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Giant Cell Tumor of the Talus: 19-Year Follow-Up of a Patient
The Journal of Foot & Ankle Surgery xxx (2014) 1–4
Contents lists available at ScienceDirect
The Journal of Foot & Ankle Surgery journal homepage: www.jfas.org
Case Reports and Series
Giant Cell Tumor of the Talus: 19-Year Follow-up of a Patient Kwang-Soon Song, MD, PhD 1, Chul Hyun Park, MD, PhD 2 1 2
Department of Orthopedic Surgery, Keimyung University Hospital, Daegu, Korea Department of Orthopedic Surgery, Yeungnam University Hospital, Daegu, Korea
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4
Giant cell tumor in small bones is a rare condition characterized by extensive bony destruction and a high recurrence rate. Intralesional excision with curettage and autologous bone grafting has been used as a standard treatment method for giant cell tumor of the bones. We report the case of a 30-year-old female with giant cell tumor of the talar body. She was followed up for 19 years after intralesional curettage and autologous bone grafting treatment. Ó 2014 by the American College of Foot and Ankle Surgeons. All rights reserved.
Keywords: ankle biopsy bone graft curettage long-term follow up steroid
Giant cell tumors (GCTs) are uncommon in the small bones of the hand, foot, and ankle (1–3), and very few such cases have been reported. More specifically, GCT of the talus is uncommon and has very rarely been reported (4,5). The standard treatment of GCT of the bone has traditionally been intralesional excision with curettage and autologous bone grafting. However, curettage alone has had a high recurrence rate. We report the case of a 30-year-old female with of GCT that had led to extensive destruction of the talus. Intralesional curettage and impact autologous bone grafting was performed, after which the pain and swelling decreased. In the 19 years of follow-up, we have found no evidence of relapse or changes in her symptoms.
Case Report A 30-year-old female had presented with gradually increasing pain and motion limitation in the right ankle of several years’ duration. She had no history of trauma to the ankle, fever, loss of appetite or weight, or similar complaints in other joints. The symptoms had gradually increased in intensity, leading to her presentation. The physical examination findings revealed restricted movements of the ankle joint, in particular, in dorsiflexion of the ankle.
Financial Disclosure: None reported. Conflict of Interest: None reported. Address correspondence to: Chul Hyun Park, MD, PhD, Department of Orthopedic Surgery, Yeungnam University Hospital, Hyunchoongro 170, Daegu, Korea. E-mail address: [email protected] (C.H. Park).
The radiographs of the ankle revealed an osteolytic expansile lesion in the body of the talus, which had ballooned medially, and the cortex had thinned out. A round bony extrusion was noted on the dorsum of the talar neck. The ankle joint space was slightly narrowed (Fig. 1). She had no signs of infection clinically, and the serum biochemistry results were within normal limits. Magnetic resonance imaging revealed an expansile soft tissue mass in the body of the talus causing cortical thinning, which was demonstrated on a plain radiograph. The joint space of the ankle and subtalar joints was relatively well preserved (Fig. 2). An open biopsy was performed, and a definite diagnosis of GCT was made (Fig. 3). The condition, prognosis, and various treatment modalities were discussed at length with the patient. The patient consented to, and underwent, intralesional curettage with autograft reconstruction by packing the cavity of the excised tumor with morselized iliac cortical and cancellous bone (Fig. 4). Involvement of the articular cartilage or evidence of a pathologic fracture was not observed intraoperatively. A protective cast was applied for a 3-month period, and the patient was subsequently mobilized to full weightbearing status. Regular follow-up examinations at 1-year intervals were performed. At 19 years postoperatively, the ankle joint space was preserved, and she had experienced no aggravation of symptoms during her activities of daily living compared with the symptom intensity at her initial visit. At the latest follow-up examination, we had found no evidence of recurrence (Fig. 5) of the GCT on radiographic inspection, despite some joint space irregulariy, and the patient’s ankle clinically functioned well with 5 of dorsiflexion and 15 of plantarflexion.
