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Journal of Bone and Soft Tissue Tumors (JBST) is the official Journal of The Indian Musculo Skeletal Oncology Society
Retrospective Study of Seven Patients with Tumoral Calcinosis
Original Article | Volume 6 | Issue 2 | JBST May-August 2020 | Page 12-16 | Kshitij Manerikar, Abhijeet Salunke, Jaymin V. Shah, Mayur Kamani, Shashank Pandya. DOI: 10.13107/jbst.2020.v06i02.25
Author: Kshitij Manerikar[1], Abhijeet Salunke[1], Jaymin V. Shah[1], Mayur Kamani[1], Shashank Pandya[1]
[1]Department of Surgical Oncology, Gujarat Cancer Research and Institute, Ahmedabad, Gujarat, India.
Address of Correspondence
Dr. Kshitij Manerikar,
A-302, Divyadeep, Ram Mandir Road, TPS-3, Borivali West, Mumbai – 400 092, Maharashtra, India.
E-mail: drkshitijmanerikar@gmail.com
Abstract
Introduction: Calcium deposition in the skin has been termed as calcinosis cutis. Tumoral calcinosis is idiopathic form of calcinosis cutis. Etiology of idiopathic calcinosis cutis is unknown. It is characterized by periarticular deposition of amorphous calcium salts around large joints. Our diligent search through literature could not find any consensus on the etiopathogenesis and treatment modalities for tumoral calcinosis.
Materials and Methods: A retrospective study of seven patients of tumoral calcinosis treated with complete surgical excision over a period of 1 year was done. Demographic details were compiled. Routine blood investigations were performed. All patients underwent radiographs and magnetic resonance imaging (MRI) scans of involved part. We did not perform computed tomography (CT) or bone scan in any of our patients. All seven patients underwent surgery and were followed up till 2 years.
Results: In our study, five were female and two were male patients ranging from 31 to 76 years. Size of swelling varied from 2 to 15 cm. Most common location was hip. Serum calcium, phosphorus, and alkaline phosphatase were normal in all patients. Radiographs showed well-outlined periarticular cluster of calcifications in the soft tissues around joint. MRI revealed round to oval multiple cystic lesions around the affected region, but not involving the joint.
Conclusion: Tumoral calcinosis is always the diagnosis of exclusion. It can be normophosphatemic or hypophosphatemic subtype. Large joints are more commonly affected. One can rely on radiographs for diagnosis. MRI for knowing exact location of lesion, its relationship with adjacent structures and planning of surgery is advocated. Complete surgical excision is the only optimum treatment of tumoral calcinosis.
Keywords: Amorphous calcium phosphate, hyperphosphatemia, X-ray film, hip joint, calcinosis, magnetic resonance imaging.
Reference:
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3. Chaabane S, Chelli-Bouaziz M, Jelassi H, Mrad K, Smida M, Ladeb MF. Idiopathic tumoral calcinosis. Acta Orthop Belg 2008;74:837-45.
4. Muddegowda P, Lingegowda J, Ramachandrarao R, Konapur PG. Calcinosis cutis: Report of 4 Cases. J Lab Physicians 2011;3:125-6.
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8. Noyez J, Murphree S, Chen K. Tumoral calcinosis, a clinical report of eleven cases. Acta Orthop Belg 1993;59:49-54.
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Clear Cell Sarcoma: A Rare Aggressive Tumor with Potential Diagnostic Challenge
Original Article | Volume 6 | Issue 2 | JBST May-August 2020 | Page 9-11 | Adam Goldman, Matthew Doscher, Neil Lancaster, Paul Lamberti. DOI: 10.13107/jbst.2020.v06i02.24
Author: Adam Goldman[1], Matthew Doscher[1], Neil Lancaster[2], Paul Lamberti[1]
[1]Department of Orthopaedics, John H. Stroger, Jr. Hospital of Cook County, Chicago, Illinois, USA,
[2]Department of Orthopaedics, Franciscan Health- Olympia Fields, Illinois, USA.
