Foot &http://fas.sagepub.com/ Ankle Specialist
Periosteal Osteosarcoma of the Calcaneum: A Case Report Daljit Singh, Ramesh Kumar Sen, Susheel Chaudhary and Sujit Kumar Tripathy Foot Ankle Spec published online 15 February 2012 DOI: 10.1177/1938640011434510 The online version of this article can be found at: http://fas.sagepub.com/content/early/2012/02/10/1938640011434510
Published by: http://www.sagepublications.com
Additional services and information for Foot & Ankle Specialist can be found at: Email Alerts: http://fas.sagepub.com/cgi/alerts Subscriptions: http://fas.sagepub.com/subscriptions Reprints: http://www.sagepub.com/journalsReprints.nav Permissions: http://www.sagepub.com/journalsPermissions.nav
>> OnlineFirst Version of Record - Feb 15, 2012 What is This?
Downloaded from fas.sagepub.com at Hinari on March 20, 2012
vol. x / no. x
Foot & Ankle Specialist
〈 Clinical Research 〉 Periosteal Osteosarcoma of the Calcaneum
Daljit Singh, MD, Ramesh Sen, MD, and Susheel Chaudhary, MD, Sujit Kumar Tripathy, MD
A Case Report
Abstract: A 30-year-old woman presented with a 6-month history of pain and swelling over the sole of her right foot. Plain radiograph showed a calcified mass in the heel pad, which appeared to be arising from the spur on inferior aspect of calcaneum. Magnetic resonance imaging showed a lesion, hypointense on T1-weeighted and hyperintense on T2-weighted images over the plantar aspect of the foot. Open biopsy of the mass was indicative of periosteal osteosarcoma, the variety that has never been reported in calcaneum. Below-knee amputation was done with no recurrence or distance metastasis seen at 24 months of follow up. Level of Evidence: Therapeutic, Level IV Keywords: periosteal osteosarcoma; calcaneum
O
steosarcoma of the foot is rare and constitutes about 0.8% of all osteosarcomas.1 Calcaneum seems to be the favored site at the foot followed by other tarsal and metatarsal bones, with phalangeal involvement being the least common.2-5 Various subtypes of calcaneum osteosarcomas have been reported in literature, such as
conventional osteosarcoma, low-grade central osteosarcoma, telangiectatic osteosarcoma, and small cell osteosarcoma.1-3,5-10 Periosteal variety of osteosarcoma of calcaneum is rare and has never been reported in literature. We report this rare variety of calcaneal osteosarcoma and its clinical and radiographic presentation.
Case Report
present over the heel and lateral aspect of the foot (Figure 1). The swelling was tender. The skin over the swelling was normal. The range of movements at the ankle joint was full and painless. Movements at the subtalar joint could not be assessed because of pain and swelling. Plain radiograph of the right foot showed a well-lineated calcified mass inferior to the calcaneum in the heel
“
A 30-year-old woman Calcaneum seems to be the favored presented with a history of pain and swelling over site at the foot followed by other tarsal and the sole of right foot for metatarsal bones, with phalangeal 6 months. Pain over the right heel pad was the iniinvolvement being the least common.” tial symptom for which she took analgesic and anti-inflammatory medication. The pain was continuous with no relation to activity and pad, which appeared to be arising from without any diurnal variation. The patient the spur on inferior aspect of calcaneal noticed swelling over the sole of her foot (Figure 2). Magnetic resonance imaging after about 1.5 months of initial sympof the right foot showed hyperintense toms. The pain and swelling increased lesion on T2-weighted and STIR (short-TI progressively. There was no history of inversion recovery) images in calcaneum. trauma, fever, or any discharging sinus. It was hypointense on T1-weighted There was no history of any other swellimages (Figure 3). The radiologist suging in the body. gested the differential diagnosis of calcaOn examination, a diffuse, bony hard neal osteomyelitis with involvement of swelling measuring around 6 ´ 4cm was surrounding muscles and soft tissue, or
DOI: 10.1177/1938640011434510. From the Department of Orthopaedics, Postgraduate Institute of Medical Education and Research, Chandigarh, India. Address correspondence to Daljit Singh, MD, Department of Orthopaedics, Postgraduate Institute of Medical Education and Research, Sector 12, Chandigarh, India; e-mail:
[email protected].. Copyright © 2009 The Author(s)
Downloaded from fas.sagepub.com at Hinari on March 20, 2012
1
2
Month xxxx
Foot & Ankle Specialist
Figure 1. Clinical photograph showing swelling over right heel.
