A rare case of Rosai-Dorfman disease presenting as solitary lesion of distal femur: A case report

Cases Series | Volume 8 | Issue 1 | JBST January – June 2022 | Page 07-10 |Ebin Rahman, Subin Sugath, Nita John.

DOI: 10.13107/jbst.2021.v08i01.67

Author: Ebin Rahman [1], Subin Sugath [1], Nita John [1]

[1] Department of Orthopaedic Oncology, Aster Medcity, Kochi, Kerala, India

Address of Correspondence
Dr. Ebin Rahman,
Senior Specialist, Department of Orthopaedic Oncology, Aster Medcity, Kochi, Kerala, India.
E-mail: rahmanebin@gmail.com


Introduction: Rosai-dorfman disease (RDD) or sinus histiocytosis with massive lymphadenopathy is a rare histiocytic proliferative disorder of unknown etiology first identified and characterized by Rosai and Dorfman in 1969. The disease is characterized by painless cervical lymphadenopathy, fever, leukocytosis, elevated erythrocyte sedimentation rate, and hypergammaglobulinemia. <500 patients have been reported worldwide. Osseous involvement without lymphadenopathy occurs in <10% of patients and is usually multifocal. Extranodal solitary osseous lesions are just 4% of this subpopulation, no more than 50 such cases have been described in the literature. Here, we report a rare case of RDD presenting as solitary lesion of the distal femur without any evidence of lymphadenopathy and normal laboratory work up.
Case Report: We report the case of a 29-year-old female who presented with pain and swelling of distal femur of 2-month duration without any evidence of lymphadenopathy, with a normal laboratory workup. Radiographical evaluation showed an epiphyseal, osteolytic, and well-defined lesion with cortical break over the medial femoral condyle and soft-tissue extension into the medial patellar recess abutting the medial patellar retinaculum. Whole body positron emission tomography did not show any other sites of abnormal increased uptake. Intralesional extended curettage, bone grafting, bone cementing, and pinning were done. Histologically, the biopsied lesion demonstrated the classic histiocyte proliferation which exhibited emperipolesis (engulfment of lymphocyte or plasma cells by large histiocyte) which are CD-68, S-100 positive, and CD-1a negative on immunohistochemistry. At 2-year follow-up, the patient is symptom free, without any new osseous/extranodal/nodal lesions.
Conclusion: RDD rarely affects bone as a primary or even secondary form of the disease. It has relatively non-specific radiological appearance causing diagnostic challenges and may lead to erroneous diagnosis of a malignancy. Biopsy and histological examination by an experienced pathologist is mandatory for the final diagnosis. The ideal treatment ranges from wait and see policy to surgery, chemotherapy, and radiotherapy.
Keywords: Rosai-Dorfman disease, Extranodal, Osseous, Histiocytosis, Emperipolesis

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How to Cite this article: Rahman E, Sugath S, John N | A rare case of Rosai-Dorfman disease presenting as solitary lesion of distal femur: A case report. | Journal of Bone and Soft Tissue Tumors | Jan-Jun 2022; 8(1): 7-10.

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Malignant Transformation Of Solitary Phalangeal Enchondroma To High Grade Chondrosarcoma- A Case Report

Cases Series | Volume 8 | Issue 1 | JBST January – June 2022 | Page 03-06 |Ebin Rahman, Subin Sugath, Nanda Kachare.

DOI: 10.13107/jbst.2021.v08i01.65

Author: Ebin Rahman [1], Subin Sugath [1], Nanda Kachare [2]

[1] Department of Orthopaedic Oncology, Aster Medcity, Kochi, Kerala, India

[2] Department of Pathology, Aster Medcity, Kochi, Kerala, India.

