahmann R, Gold RH, Eckardt JJ, Mirra JM. Case report 337: cystic chondroblastoma of calcaneus. Skeletal Radiol 14:301–304, 1985. 21. Fink BR, Temple HT, Chiricosta FM, Mizel MS, Murphey MD. Chondroblastoma of the foot. Foot Ankle Int 18:236–242, 1997. 22. Dhatt SS, Bhagwat KR, Kumar V, Dhillon MS. Chondroblastoma in a metatarsal treated with autogenous fibular graft: a case report. J Foot Ankle Surg 51:356–361, 2012. 23. Fang Z, Chen M. Chondroblastoma associated with aneurysmal cyst of the navicular bone: a case report. World J Surg Oncol 11:50, 2013. ral G, Yıldırım A, Kandemir O. Chondroblastoma of the 24. Arıkan M, Aktas E, Tog medial cuneiform bone in a 32-year-old woman. Am J Case Rep 28:317–321, 2014. 25. Kricun ME, Kricun R, Haskin ME. Chondroblastoma of the calcaneus: radiographic features with emphasis on location. AJR Am J Roentgenol 128:613–616, 1977. 26. Barbera C, Pinotti N, Klein MJ, Lewis MM. An unusual case of cystic chondroblastoma of the calcaneus: a case report. Bull Hosp Jt Dis Orthop Inst 48:88–92, 1988.
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