Figure 2. Plain radiograph lateral view of the foot showing a well-defined mass arising from calcaneal tuberosity.
serum calcium, renal function tests, and liver function tests were within the normal range. Open biopsy of the lesion was performed through lateral aspect of the foot. The biopsy was performed under tourniquet. Full thickness flap was raised and tissue was taken from the lesion. Wound was closed after maintaining hemostasis. Specimen showed multiple small fragments predominantly composed of nodular lesion revealing cartilaginous differentiation. Periphery of these nodules showed aggregates and clusters of osteoblasts showing significant nuclear atypia and hyperchromasia. Many mitotic figures were seen, including atypical ones (Figure 4). The findings were suggestive of periosteal osteosarcoma.
and a moderate amount of cytoplasm (Figure 5). In addition, there were areas of reactive new bone formation. The features were confirmative of periosteal osteosarcoma. Figure 5. Photomicrograph showing spindleshaped cells and chondroid cells (hematoxylin and eosin ×100).
Figure 4. Photomicrograph showing nodular lesions with aggregates and clusters of osteoblasts showing significant nuclear atypia and hyperchromasia at their periphery (hematoxylin and eosin ×20).
Figure 3. Magnetic resonance image of the right foot showing hypointense lesion on T1weighted image..
posttraumatic marrow edema with swelling in the surrounding muscles and soft tissue. But these were not clinically correlating as there was no history and clinical features suggestive of infection or trauma. Routine hematological and biochemical investigations showed no abnormality. Alkaline phosphatase, lactate dehydrogenase,
Three-phase bone scan revealed no evidence of any skip metastasis. Contrastenhanced tomographies of the chest and abdomen ruled out any distant metastasis. The patient was admitted and explained the nature of the disease. Below-knee amputation was performed after taking consent from the patient. The tumor was sent for histopathological examination that showed lobules of cartilaginous tissue at the periphery of which there was new bone formation by the tumor cells. The tumor cells were oval- to spindle-shaped with hyperchromatic nuclei
Downloaded from fas.sagepub.com at Hinari on March 20, 2012
Patient received a full course of postoperative chemotherapy. The chemotherapy regimen incorporated doxorubicin, methotrexate, and cisplatin. Simultaneously, patient was put on a program of aggressive physiotherapy to train the amputated stump for a period of 4 weeks. Patient was back to her work using a belowknee prosthesis at the end of 4 months of her initial presentation to the institute. At a follow-up of 24 months, there was no evidence of any skip metastasis as revealed on bone scan. The patient was informed that the data would be submitted for publication, and she gave her consent.
Discussion Most of the osteosarcomas of calcaneum reported in the literature are of conventional type, with its various histological types, including osteoblastic, chondroblastic, or fibroblastic types.11 Cases of low-grade central osteosarcoma have been reported in few studies with relatively benign features on radiographs.2,3,9,10 There are few cases of telangiectatic variety of calcaneal osteosarcoma reported with radiological features similar to aneurysmal bone cyst.6 A
vol. x / no. x
single case of small cell osteosarcoma is reported with osteolytic lesion in calcaneum and destruction of inferior portion of calcaneum seen on radiograph.7 Periosteal osteosarcoma is an intermediate-grade malignancy that arises on the surface of the bone. The most common locations are the diaphyses of the femur and tibia. Histological examination of periosteal osteosarcoma shows strands of osteoid-producing spindle cells radiating between lobules of cartilage.12 To our knowledge, this is the first case of periosteal osteosarcoma of the calcaneum. Heel pain was the initial symptom in the present case. The common differential diagnoses of unilateral heel pain include plantar fasciitis, heel spur, tarsal tunnel syndrome, heel cord tendinitis, and retrocalcaneal bursitis. These present in middle-aged patients with longstanding history of heel pain. Although swelling may be associated with many of these disorders, progressive increase in size of swelling is not a feature.13 Owing to the rarity of osteosarcoma at this site, its diagnosis is often missed or delayed. Radiograph was done when the patient presented to us at 6 months, which showed a mass on the inferior aspect of the calcaneum. The possible differentials were myositis ossificans, osteochondroma, or a calcified soft tissue mass. As the calcaneal osteosarcoma with such radiological feature was never reported, it was not kept in the differential diagnosis. Magnetic resonance imaging is the best test to measure the extent of the tumor within the bone and the soft tissue. However, magnetic resonance imaging is nonspecific and is not useful in differentiating benign from malignant lesions.14 Magnetic resonance imaging in the present case was not helpful in suspecting the diagnosis. Although open biopsy has a greater risk of tumor spillage, hematoma, and infection, it has the highest diagnostic accuracy.15 The final diagnosis in the present case was only reached after the histopathological examination, which showed spindle-shaped cells with hyperchromatic nuclei and cartilaginous nodules and new bone formation. The presence of “woven bone” or
Foot & Ankle Specialist
“malignant osteoid” helps in confirming the diagnosis. The standard treatment of patients with conventional osteosarcoma consists of combination of chemotherapy and surgery. Periosteal osteosarcoma is an intermediate-grade malignancy and in general, chemotherapy is included in the treatment protocol of this tumor. Although the advent of adjuvant and neoadjuvant chemotherapy has increased the survival of patients with conventional osteosarcomas, the benefit of chemotherapy in periosteal osteosarcoma is doubtful and several studies have concluded that it does not improve the outcome or probability of survival.16-18 The most common surgery performed for calcaneal osteosarcomas is below-knee amputation2,3 with only a few reports of limb salvage surgery.9,10 As the periosteal variety of calcaneum osteosarcoma and its treatment has never been mentioned in the literature, we planned to go for below-knee amputation followed by full course of chemotherapy. At 24 months’ follow-up, there was no evidence of any distant metastasis or recurrence of tumor. Periosteal osteosarcoma of calcaneum is a rare variety. It should be borne in mind during the differential diagnosis of any mass arising from calcaneum so as to detect it as early as possible. Open biopsy is the best investigation in reaching at an early diagnosis and initiation of the appropriate treatment; which ultimately results in long-term survival of the patient.