1067-2516/$ - see front matter Ó 2014 by the American College of Foot and Ankle Surgeons. All rights reserved. http://dx.doi.org/10.1053/j.jfas.2014.04.003
2
K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
Fig. 1. (A) Anteroposterior and (B) lateral radiographs of our 30-year-old female patient showing an expansile osteolytic lesion in the body of the talus, which had ballooned medially, and the cortex had thinned out. A round bony extrusion was noted on the dorsum of the talar neck.
Discussion GCT is most commonly seen in the distal femur, proximal tibia, distal radius, and proximal humerus, in descending order of frequency (6). GCT is uncommon in the small bones of the foot or ankle (1–3), and GCT of the talus has been an even less common (1,4,5,7,8). Kinley et al (9) reported 35 cases of GCT involving the tarsal bones, with the most common site being the talus. Among the bony tumors involving the talus, GCT was the most common (10). The age of onset has been debated. Wold and Swee (4) stressed that, in contrast to GCT of the long bones, most GCTs of the small bones will occur in a young age group and will tend to be multicentric. However, Ramdas et al (7) reported that GCT of the talus has usually been seen in skeletally mature adults. Finally, osteoclastoma GCT of the talus is a rare entity and more commonly seen in the third decade of life (2).
GCT of the tarsal bones is uncommon, and the therapeutic options are ill defined (10). Traditionally, the standard treatment of GCT of the bone has been intralesional excision with curettage and autograft reconstruction by packing the cavity of the excised tumor with morselized iliac cortical and cancellous bone. Many investigators have reported satisfactory results with intralesional curettage and bone grafting (2). However, curettage alone has had a high rate of recurrence, and the use of adjuvant agents such as methyl methacrylate (bone cement), cryotherapy, and phenol have been reported. Several procedures, including fresh frozen osteochondral allografts (11), partial (3) or total excision (3,10) of the talus, and arthrodesis (3), have also been reported. The classic treatment of choice has been intralesional curettage. Recently, the use of cementing and cryotherapy has been increasing, with encouraging results. Recurrence has been very common for this benign, but locally aggressive, neoplasm (2). Chemical treatments such as phenol or cytotoxic agents such as chlorpactin have been
Fig. 2. Coronal (A) and sagittal (B) magnetic resonance images showing an expansile soft tissue mass in the body of the talus causing cortical thinning. The ankle and subtalar joint spaces were relatively well preserved.
K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
3
Fig. 3. Histopathologic findings with hematoxylin and eosin (H&E) stain. (A) A section of the yellow fragile tissue of the right talus lesion showing marked infiltration of foamy cells with spindle stromal cells and large multinucleated giant cells (H&E stain, original magnification 200). (B) Fibroblast-like spindle-cell proliferation, abundant collagen deposit, and foamy cell infiltration (H&E stain, original magnification 200). (C) A section of the yellow fragile tissue of the right talus showing marked infiltration of foamy cells, spindle stromal cells, and large multinucleated giant cells (H&E stain, original magnification 400).
applied to the tumor bed to decrease relapse rates, with varying results (12). The local recurrence rate has ranged from 27% to as high as 40% to 60% for intralesional procedures (5,13). The interval to local recurrence development after treatment has varied in published reports. Campanacci et al (5) reported that 90% of recurrences had
developed in the first 3 years after surgery. Although most recurrences of GCT of the bone can be expected within 2 years, some patients will remain at risk for a much longer period, up to 19 to 30 years, after curettage and bone grafting (12). In the presented case, no evidence of relapse or a change in pain intensity was observed at the most recent follow-up visit 19 years after surgery.
Fig. 4. (A) Anteroposterior and (B) lateral radiographs immediately postoperatively demonstrating changes after intralesional curettage with autograft reconstruction.
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K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
Fig. 5. (A) Anteroposterior and (B) lateral radiographs at 19 years postoperatively showing no interval changes from the radiographic study at 13 years of follow-up.
