Abstract

Pure cutaneous Rosai-Dorfman is distinguished from classic Rosai-Dorfman disease by isolated skin involvement without lymphadenopathy or systemic symptoms. Herein, we report a case of a 30 year-old-man with 16 months history of a slowly enlarging, asymptomatic, purple plaque on her left cheek following cutaneous leishmaniasis successfully treated. The histopathological examination showed dense infiltration of inflammatory cells involving the entire dermis, consisting of large macrophages with emperipolesis, S100 and CD68 positive, lymphocytes and plasma cells. The patient was treated with intralesional triamcinolone with a significant improvement. The present article aimed to emphasise the clinical, histological differential diagnosis and to share the tumoral presentation of cutaneous Rosai-Dorfman disease.

Highlights

  • Rosai- Dorfman disease, known as sinus histiocytosis with massive lymphadenopathy is a rare benign proliferative disorder of histiocytes

  • We present on exceptionally florid case of purely cutaneous Rosai-Dorfman disease following cutaneous leishmaniasis and succinctly review the clinical and histologic features, differential diagnosis, and course

  • Infectious agents, including human herpesvirus (HHV) 6, Epstein-Barr virus (EBV), parvovirus B19, herpes simplex virus, Brucella, Klebsiella rhinoscleromatis, and Nocardia have been implicated on the basis of temporal association and positive serology in some affected patients [4]

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Summary

INTRODUCTION

Rosai- Dorfman disease, known as sinus histiocytosis with massive lymphadenopathy is a rare benign proliferative disorder of histiocytes. A 30 year-old man presented with 16 months history of a slowly enlarging, asymptomatic, purple plaque on her left cheek. He denied insect bite and traumatic injury. He had a past history of cutaneous leishmaniasis over her left cheek treated with systemic meglumine antimoniate therapy successfully. Physical examination revealed a 5*7cm, violaceous plaque studded with multiple, soft, reddish brown papular and nodular lesions with hummocky surface and lymphomatouslike appearance over left cheek (Fig. 1). He had no mucosal involvement and no lymphadenopathy.

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