5.
Lopez-Barea F, Contreras F, SanchezHerrera S. Case report 540: grade III conventional sclerosing osteosarcoma of the calcaneus. Skeletal Radiol. 1989;18:237-240.
6.
Leithner A, Bodo K, Scheipl S, Radl R, Kastner N, Windhager R. Two cases of calcaneal osteosarcomas presenting as aneurysmal bone cysts. Foot Ankle Int. 2004;25:815-818.
7.
Park YK, Ryu KN, Ahn JH, Yang MH. A small cell osteosarcoma on the calcaneus: a case report. J Korean Med Sci. 1995;10:147-151.
8.
Wozniak W, Raciborska A, Walenta T, Szafranski A, Szymborska A, Bajor M. New technique of surgical treatment of malignant calcaneal tumours. Orthop Traumatol Rehabil. 2007;9:273-276.
9.
Chou LB, Malawer MM. Osteosarcoma of the calcaneus treated with prosthetic replacement with twelve years of follow-up: a case report. Foot Ankle Int. 2007;28:841-844.
References
15. Skrzynski MC, Biermann JS, Montag A, Simon MA. Diagnostic accuracy and charge-savings of outpatient core needle biopsy compared with open biopsy of musculoskeletal tumors. J Bone Joint Surg Am. 1996;78:644-649.
1.
Wu KK. Tumour review: osteogenic sarcoma of the foot. J Foot Surg. 1987;26:269-271.
2.
Choong PF, Qureshi AA, Sim FH, Unni KK. Osteosarcoma of the foot: a review of 52 patients at the Mayo Clinic. Acta Orthop Scand. 1999;70:361-364.
3.
4.
Biscaglia R, Gasbarrini A, Bohling T, Bacchini P, Bertoni F, Picci P. Osteosarcoma of the bones of the foot: an easily misdiagnosed malignant tumor. Mayo Clin Proc. 1998;73:842-847. Sneppen O, Dissing I, Heerfordt J, Schlodt T. Osteosarcoma of the metatarsal bones: review of the literature and report of a case. Acta Orthop Scand. 1978;49:220-223.
Downloaded from fas.sagepub.com at Hinari on March 20, 2012
10. Li J, Guo Z, Pei GX, Wang Z, Chen GJ, Wu ZG. Limb salvage surgery for calcaneal malignancy. J Surg Oncol. 2010;102:48-53. 11. Rosenberg ZS, Lev S, Schmahmann S, Steiner GC, Beltran J, Present J. Osteosarcoma: subtle, rare and misleading plain film features. AJR Am J Roentgenol. 1995;165:1209-1214. 12. Ritts GD, Pritchard DJ, Unni KK, Beabout JW, Eckardt JJ. Periosteal osteosarcoma. Clin Orthop Relat Res. 1987;219:299-307. 13. Murphy A. Painful heel. In: Canale ST, Beaty JH, eds. Campbell’s Operative Orthopaedics. 11th ed. St Louis, MO: Mosby Elsevier; 2008:4815-4817. 14. Kalmar JA, Eick JJ, Merritt CR, et al. A review of applications of MRI in soft tissue and bone tumors. Orthopedics. 1988;11:417-425.
16. Grimer RJ, Bielack S, Flege S, et al. Periosteal osteosarcoma: a European review of outcome. Eur J Cancer. 2005;41:2806-2811. 17. Cesari M, Alberghini M, Vanel D, et al. Periosteal osteosarcoma: a singleinstitution experience. Cancer. 2011;117:1731-1735. 18. Rose PS, Dickey ID, Wenger DE, Unni KK, Sim FH. Periosteal osteosarcoma: long-term outcome and risk of late recurrence. Clin Orthop Relat Res. 2006;453:314-317.
3