However, the patient will need to be monitored for recurrence development for as long as possible. References 1. Selek H, Ozer H, Turanli S, Erdem O. Giant cell tumor of the talar neck. J Am Podiatr Med Assoc 97:225–228, 2007. 2. Bapat MR, Narlawar RS, Pimple MK, Bhosale PB. Giant cell tumour of talar body. J Postgrad Med 46:110–111, 2000. 3. Dhillon MS, Singh B, Gill SS, Walker R, Nagi ON. Management of giant cell tumor of the tarsal bones: a report of nine cases and a review of the literature. Foot Ankle 14:265–272, 1993. 4. Wold LE, Swee RG. Giant cell tumor of the small bones of the hands and feet. Semin Diagn Pathol 1:173–184, 1984. 5. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am 69:106–114, 1987.
6. Stoker DJ. Bone tumours: general characteristics benign lesions. In: Diagnostic Radiology: A Textbook of Medical Imaging, pp. 629–1660, edited by RG Grainger, DJ Allison, Churchill Livingston, New York, 1997. 7. Ramdas A, Rao KR, Thomas A, Varghese RG, Gnanadoss JJ. Giant cell tumour of talusda case report. J Indian Med Assoc 105:96–98, 2007. 8. Sharma S, Wani IH, Gupta N, Mahajan N, Salaria AQ. Giant cell tumor of talus: a case report. Cases J 2:74, 2009. 9. Kinley S, Wiseman F, Wertheimer SJ. Giant cell tumor of the talus with secondary aneurysmal bone cyst. J Foot Ankle Surg 32:38–46, 1993. 10. Dhillon MS, Singh B, Singh DP, Prabhu V, Nagi ON. Primary bone tumors of the talus. J Am Podiatr Med Assoc 84:379–384, 1994. 11. Schoenfeld AJ, Leeson MC, Grossman JP. Fresh-frozen osteochondral allograft reconstruction of a giant cell tumor of the talus. J Foot Ankle Surg 46:144–148, 2007. 12. Scully SP, Mott MP, Temple HT, O’Keefe RJ, O’Donnell RJ, Mankin HJ. Late recurrence of giant-cell tumor of bone: a report of four cases. J Bone Joint Surg Am 76:1231–1233, 1994. 13. Carrasco CH, Murray JA. Giant cell tumors. Orthop Clin North Am 20:395–405, 1989.
Contents lists available at ScienceDirect
The Journal of Foot & Ankle Surgery journal homepage: www.jfas.org
Case Reports and Series
Giant Cell Tumor of the Talus: 19-Year Follow-up of a Patient Kwang-Soon Song, MD, PhD 1, Chul Hyun Park, MD, PhD 2 1 2
Department of Orthopedic Surgery, Keimyung University Hospital, Daegu, Korea Department of Orthopedic Surgery, Yeungnam University Hospital, Daegu, Korea
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4
Giant cell tumor in small bones is a rare condition characterized by extensive bony destruction and a high recurrence rate. Intralesional excision with curettage and autologous bone grafting has been used as a standard treatment method for giant cell tumor of the bones. We report the case of a 30-year-old female with giant cell tumor of the talar body. She was followed up for 19 years after intralesional curettage and autologous bone grafting treatment. Ó 2014 by the American College of Foot and Ankle Surgeons. All rights reserved.
Keywords: ankle biopsy bone graft curettage long-term follow up steroid
Giant cell tumors (GCTs) are uncommon in the small bones of the hand, foot, and ankle (1–3), and very few such cases have been reported. More specifically, GCT of the talus is uncommon and has very rarely been reported (4,5). The standard treatment of GCT of the bone has traditionally been intralesional excision with curettage and autologous bone grafting. However, curettage alone has had a high recurrence rate. We report the case of a 30-year-old female with of GCT that had led to extensive destruction of the talus. Intralesional curettage and impact autologous bone grafting was performed, after which the pain and swelling decreased. In the 19 years of follow-up, we have found no evidence of relapse or changes in her symptoms.
Case Report A 30-year-old female had presented with gradually increasing pain and motion limitation in the right ankle of several years’ duration. She had no history of trauma to the ankle, fever, loss of appetite or weight, or similar complaints in other joints. The symptoms had gradually increased in intensity, leading to her presentation. The physical examination findings revealed restricted movements of the ankle joint, in particular, in dorsiflexion of the ankle.