Address of Correspondence
Dr. Adam Goldman,
Orthopedic Surgery Resident, Franciscan Health Olympia Fields
20201 S. Crawford Ave, Olympia Fields, IL 60461.
E-mail: adamgoldman91@gmail.com
Abstract
Introduction: Synovial sarcomas of the hand are extremely rare, high grade, slow growing, malignant soft-tissue neoplasms. They represent only a fraction of soft-tissue sarcomas, are usually found in the lower extremities around tendon sheaths, bursae, and joint capsules, and carry a significant risk for recurrence and eventual metastasis to the lymph nodes and lungs. There are limited reports of synovial sarcomas of the hand.
Case Report: A 67-year-old male presented with a slow-growing soft-tissue mass of the proximal right long finger over the course of one year. Initial excision was performed, which provided a diagnosis of monophasic synovial cell sarcoma. Subsequently, the patient underwent further staging with an eventual ray resection of the right long finger and an index ray transfer.
Conclusion: There are unique approaches to a patient with a highly malignant neoplasm in the hand. By performing a ray resection and index ray transfer, the patient was spared the potential side effects of radiation therapy and provided a quality functional and cosmetic outcome. The patient returned to work 4 weeks postoperatively. To the best of our knowledge, this treatment has not been used for a synovial sarcoma of the hand.
Keywords: Hand, synovial sarcoma, soft-tissue sarcoma, soft-tissue neoplasms
Reference:
1. Siegel HJ, Sessions W, Casillas MA Jr., Said-Al-Naief N, Lander PH, Lopez-Ben R. Synovial sarcoma: Clinicopathologic features, treatment, and prognosis. Orthopedics 2007;30:1020-7.
2. Rosa AC, Machado MM, Filho LEG, Albertotti F, Sato E, Figueiredo MAJ et al. Sarcoma sinovial fibroso do pe: Relato de caso. Radiol Bras 2002;35:51-4.
3. Michal M, Fanburg-Smith JC, Lasota J, Fetsch JF, Lichy J, Miettinen M. Minute synovial sarcomas of the hands and feet: A clinicopathologic study of 21 tumors less than 1 cm. Am J Surg Pathol 2006;30:721-6.
4. Okcu MF, Munsell M, Treuner J, Mattke A, Pappo A, Cain A, et al. Synovial sarcoma of childhood and adolescence: A multicenter, multivariate analysis of outcome. J Clin Oncol 2003;21:1602-11.
5. Pradhan A, Cheung YC, Grimer RJ, Peake D, Al-Muderis OA, Thomas JM, et al. Soft-tissue sarcomas of the hand: Oncological outcome and prognostic factors. J Bone Joint Surg Br 2008;90:209-14.
6. Outani H, Hamada K, Oshima K, Joyama S, Naka N, Araki N, et al. Clinical outcomes for patients with synovial sarcoma of the hand. Springerplus 2014;3:649.
7. Sahoo TK, Dhal I, Das SK, Majumdar SK, Parida DK. Synovial sarcoma of palmar aspect of hand and survival: A rare case report. J Clin Diagn Res 2017;11:XD9-11.
8. Eilber FC, Dry SM. Diagnosis and management of synovial sarcoma. J Surg Oncol 2008;97:314‐20.
9. Italiano A, Penel N, Robin YM, Bui B, Le Cesne A, Piperno-Neumann S, et al. Neo/adjuvant chemotherapy does not improve outcome in resected primary synovial sarcoma: A study of the French sarcoma group. Ann Oncol 2009;20:425-30.
10. Maia DC, Menezes CK, Bastos TC, de Lima Ferreira LC, Francesconi F. Poorly differentiated synovial sarcoma in the wrist: Case report. An Bras Dermatol 2014;89:816-18.
11. Puhaindrain ME, Healy JH, Athanasian EA. Single ray amputations for tumors of the hand. Clin Orthop Relat Res 2010;468:1390-5.
