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A 55\u2010year\u2010old female presented with a 7\u2010year history of cutaneous nodules involving the trunk and extremities. A prior skin biopsy specimen at a different institution had demonstrated a dense dermal lymphohistiocytic infiltrate with histiocytes containing GMS+ forms, favored to represent cryptococcal organisms, with a differential diagnosis including other infections with parasitized histiocytes. Despite antibiotic therapy, lesions persisted. After a presentation to our institution, a biopsy specimen showed a diffuse infiltrate, including histiocytes with voluminous pale cytoplasm with focal emperipolesis of inflammatory cells and S100 immunohistochemical positivity. Clinical and radiologic examinations did not identify significant extracutaneous involvement. A genetic study performed on the biopsy specimen identified a K57Q mutation of <jats:italic>MAP2K1<\/jats:italic>. The presence of this mutation correlated with prior reports of <jats:italic>MAP2K1<\/jats:italic> mutation in classic RDD, thereby supporting our histopathologic diagnosis of RDD over an infectious process and further illuminating options for targeted therapies. At 3\u2010year follow\u2010up, the patient has been managed with a course of systemic corticosteroids and excision of bothersome lesions. Consideration of systemic therapy is ongoing.<\/jats:p>","DOI":"10.1111\/cup.14700","type":"journal-article","created":{"date-parts":[[2024,8,10]],"date-time":"2024-08-10T02:59:12Z","timestamp":1723258752000},"page":"942-947","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Cutaneous Rosai\u2013Dorfman disease with <i>MAP2K1<\/i> mutation, initially mimicking an infection with parasitized histiocytes"],"prefix":"10.1111","volume":"51","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3613-1542","authenticated-orcid":false,"given":"Joseph","family":"Gillam","sequence":"first","affiliation":[{"name":"Department of Pathology Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ruchi","family":"Desai","sequence":"additional","affiliation":[{"name":"Division of Hematology\u2010Oncology Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Raphael J.","family":"Louie","sequence":"additional","affiliation":[{"name":"Department of Surgery Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Scott A.","family":"Turner","sequence":"additional","affiliation":[{"name":"Department of Pathology Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Grace Y.","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Pathology Beaumont Hospital  Royal Oak Michigan USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Julio A.","family":"Diaz\u2010Perez","sequence":"additional","affiliation":[{"name":"Department of Pathology Virginia Commonwealth University Health System  Richmond Virginia USA"},{"name":"Department of Dermatology Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7306-8813","authenticated-orcid":false,"given":"Mark C.","family":"Mochel","sequence":"additional","affiliation":[{"name":"Department of Pathology Virginia Commonwealth University Health System  Richmond Virginia USA"},{"name":"Department of Dermatology Virginia Commonwealth University Health System  Richmond Virginia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,8,9]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41572\u2010021\u201000307\u20109"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/00000372\u2010200210000\u201000001"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2004.04.030"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097\u20100142(197211)30:5<1174::AID\u2010CNCR2820300507>3.0.CO;2\u2010S"},{"key":"e_1_2_6_6_1","unstructured":"Thermo Fisher Scientific.Oncomine Comprehensive Assay v3. 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