Address of Correspondence
Dr. Ebin Rahman,
Department of Orthopaedic Oncology, Aster Medcity, Kochi, Kerala, India.
E-mail: rahmanebin@gmail.com


Introduction: Chondrosarcoma (CS) is a malignant neoplasm of mesenchymal origin characterized by the formation of cartilaginous matrix by neoplastic cells. It may arise as a primary lesion or occur secondarily at the site of a previous benign lesion such as enchondroma. Primary CS is rare in the hand with a frequency of <0.5–3.2% of all CS. Malignant transformation of a benign solitary enchondroma to secondary CS, especially in the hand, is extremely rare. This transformation is seen more often in multiple enchondromatosis than in solitary lesions, but even this event is rare in the hands. Here, we report a case of solitary enchondroma of proximal phalanx of middle finger initially treated with intralesional curettage and bone grafting, later recurring to high grade (Grade 3) CS.
Case Report: We report the case of 44-year-old lady presenting with pain and progressive fusiform swelling of the entire right middle finger of 4 months duration. She had a similar history 5 years back for which she had underwent intralesional curettage and bone graft substitute application. Curetted specimen was reported as enchondroma. Radiographical evaluation showed an expansile, intramedullary, and lytic lesion severe cortical destruction over proximal phalanx of the right middle finger. Magnetic resonance imaging detailed the soft-tissue component. She underwent a core needle biopsy which reported as low-grade CS arising from enchondroma. Resection of middle ray including distal 2/3 of 3rd metacarpal was performed. Excised specimen was reported as high-grade CS (Grade 3) with tumor free margins. At 2-year follow-up, she is symptom free without any local recurrence or distant lesions.
Conclusion: Diagnosis of CS in the hand can pose problems to clinicians and pathologists alike. To definitely set a diagnosis of CS in the hand, comparison of histological and radiological finding is paramount. CS are malignancies resistant to both radiation and chemotherapy. CS of the hand is characterized by local recurrence and very low metastatic potential. Hence, wide excision is the recommended approach.
Keywords: Phalanx, Enchondroma, Chondrosarcoma, High grade, Amputation.

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How to Cite this article: Rahman E, Sugath S, Kachare N. | Malignant transformation of solitary phalangeal enchondroma to high grade chondrosarcoma – A case report. | Journal of Bone and Soft Tissue Tumors | Jan-Jun 2022; 8(1): 3-6.

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Vertebral Osteosarcoma – A Report of Five Cases

Case Report | Volume 7 | Issue 3 | JBST September – December 2021 | Page 13-15 | Chitturi Ramya, Jayasree Kattoor, Narayanan Geetha, Muraleedharan Venugopal. DOI:0.13107/jbst.2021.v07i03.59

Author: Chitturi Ramya [1], Jayasree Kattoor [2], Narayanan Geetha [3], Muraleedharan Venugopal [4]

[1] Department of Pathology, NRI Medical College, Chinakakani, Andhra Pradesh, India.

[2] Department of Pathology, Regional Cancer Centre, Trivandrum, Kerala, India.

[3] Department of Medical Oncology, Regional Cancer Centre, Trivandrum, Kerala, India.

[4] Department of Radiology, Regional Cancer Centre, Trivandrum, Kerala, India.

Address of Correspondence
Dr. Jayasree Kattoor,
Department of Pathology, Regional Cancer Centre, Trivandrum, Kerala, India.
E-mail: jayasreeramdas@gmail.com


Background: Vertebral osteosarcoma is rare, accounting for 3%–5% of all osteosarcomas. It tends to occur in a slightly older age group. The prognosis is poor compared to osteosarcoma of extremities.
Case Report: We present five cases of vertebral osteosarcomas. The patient’s age ranged from 16 years to 54 years. There were four females and one male. They presented with pain, swelling, or weakness of limbs. Destructive lesions, mixed lytic and sclerotic lesions involving vertebral bodies, and/or pedicles were seen on imaging. A definitive diagnosis could not be made in two cases. On histopathological examination, all the cases turned out to be osteosarcomas.
Conclusion: Osteosarcoma of vertebrae has high rate of recurrence, metastasis, and mortality. Differentiation of vertebral osteosarcoma from other common bony lesions involving the spine is of utmost importance as the treatment is entirely different. Combination therapies including surgery, radiation, and chemotherapy achieve adequate short-term survival rates for vertebral osteosarcoma.