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Fracture Prosthesis in Giant Cell Tumor of Distal Radius: Precautions and Management
Original Article | Volume 6 | Issue 2 | JBST May-August 2020 | Page 5-8 | Mahesh Kulkarni, Sourab Shetty, Monappa Naik, Sandeep Vijayan. DOI: 10.13107/jbst.2020.v06i02.23
Author: Mahesh Kulkarni[1], Sourab Shetty[1], Monappa Naik[1], Sandeep Vijayan[1]
[1]Department of Orthopaedics, Kasturba Medical College, Manipal Academy of Higher Education, Manipal, Udupi, Karnataka, India.
Address of Correspondence
Dr. Sourab Shetty,
Department of Orthopaedics, Kasturba Medical College, Manipal Academy of Higher Education, Manipal – 576 104, Udupi, Karnataka, India.
E-mail: sourab.shetty.ss@gmail.com
Abstract
Introduction: Distal radius is one of the common locations for giant cell tumor (GCT) in the second to the fourth decade of life. Based on the extent of the tumor, various treatment options are available such as curettage and bone grafting or use of bone cement, various ablation techniques, excision, and vascularized or non-vascularized fibular graft. However, when subchondral bone and articular surface are involved and the patient is not ready for the fibular grafting, one of the methods described is excision and use of custom-made acrylic prosthesis, of which fracture prosthesis is a complication. Here, we are reporting a case who presented with a fractured prosthesis, the way we managed it and the precautions that can be taken while use of the prosthesis.
Case Report: We are presenting a case of a 42-year-old male who came back after 2 years of implantation with a fractured prosthesis, which happened during a routine activity. There was no evidence of recurrence of the tumor. A discussion about the cause, the management done, precautions to be taken while implanting acrylic prosthesis, and a follow-up of the patient for 3 years is also done.
Conclusion: Acrylic can be used as a cost-efficient material for a prosthesis in both primary excision and revisions in distal radius GCT with good functional results if specific precautions are taken preoperatively and intraoperatively.
Keywords: Acrylic prosthesis, broken prosthesis, fractured prosthesis, radius giant cell tumor.
Reference:
1. Renard AJ, Veth RP, Pruszczynski M, Wobbes T, Lemmens JA, van Horn JR. Giant cell tumor of bone: Oncologic and functional results. J Surg Oncol 1994;57:243-51.
2. Ekardt JJ, Grogan TJ. Giant cell tumor of bone. Clin Orthop Relat Res 1986;204:43-48.
3. Jaffe HL, Lichtenstein L, Portis RB. Giant cell tumor of the bone: Its pathological appearance, grading, supposed variant and treatment. Arch Pathol 1940;30:993-1031.
4. Klenke FM, Wenger DE, Inwards CY, Rose PS, Sim FH. Giant cell tumor of bone: Risk factors for recurrence. Clin Orthop Relat Res 2011;469:591-9.
5. O’Donnell RJ, Springfield DS, Motwani HK, Ready JE, Gebhardt MC, Mankin HJ. Recurrence of giant-cell tumors of the long bones after curettage and packing with cement. J Bone Joint Surg Am 1994;76:1827-33.
6. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am 1987;69:106-14.
7. Sanerkin NG. Malignancy, aggressiveness, and recurrence in giant cell tumor of bone. Cancer 1980;46:1641-9.
8. Trieb K, Bitzan P, Lang S, Dominkus M, Kotz R. Recurrence of curetted and bone-grafted giant-cell tumours with and without adjuvant phenol therapy. Eur J Surg Oncol 2001;27:200-2.
9. Briggs TW, Cobb J, McAuliVe T, Pringle J, Kemp H. Giant cell tumours of bone. J Bone Joint Surg 1990;72B:937.
10. Griend RA, Funderburk CH. The treatment of giant-cell tumors of the distal part of the radius. J Bone Joint Surg Am 1993;75A:899-908.
11. Gold AM. Use of a prosthesis for the distal portion of the radius following resection of a recurrent giant-cell tumor. J Bone Joint Surg 1957;39A:1374.