Financial Disclosure: None reported. Conflict of Interest: None reported. Address correspondence to: Chul Hyun Park, MD, PhD, Department of Orthopedic Surgery, Yeungnam University Hospital, Hyunchoongro 170, Daegu, Korea. E-mail address: [email protected] (C.H. Park).
The radiographs of the ankle revealed an osteolytic expansile lesion in the body of the talus, which had ballooned medially, and the cortex had thinned out. A round bony extrusion was noted on the dorsum of the talar neck. The ankle joint space was slightly narrowed (Fig. 1). She had no signs of infection clinically, and the serum biochemistry results were within normal limits. Magnetic resonance imaging revealed an expansile soft tissue mass in the body of the talus causing cortical thinning, which was demonstrated on a plain radiograph. The joint space of the ankle and subtalar joints was relatively well preserved (Fig. 2). An open biopsy was performed, and a definite diagnosis of GCT was made (Fig. 3). The condition, prognosis, and various treatment modalities were discussed at length with the patient. The patient consented to, and underwent, intralesional curettage with autograft reconstruction by packing the cavity of the excised tumor with morselized iliac cortical and cancellous bone (Fig. 4). Involvement of the articular cartilage or evidence of a pathologic fracture was not observed intraoperatively. A protective cast was applied for a 3-month period, and the patient was subsequently mobilized to full weightbearing status. Regular follow-up examinations at 1-year intervals were performed. At 19 years postoperatively, the ankle joint space was preserved, and she had experienced no aggravation of symptoms during her activities of daily living compared with the symptom intensity at her initial visit. At the latest follow-up examination, we had found no evidence of recurrence (Fig. 5) of the GCT on radiographic inspection, despite some joint space irregulariy, and the patient’s ankle clinically functioned well with 5 of dorsiflexion and 15 of plantarflexion.
1067-2516/$ - see front matter Ó 2014 by the American College of Foot and Ankle Surgeons. All rights reserved. http://dx.doi.org/10.1053/j.jfas.2014.04.003
2
K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
Fig. 1. (A) Anteroposterior and (B) lateral radiographs of our 30-year-old female patient showing an expansile osteolytic lesion in the body of the talus, which had ballooned medially, and the cortex had thinned out. A round bony extrusion was noted on the dorsum of the talar neck.
Discussion GCT is most commonly seen in the distal femur, proximal tibia, distal radius, and proximal humerus, in descending order of frequency (6). GCT is uncommon in the small bones of the foot or ankle (1–3), and GCT of the talus has been an even less common (1,4,5,7,8). Kinley et al (9) reported 35 cases of GCT involving the tarsal bones, with the most common site being the talus. Among the bony tumors involving the talus, GCT was the most common (10). The age of onset has been debated. Wold and Swee (4) stressed that, in contrast to GCT of the long bones, most GCTs of the small bones will occur in a young age group and will tend to be multicentric. However, Ramdas et al (7) reported that GCT of the talus has usually been seen in skeletally mature adults. Finally, osteoclastoma GCT of the talus is a rare entity and more commonly seen in the third decade of life (2).
GCT of the tarsal bones is uncommon, and the therapeutic options are ill defined (10). Traditionally, the standard treatment of GCT of the bone has been intralesional excision with curettage and autograft reconstruction by packing the cavity of the excised tumor with morselized iliac cortical and cancellous bone. Many investigators have reported satisfactory results with intralesional curettage and bone grafting (2). However, curettage alone has had a high rate of recurrence, and the use of adjuvant agents such as methyl methacrylate (bone cement), cryotherapy, and phenol have been reported. Several procedures, including fresh frozen osteochondral allografts (11), partial (3) or total excision (3,10) of the talus, and arthrodesis (3), have also been reported. The classic treatment of choice has been intralesional curettage. Recently, the use of cementing and cryotherapy has been increasing, with encouraging results. Recurrence has been very common for this benign, but locally aggressive, neoplasm (2). Chemical treatments such as phenol or cytotoxic agents such as chlorpactin have been
Fig. 2. Coronal (A) and sagittal (B) magnetic resonance images showing an expansile soft tissue mass in the body of the talus causing cortical thinning. The ankle and subtalar joint spaces were relatively well preserved.