Keywords: Osteosarcoma, Spine, Vertebra.


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How to Cite this article: Ramya C, Kattoor J, Geetha N, Venugopal M | Vertebral Osteosarcoma – A Report of Five Cases. | Journal of Bone and Soft Tissue Tumors | Sep-Dec 2021; 7(3): 13-15.

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Trabecular Metal Augmented Reconstruction of Acetabular Defect after Removal of Periacetabular Giant Cell Tumors in A Young Male: A Case Report with Review of Literature

Case Report | Volume 7 | Issue 3 | JBST September – December 2021 | Page 8-12 | Apurv Gabrani, Hitesh Dawar, Surbhit Rastogi, Deepak Raina. DOI:0.13107/jbst.2021.v07i03.57

Author: Apurv Gabrani [1], Hitesh Dawar [1], Surbhit Rastogi [1], Deepak Raina[1]

[1] Department of Orthopedic, Indian Spinal Injuries Centre, Vasant Kunj, New Delhi, India

Address of Correspondence
Dr. Apurv Gabrani,
Department of Orthopedic, Indian Spinal Injuries Centre, Vasant Kunj, New Delhi, India.
E-mail: apurvgabrani@gmail.com


Introduction: Giant cell tumors (GCTs) are benign, epiphyseal, locally aggressive bone tumors. Pelvic GCTs are sparingly encountered and present a challenging scenario for the surgeon to not only diagnose but also to treat appropriately. Multiple treatment options are available as per the literature but no single approach is universally accepted. Moreover, due to the complex anatomy of the pelvis, novel treatment options have also been tried.
Case Report: A 30-year-old male presented to the outpatient clinic with complaints of gradually progressive dull aching pain in his right hip for 6 months. On radiological investigation, he was found to have a lytic lesion in the right periacetabular region. Image-guided biopsy revealed GCT on histopathological examination. The final management included extended curettage of the tumor and reconstruction of the remaining defect with trabecular metal (TM) augment supplemented with reconstruction plate. The patient resumed full activity and remains asymptomatic and disease free at 5 years follow-up.
Conclusion: To the best of our knowledge, this is the first reported case for the use of TM augment in reconstruction of bone defect following curettage for GCT in pelvis.

Keywords: Giant cell tumor pelvis, trabecular metal, acetabular reconstruction.


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11. Zheng K, Yu X, Hu Y, Wang Z, Wu S, Ye Z. Surgical treatment of pelvic giant cell tumor: Amulti-center study. World J Surg Oncol 2016;14:104.
12. Zheng K, Wang Z, Wu S, Ye Z, Xu S, Xu M, et al. Giant cell tumor of the pelvis: Asystematic review. Orthop Surg 2015;7:102-7.

13. Breitenseher M, Dominkus M, Scharitzer M, Lechner M, Trieb K, Imhof H, et al. Diagnostic imaging of giant cell tumors. Radiologe 2001;41:568.
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15. Goto T, Lijima T, Kawano H, Yamamoto A, Arai M, Matsuda K, et al. Serum acid phosphatase as a tumour marker in giant cell tumuor of bone. Arch Orthop Trauma Surg 2001;121:411-3.
16. Caudell JJ, Ballo MT, Zagars GK, Lewis VO, Weber KL, Lin PP, et al. Radiotherapy in the management of giant cell tumor of bone. Int J Radiat Oncol Biol Phys 2003;57:158-65.
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22. Patradul A, Kitidurmrongsook P, Parkpian V, Ngarmukos C. Allograft replacement in giant cell tumor of the hand. Hand Surg 2001;6:59-65.

