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September 15, 2022

You can use pre-built Dataflow Templates or custom Dataflow Connectors, which remove the need to write and maintain integration code yourself. Live Nation confirmed the data breach in a filing with government regulators late on Friday after the markets closed. Join 1,000+ founders and VCs for all-day bootcamp. Together, these signals show a market moving […]

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

June 10, 2022

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 […]

Professional Database Engineer Certification

September 15, 2022

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

June 10, 2022

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

June 10, 2022

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

Abstract

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

Reference:
1. Rosai J, Dorfman RF. Sinus histiocytosis with massive lymphadenopathy: A newly recognized benign clinicopathological entity. Arch Pathol 1969;87:63-70.
2. Pulsoni A, Anghel G, Falcucci P, Matera R, Pescarmona E, Ribersani M, et al. Treatment of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): Report of a case and literature review. Am J Hematol 2002;69:67-71.
3. Demicco EG, Rosenberg AE, Bjornsson J, Rybak LD, Unni KK, Nielsen GP. Primary Rosai-Dorfman disease of bone: A clinicopathologic study of 15 cases. Am J Surg Pathol 2010;34:1324-33.
4. Foucar E, Rosai J, Dorfman R. Sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease): Review of the entity. Semin Diagn Pathol 1990;7:19-73.
5. Bhandari A, Patel PR, Patel MP. Extranodal Rosai-Dorfman disease with multiple spinal lesions: Arare presentation. Surg Neurol 2006;65:308-11.
6. George J, Stacy G, Peabody T, Montag A. Rosai-Dorfman disease manifesting as a solitary lesion of the radius in a 41-year-old woman. Skeletal Radiol 2003;32:236-9.
7. Bachmann KR, Dragoescu EA, Foster WC. Extranodal Rosai-Dorfman disease presenting as incidental bone tumor: A case report. Am J Orthop (Belle Mead NJ) 2010;39:E123-5.
8. Abou-Zeid AH, Herwadkar A, du Plessis D, Gnanalingham KK. Isolated extradural Rosai-Dorfman disease of the thoracic spine: A rare cause of spinal cord compression: Case report. Neurosurgery 2010;67:E514-5.
9. Duijsens HM, Vanhoenacker FM, ter Braak BP, Hogendoorn PC, Kroon HM. Primary intraosseous manifestation of Rosai-Dorfman disease: 2 cases and review of literature. JBR-BTR 2014;97:84-9.
10. Mehraein Y, Wagner M, Remberger K, Füzesi L, Middel P, Kaptur S, et al. Parvovirus B19 detected in Rosai-Dorfman disease in nodal and extranodal manifestations. J Clin Pathol 2006;59:1320-6.
11. Lin J, Lazarus M, Wilbur A. Sinus histiocytosis with massive lymphadenopathy: MRI findings of osseous lesions. Skeletal Radiol 1996;25:279-82.
12. Ruggiero A, Attinà G, Maurizi P, Mulè A, Tarquini E, Barone G, et al. Rosai-Dorfman disease: Two case reports and diagnostic role of fineneedle aspiration cytology. J Pediatr Hematol Oncol 2006;28:103-6.
13. Walker PD, Rosai J, Dorfman RF. The osseous manifestations of sinus histiocytosis with massive lymphadenopathy. Am J Clin Pathol 1981;75:131-9.

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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