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A pure Cutaneous Rosai-Dorfman disease: case report and a review of the literature
Fatemeh Montazer , Saeedeh Farahani , Zoha Shaka , Zeinab Aryanian , Azadeh Goodarzi *
Department of Dermatology, Rasool Akram Medical Complex, Iran University of Medical Sciences, Tehran, Iran , azadeh_goodarzi1984@yahoo.com
Abstract:   (344 Views)
Background: Cutaneous Rosai-Dorfman disease (CRDD) is a rare variant of benign histiocytic proliferative disorder limited to the skin. The underlying etiology is still unclear, but it had been claimed that infections, immunodeficiencies, and autoimmune disorders might have a role in the etiology of this disorder. The characteristic presentation of RDD is lymphadenopathy due to abnormal production and accumulation of histiocytes in lymph nodes; however, the extra-nodal areas could also be affected, such as cutaneous.Herein we presented a 45-year-old Iranian womanpresented with an atypical pure cutaneous Rosai Dorfman disease, in addition to a summarized list of atypical cases of RDD that are reported as pure cutaneous RDD with atypical presentation.
Case presentation:  Herein we presented a 45-year-old woman referred to us with an ulcerative nodule with a size of 5×5 on her buttock, gradually growing over one year. After the primary evaluations, a biopsy specimen was obtained, and Histologic studies revealed a dense cellular infiltrate involving the dermis and the subcutis, which was composed of abundant sheets of large histiocytes with admixtures of lymphocytes, plasma cells, neutrophils, and eosinophils within their cytoplasm -known as the emperipolesis phenomenon. The immunohistochemistry staining was positive for S100 and CD68 and negative for CD1a
Conclusion: we presented a patient with an isolated ulcerative dermal-based lesion that hadtypical cytomorphologic and immunohistochemical features for pure CRDDwithout any systematic presentation. Thus, the diagnosis of CRDD was confirmed for her condition.the diagnosis of CRDD was confirmed based on these histopathological findings.

Keywords: pure cutaneous Rosai Dorfman disease, histiocytosis, atypical cutaneous presentation, non-Langerhans cell histiocytosis, review, case report, histopathology
Type of Study: Review Article | Subject: Dermathology
Received: 2021/05/29 | Accepted: 2021/11/16
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Caspian Journal of Internal Medicine
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