12. Blake SM, Gie GA. Large pelvic giant cell tumor: A Case report and a review of current treatment modalities. J Arthroplasty 2004;19:1050-4.
13. Errani C, Ruggieri P, Asenzio MA, Toscano A, Colangeli S, Rimondi E, et al. Giant cell tumor of the extremity: A review of 349 Cases from a single institution. Cancer Treat Rev 2010;36:1-7.
14. McGough RL, Rutledge J, Lewis VO, Lin PP, Yasko AW. Impact severity of local recurrence in giant cell tumor of bone. Clin Orthop Relat Res 2005;438:116-22.
15. Saraf S, Goel S. Complications of resection and reconstruction in giant cell tumour of distal end of radius-an analysis. Indian J Orthop 2005;39:206.
16. Chadha M, Arora SS, Singh AP, Gulati D, Singh AP. Autogenous non-vascularized fibula for treatment of giant cell tumor of distal end radius. Arch Orthop Trauma Surg 2010;130:1467-73.
17. Boons HW, Keijser LC, Schreuder BH, Pruszczynski M, Lemmens JA, Veth RP. Oncologic and functional results after treatment of giant cell tumors of bone. Arch Orthop Trauma Surg 2002;122:17-23.
18. Beyli MS, von Fraunhofer JA. An analysis of causes of fracture of acrylic resin dentures. J Prosthet Dent 1981;46:238-41.
19. Beyli MS, von Fraunhofer JA. Repair of fractured acrylic resin. J Prosthet Dent 1980;44:497-503.
20. Vallittu PK, Lassila VP. Effect of metal strengthener’s surface roughness on fracture resistance of acrylic denture base material. J Oral Rehabil 1992;19:385-91.
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Resection and Reconstruction of Calcaneal Tumors – A Review
Original Article | Volume 6 | Issue 2 | JBST May-August 2020 | Page 2-4 | Subbiah Shanmugam, Sujay Susikar, Murali Kannan. DOI: 10.13107/jbst.2020.v06i02.22
Author: Subbiah Shanmugam[1], Sujay Susikar[1], Murali Kannan[1]
[1]Department of Surgical Oncology, Associate Professor, Centre for Oncology, Government Royapettah Hospital, Chennai, Tamil Nadu, India.
Address of Correspondence
Dr. Sujay Susikar,
Department of Oncology, Centre for Oncology, Government Royapettah Hospital, Chennai, Tamil Nadu, India.
E-mail: sujaysusikar@gmail.com
Abstract
Introduction: Calcaneum is a rare site for Ewing’s sarcoma. The treatment includes neoadjuvant systemic therapy followed by surgical resection and adjuvant systemic therapy. The reconstruction options in this era of limb salvage for bone tumors are not much established in calcaneal tumors in view of limited reporting. Various other reconstruction options available are allograft, iliac crest autograft, and custom-made prosthesis.
Case Report: We have reported a 13-year-old female patient with Ewing’s sarcoma of calcaneum diagnosed by imaging and biopsy. The patient underwent neoadjuvant chemotherapy and then had undergone total calcanectomy with allograft reconstruction. The post-operative outcomes are fair, and the patient is on adjuvant chemotherapy at present.
Conclusion: Biological reconstruction in the form of allograft is a reliable option with regard to the functional outcomes for calcaneal resections.
Keywords: Calcaneum, Ewing sarcoma, total calcanectomy, allograft.
Reference:
1. Sherif PA, Santa A. Ewing’s sarcoma of the calcaneum. Indian J Med Paediatr Oncol 2017;38:542-4.
2. Imanishi J, Choong PF. Three-dimensional printed calcaneal prosthesis following total calcanectomy. Int J Surg Case Rep 2015;10:83-7.
3. Ottolenghi CE, Petracchi LJ. Chondromyxosarcoma of the calcaneus; report of a case of total replacement of involved bone with a homogenous refrigerated calcaneus. J Bone Joint Surg 1953;35:211-4.
