• Zhanzhan Zhang, Aobo Zhang, Tao Zhang, and Zongmao Zhao.
• Department of Neurosurgery, The Second Hospital of Hebei Medical University, Shijiazhuang, China.
• Medicine (Baltimore). 2022 Dec 30; 101 (52): e32553e32553.

RationaleRosai Dorfman disease is a rare benign histiocytoproliferative disorder that occurs in the intracranial area, which occurrs typically in lymph nodes. Extrapnodal Rosai Dorfman disease rarely develops in the central nervous system and is often a focal lesion based on the dura. Based on imaging and clinical symptoms, RDD may be misdiagnosed as meningioma, and some lesions can also occur in the brain parenchyma. In the case of benign disease, the final diagnosis is made by pathological tissue diagnosis. For chronic diseases, progression may be chronic or remitting and relapsing.Patient ConcernsA 54-years-old man was hospitalized after experiencing paroxic convulsions and being unconsciousness. A head magnetic resonance imaging demonstrates a strip of lesions in the right parietal lobe. No obvious abnormality is found in the laboratory data.DiagnosesWe diagnosed meningioma of right parietal lobe and secondary epilepsy, and prescribed oral sodium valproate to treat him.InterventionsThe lesion is located in the right parietal lobe on neuroimaging prior to surgery, which was taken for immunohistochemical examination.OutcomesIf it is found that immunohistochemistry reveals histiocytes are positive for CD68, S-100, but negative for CD1a, it is identified as RDD. For patients who are seizure-free following surgery, symptomatic management is used. Following parietal lesion resection, patients are seizure-free during the follow-up period (44 months).LessonsBasing on studying and summarizing relevant literatures, RDD is described in the report in terms of its diagnosis, pathology, treatment, and clinical outcome, in order to improve the diagnosis and identification of intracranial RDD by physicians.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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