23. Meneghini RM, Meyer C, Buckley CA, Hanssen AD, Lewallen DG. Mechanical stability of novel highly porous metal acetabular components in revision total hip arthroplasty. J Arthroplasty 2010;25:337-41.
24. Grappiolo G, Loppini M, Longo UG, Traverso F, Mazziotta G, Denaro V. Trabecular metal augments for the management of paprosky Type III defects without pelvic discontinuity. J Arthroplasty 2015;30:1024-9.
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How to Cite this article: Gabrani A, Dawar H, Rastogi S, Raina D | Trabecular metal augmented reconstruction of acetabular defect after removal of periacetabular giant cell tumors in a young male: A case report with review of literature. | Journal of Bone and Soft Tissue Tumors | Sep-Dec 2021; 7(3): 8-12.

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Elderly patients with pathological fractures in distal third femur treated with intramedullary nailing: A case series

Case Report | Volume 7 | Issue 3 | JBST September – December 2021 | Page 5-7 | Neetin P Mahajan, Pranay Pravin Kondewar, Sudip Subhash Chavan, Habung Chobing, Lalkar Gadod. DOI:10.13107/jbst.2021.v07i03.55

Author: Neetin P Mahajan [1], Pranay Pravin Kondewar[1], Sudip Subhash Chavan[1], Habung Chobing[1], Lalkar Gadod [1]

[1] Department of Orthopaedics, Grant Govt. Medical College and Sir J.J. Hospital, Mumbai, Maharashtra, India.

Address of Correspondence
Dr. Pranay Pravin Kondewar,
Department of Orthopaedics, Grant Govt. Medical College and Sir J.J. Hospital, Mumbai, Maharashtra, India.
E-mail: pranaypk1@gmail.com


Introduction: Pathological fractures of femur are rare and are difficult to manage. Proximal weight bearing portion is more common site for these fractures and are least common in distal third of shaft. Depending on the location and origin of primary tumor or benign lesion, we decide the management. At risk, bone with impending fracture can be managed conservatively. Displaced fractures need surgical intervention. MIREL scoring is used to decide plan of management. Closed technique like intramedullary nailing is preferred over the open technique and gives good results without post-operative morbidity. Radiological investigations are necessary to find out the primary tumor source in metastatic lesions these includes plain radiographs, CT scan, MRI and PET scan. Lab investigations also aid in finding source of primary tumor.
Case Report: This is a case series of 3 patients who had a pathological fracture of the distal third shaft of femur. These patients had metastasis from primary tumor elsewhere in body. All the patients were operated with intramedullary nailing and fixation was done with the aim of reducing pain, avoiding complications such as bed sore, infection, deep venous thrombosis, and regaining the functional outcome as much as possible and improves the quality of life.
Conclusion: All 3 patients with a pathological femur fracture operated with intramedullary nailing were shown to have a significant reduction of pain, regaining of the range of motion of the knee joint and a better quality of life with improvement in performance of activities of daily living.

Keywords: Bony metastasis, pathological fracture, intra-medullary nail, palliative surgery.