4. Scoccianti G, Campanacci DA, Innocenti M, Beltrami G, Capanna R. Total calcanectomy and reconstruction with vascularized iliac bone graft for osteoblastoma: A report of two cases. Foot Ankle Int 2009;30:716-20.
5. Kurvin LA, Volkering C, Kessler SB. Calcaneus replacement after total calcanectomy via vascularized pelvis bone. Foot Ankle Surg 2008;14:221-4.
6. Anacak Y, Sabah D, Demirci S, Kamer S. Intraoperative extracorporeal irradiation and re-implantation of involved bone for the treatment of musculoskeletal tumors. J Exp Clin Cancer Res 2007;26:571.
7. 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.
8. Brenner P, Zwipp H, Rammelt S. Vascularized double barrel ribs combined with free serrate us anterior muscle transfer for homologous restoration of the hind foot after calcanectomy. J Trauma Acute Care Surg 2000;49:331-5.
9. Elsalanty ME, Genecov DG. Bone grafts in craniofacial surgery. Craniomaxillofac Trauma Reconstr 2009;2:125-34.
10. 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;28:841-4.
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Aneurysmal Bone Cyst – Review
Original Article | Volume 6 | Issue 1 | JBST Jan-April 2020 | Page 17-20| DOI: 10.13107/jbst.2020.v06i01.009
Author: Nitin Shetty[1], Prateek Hegde[2], Hemant Singh[3], Ashish Gulia[4]
[1]Department of Radio-Diagnosis, Tata Memorial Centre, Homi Bhabha National Institute (HBNI), Mumbai, India.
[2]Department of Surgical Oncology, Tata Memorial Centre, HBNI, Mumbai, India.
Address of Correspondence
Dr. Ashish Gulia
Tata Memorial Centre, Homi Bhabha National Institute, Dr. E Borges Road, Parel, Mumbai – 400 012, India.
E- mail: aashishgulia@gmail.com
Abstract
Background: Aneurysmal bone cyst (ABC) is a rare, benign, expansile lesion that produces blood-filled cavities inside the bone. The term, ‘Aneurysmal bone cyst’ was first time used by Jaffe and Lichtenstein in 1942 [1]. The name is a misnomer, as they are neither aneurysmal nor are, they truly cystic, as these lesions do not have an endothelial lined cyst wall. ABC is a disease of childhood or young adulthood with a median age of 13 years, with an incidence of 0.14 per 105 individuals with slight female preponderance [2].
Pathophysiology: ABC usually present as a solitary lesion either as a primary neoplasm (translocation driven) or a secondary lesion arising adjacent to previous bony lesions like giant cell tumours (GCT), osteoblastomas, chondroblastomas [3]. Few authors have proposed post traumatic hypothesis whereas others feel it could due to an hemodynamic disturbance especially venous impedance.
Primary ABCs: Primary ABC is one where it occurs in a bone without any previously known lesion. But now there has been identification of TRE17 also known as USP6 (ubiquitin-specific protease 6) gene on chromosome 17p13.Pathogenesis of some primary ABC involves transcriptional up-regulation of USP6 when there is the chromosomal translocation t(16;17)(q22;p13) which fuses the promoter region of the osteoblast cadherin 11 gene (CDH11) on chromosome 16q22 to the entire coding sequence of the ubiquitin protease USP6 gene on chromosome 17p13 [4].
Secondary ABCs: Approximately one third of the ABCs appear secondary to other pre-existing bone tumours, most commonly from GCT, which accounts for 19-39% of these cases [5]. Other common precursor lesions are chondroblastomas, chondromyxoid fibroma, fibrous dysplasia, osteoblastomas, haemangioendothelioma, angioma, fibroxanthoma (nonossifying fibroma), solitary bone cyst, fibrous histiocytoma, eosinophilic granuloma, radiation osteitis, osteosarcoma, trauma (including fracture), fibrosarcoma and even metastatic carcinoma [3, 5].
ABCs have been likened to a “blood-filled sponge”, composed of blood-filled, anastomosing, cavernomatous spaces, separated by a cyst like wall composed of fibroblasts, myofibroblasts, osteoclast like giant cells, osteoid and woven bone.
