Journal of Foot and Ankle Surgery (Asia Pacific)

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VOLUME 9 , ISSUE 1 ( January-March, 2022 ) > List of Articles

CASE REPORT

A Rare Case of Rosai-Dorfman Disease in Calcaneum and Study of Literature

Amit Gupta, Ravi Bhadiyadra, Ramesh Deshpande, Aditya Menon, Vikas M Agashe

Keywords : Brodie\'s abscess, Calcaneal osteomyelitis, Case report, Foot tumors, Heel pain, Histiocytosis, Immunohistochemical staining, Lytic, Osteoarticular tuberculosis-foot and ankle tuberculosis

Citation Information : Gupta A, Bhadiyadra R, Deshpande R, Menon A, Agashe VM. A Rare Case of Rosai-Dorfman Disease in Calcaneum and Study of Literature. J Foot Ankle Surg Asia-Pacific 2022; 9 (1):40-45.

DOI: 10.5005/jp-journals-10040-1165

License: CC BY-NC 4.0

Published Online: 31-12-2021

Copyright Statement:  Copyright © 2022; The Author(s).


Abstract

Background: Pain and osteolytic lesions on X-rays with non-specific changes on histopathology and negative culture are common scenarios; patients often treated with empirical antibiotics or anti-tuberculosis therapy (ATT), especially in regions where tuberculosis (TB) is endemic. The policy of “Diagnosis before treatment” should be the dictum in such cases. We report a rare case of Rosai-Dorfman disease (RDD) of calcaneum diagnosed by following these guidelines. Case description: A 17-year-old female presented with left heel pain for 1 year. She was diagnosed with Brodie\'s abscess of calcaneum at another facility on imaging. Empirical antibiotics were started after debridement, as cultures were negative and histopathology was not done. Symptoms recurred in 2 months. At presentation, careful clinical evaluation, the site of tenderness was found to be 2–3 cm distal to the prior incision. MRI revealed a large lesion with post-contrast enhancement. CT-guided biopsy from an appropriate site at our facility was inconclusive as there were no signs of infection or malignancy. Subsequently, thorough debridement was done using a different incision excising the biopsy scar. Tissue cultures were negative. Histopathology showed areas of dense lymphohistiocytic infiltrate with lymphocytic emperipolesis within their cytoplasm. There were no granulomas. On immunohistochemical staining, the macrophages strongly expressed S100 but not CD1a. Accordingly, a diagnosis of RDD was made. The surgical site healed with an uneventful postoperative period. She was advised protected weight-bearing for 6 weeks. Pain resolved and no further treatment had to be given. The patient was asymptomatic at a 3-year follow-up and the radiograph showed complete consolidation of the cavity with no evidence of recurrence. Conclusion: Primary intraosseous RDD is an unusual manifestation of a rare disease. Careful assessment of clinical details, inputs and help from imaging consultants, sending adequate tissue samples from appropriate sites for both culture and histopathology, and specialized staining techniques helped accurately diagnose this condition. Clinical significance: The case highlights the challenges faced in diagnosing a lytic lesion in the foot and the importance of avoiding empirical medication before obtaining a diagnosis despite negative percutaneous image-guided biopsies.


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