K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
3
Fig. 3. Histopathologic findings with hematoxylin and eosin (H&E) stain. (A) A section of the yellow fragile tissue of the right talus lesion showing marked infiltration of foamy cells with spindle stromal cells and large multinucleated giant cells (H&E stain, original magnification 200). (B) Fibroblast-like spindle-cell proliferation, abundant collagen deposit, and foamy cell infiltration (H&E stain, original magnification 200). (C) A section of the yellow fragile tissue of the right talus showing marked infiltration of foamy cells, spindle stromal cells, and large multinucleated giant cells (H&E stain, original magnification 400).
applied to the tumor bed to decrease relapse rates, with varying results (12). The local recurrence rate has ranged from 27% to as high as 40% to 60% for intralesional procedures (5,13). The interval to local recurrence development after treatment has varied in published reports. Campanacci et al (5) reported that 90% of recurrences had
developed in the first 3 years after surgery. Although most recurrences of GCT of the bone can be expected within 2 years, some patients will remain at risk for a much longer period, up to 19 to 30 years, after curettage and bone grafting (12). In the presented case, no evidence of relapse or a change in pain intensity was observed at the most recent follow-up visit 19 years after surgery.
Fig. 4. (A) Anteroposterior and (B) lateral radiographs immediately postoperatively demonstrating changes after intralesional curettage with autograft reconstruction.
4
K.-S. Song, C.H. Park / The Journal of Foot & Ankle Surgery xxx (2014) 1–4
Fig. 5. (A) Anteroposterior and (B) lateral radiographs at 19 years postoperatively showing no interval changes from the radiographic study at 13 years of follow-up.
However, the patient will need to be monitored for recurrence development for as long as possible. References 1. Selek H, Ozer H, Turanli S, Erdem O. Giant cell tumor of the talar neck. J Am Podiatr Med Assoc 97:225–228, 2007. 2. Bapat MR, Narlawar RS, Pimple MK, Bhosale PB. Giant cell tumour of talar body. J Postgrad Med 46:110–111, 2000. 3. Dhillon MS, Singh B, Gill SS, Walker R, Nagi ON. Management of giant cell tumor of the tarsal bones: a report of nine cases and a review of the literature. Foot Ankle 14:265–272, 1993. 4. Wold LE, Swee RG. Giant cell tumor of the small bones of the hands and feet. Semin Diagn Pathol 1:173–184, 1984. 5. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am 69:106–114, 1987.
6. Stoker DJ. Bone tumours: general characteristics benign lesions. In: Diagnostic Radiology: A Textbook of Medical Imaging, pp. 629–1660, edited by RG Grainger, DJ Allison, Churchill Livingston, New York, 1997. 7. Ramdas A, Rao KR, Thomas A, Varghese RG, Gnanadoss JJ. Giant cell tumour of talusda case report. J Indian Med Assoc 105:96–98, 2007. 8. Sharma S, Wani IH, Gupta N, Mahajan N, Salaria AQ. Giant cell tumor of talus: a case report. Cases J 2:74, 2009. 9. Kinley S, Wiseman F, Wertheimer SJ. Giant cell tumor of the talus with secondary aneurysmal bone cyst. J Foot Ankle Surg 32:38–46, 1993. 10. Dhillon MS, Singh B, Singh DP, Prabhu V, Nagi ON. Primary bone tumors of the talus. J Am Podiatr Med Assoc 84:379–384, 1994. 11. Schoenfeld AJ, Leeson MC, Grossman JP. Fresh-frozen osteochondral allograft reconstruction of a giant cell tumor of the talus. J Foot Ankle Surg 46:144–148, 2007. 12. Scully SP, Mott MP, Temple HT, O’Keefe RJ, O’Donnell RJ, Mankin HJ. Late recurrence of giant-cell tumor of bone: a report of four cases. J Bone Joint Surg Am 76:1231–1233, 1994. 13. Carrasco CH, Murray JA. Giant cell tumors. Orthop Clin North Am 20:395–405, 1989.