1. Feng H, Wang J, Xu J, Chen W, Zhang Y. The surgical management and treatment of metastatic lesions in the proximal femur: A mini review. Medicine (Baltimore) 2016;95:e3892.
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3. Coleman RE. Bone cancer in 2011: Prevention and treatment of bone metastases. Nat Rev Clin Oncol 2012;9:76-8.
4. Yin JJ, Selander K, Chirgwin JM, Dallas M, Grubbs BG, Wieser R, et al. TGF-beta signaling blockade inhibits PTHrP secretion by breast cancer cells and bone metastases development. J Clin Invest 1999;103:197-206.
5. Piccioli A, Ventura A, Maccauro G, Spinelli MS, Del Bravo V, Rosa MA. Local adjuvants in surgical management of bone metastases. Int J Immunopathol Pharmacol 2011;24 Suppl 1:129-32.
6. Heisterberg L, Johansen TS. Treatment of pathological fractures. Acta Orthop Scand 1979;50:787-90.
7. Feng H, Feng J, Li Z, Feng Q, Zhang Q, Qin D, et al. Percutaneous femoroplasty for the treatment of proximal femoral metastases. Eur J Surg Oncol 2014;40:402-5.
8. Hunt KJ, Gollogly S, Randall RL. Surgical fixation of pathologic fractures: An evaluation of evolving treatment methods. Bull Hosp Jt Dis 2006;63:77-82.
9. Ruggieri P, Mavrogenis AF, Casadei R, Errani C, Angelini A, Calabrò T, et al. Protocol of surgical treatment of long bone pathological fractures. Injury 2010;41:1161-7.
10. Willeumier JJ, van der Linden YM, van de Sande MA, Dijkstra PS. Treatment of pathological fractures of the long bones. EFORT Open Rev 2016;1:136-45.
11. Sarahrudi K, Greitbauer M, Platzer P, Hausmann JT, Heinz T, Vécsei V. Surgical treatment of metastatic fractures of the femur: A retrospective analysis of 142 patients. J Trauma 2009;66:1158-63.

How to Cite this article: Mahajan NP, Kondewar PP, Chavan SS, Gadod L | Elderly patients with pathological fractures in distal third femur treated with intramedullary nailing: A case series. | Journal of Bone and Soft Tissue Tumors | Sep-Dec 2021; 7(3): 5-7.

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Intraosseous Schwannoma of the Thoracic Spine: A Case Report

Case Report | Volume 7 | Issue 3 | JBST September – December 2021 | Page 2-4 | Ikuo Kudawara, Hiroyuki Aono. DOI: DOI:10.13107/jbst.2021.v07i03.53

Author: Ikuo Kudawara[1], Hiroyuki Aono[1]

[1] Department of Orthopaedic Surgery, National Hospital Organization Osaka National Hospital, 2-1-14, Hoenzaka, Chuo-ku, Osaka 540-0006, Japan.

Address of Correspondence
Dr. Ikuo Kudawara,
Department of Orthopaedic Surgery, National Hospital Organization Osaka National Hospital, Japan.
E-mail: kudawara.ikuo.rf@mail.hosp.go.jp


Introduction: Primary schwannoma of the bone is extremely rare. Spinal schwannoma usually rises in the nerve root or the cauda equina and their branches that occasionally scallop on the adjacent bone. On radiology, their features often mimic those of bone tumors such as osteoblastoma, hemangioma, aneurysmal bone cyst, plasmacytoma, or bone metastasis. Therefore, the diagnosis of pure spinal schwannoma of the bone should be evaluated carefully when referring to radiological and histopathological findings.
Case Report: A 64-year-old female presented with back pain and lower leg dysesthesia. The tumor within the left lamina of the 12th thoracic spine showed an expansile and lytic lesion on computed tomography (CT) and intermediate signals on the T1-weighted image and high signals on the T2-weighted image on magnetic resonance imaging. In addition, an old compression fracture in the same vertebra was observed. ACT-guided biopsy specimen suggested schwannoma. The tumor was successfully excised, and afterward, remission of the symptoms was observed. A definitive diagnosis of intraosseous schwannoma was made. There has been no recurrence in the 6 years following surgery.
Conclusion: We have presented the radiological and histopathological findings as well as the clinical outcomes of an unusual case of intraosseous schwannoma of the posterior element of the 12th thoracic spine. Pre-operative CT-guided biopsy was useful in making a diagnosis and planning a surgical strategy.

Keywords: Schwannoma, Thoracic spine, Bone.


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How to Cite this article: Kudawara I, Aono H | Intraosseous schwannoma of the thoracic spine: A case report. | Journal of Bone and Soft Tissue Tumors | Sep-Dec 2021; 7(3): 2-4.

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