In approximately one third of cases, a characteristic reticulated lacy chondroid like material, described as a calcified matrix with a chondroid aura, is seen [6]. These are called as “solid aneurysmal bone cyst”. The term “solid aneurysmal bone cyst,” was coined by Sanerkin et al. in 1983 [7].
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15. Reddy KIA, Sinnaeve F, Gaston CL, Grimer RJ, Carter SR.Aneurysmal bone cysts: do simple treatments work? Clin OrthopRelat Res. 2014;472(6):1901–1910.
16. Steffner RJ, Liao C, Stacy G, Atanda A, Attar S, Avedian R, et al. Factors associated with recurrence of primary aneurysmal bone cysts: is argon beam coagulation an effective adjuvant treatment? J Bone Joint Surg Am. 2011;93(21):1221–1229.
17. Park HY, Yang SK, Sheppard WL, Hegde V, Zoller SD, Nelson SD, et al. Current management of aneurysmal bone cysts. Curr Rev Musculoskelet Med. 2016;9(4):435-444.
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19. Flont P, Kolacinska-Flont M, Niedzielski K. A comparison of cyst wall curettage and en bloc excision in the treatment of aneurysmal bone cysts. World J Surg Oncol.2013;11:109.
20. Elsayad K, Kriz J, Seegenschmiedt H, Imhoff D, Heyd R, Eich HT, et al.Radiotherapy for aneurysmal bone cysts: a rare indication. Strahlenther Onkol.2017;193(4):332-340.
21. Batisse F, Schmitt A, Vendeuvre T, Herbreteau D, Bonnard C. Aneurysmal bone cyst: A 19-case series managed by percutaneous sclerotherapy. Orthop Traumatol Surg Res.2016;102(2):213-216.
22. Varshney MK, Rastogi S, KhanSA, Trikha V. Is sclerotherapy better than intralesional excision for treating aneurysmal bone cysts? Clin Orthop Relat Res. 2010;468(6):1649-1659.
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Editorial IMSOS Issue January – April 2020
Original Article | Volume 6 | Issue 1 | JBST January-April 2020 | Page 21 | DOI: 10.13107/jbst.2020.v06i01.010
Author: Dr. Yogesh Panchwagh[1] , Dr. Ashish Gulia[2] & Dr. Ashok Shyam[3, 4]
1. Orthopaedic Oncology Clinic, Pune, India,
2. Orthopedic Oncology Services, Department of Surgical Oncology,
Tata Memorial Hospital, Mumbai,
3. Indian Orthopaedic Research Group, Thane, India,
4. Sancheti Institute for Orthopaedics &Rehabilitation, Pune, India.
Address of Correspondence:
Dr. Yogesh Panchwagh.
Orthopaedic Oncology Clinic, 101, Vasant plot 29, Bharat Kunj
Society -2, Erandwana, Pune – 38, India.
Email: drpanchwagh@gmail.com
We are pleased to bring you the IMSOS issue for 2020. JBST is the official publication of the Indian musculoskeletal oncology society (IMSOS). This is the second consecutive year that we are publishing a special IMSOS issue, which typically coincides with the IMSOS annual conference in early March.
Since the last year, JBST editorial board has 2 IMSOS life members selected as IMSOS representatives. Dr. Akshay Tiwari and Dr. Dominic Putthoor are the current IMSOS representatives on the JBST editorial board. Both have a vast experience of treating bone and soft tissue tumors and bring along considerable credibility to the work published in the issue.
IMSOS annual conference this year is being held at Bengaluru. The organising secretary Dr. Suman Byregowda and his team have put up an exceptional scientific program. The current IMSOS issue being released online to coincide with the annual conference promises to add to the academic value that IMSOS had aimed at. The conference is being held on the background of the Covid 19 scare that is looking to grip India. Hopefully the great academic content, warm hospitality and gracious hosts will not let the scare dampen the fervor and camaraderie.
JBST wishes IMSOS 2020 the very best.
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