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Here we report a case of primary CNS HS with unusual histopathological characteristics. A 65\u2010year\u2010old woman presented with CNS HS in the left frontal lobe region, showing two distinct histological patterns. Approximately half of the lesion displayed histological characteristics typical of HS, including diffuse invasion of large round\u2010to\u2010ovoid pleomorphic cells, with mitotic figures (Ki\u201067 index: 30%) and coagulative necrotic foci. The other half exhibited prominent proliferation of histiocytic cells between the trabeculae of reactive glial cells, with rare mitotic figures (Ki\u201067 index: &lt; 1%) and no necrotic foci. There were transitions between two morphologies. The HS tumor cells and the histiocytic cells between the trabeculae of reactive glial cells possessed nearly identical histomorphologic and immunophenotypic features, although the HS tumor cells showed a more pronounced degree of cytologic atypia and mitotic activity. To our knowledge, this is the first reported case of HS with prominent proliferation of the histiocytic cells between the trabeculae of reactive glial cells. Here we present the detailed histological, immunohistochemical, and molecular findings. Investigating cases of HS may provide insight into the pathogenesis of this disease.<\/jats:p>","DOI":"10.1111\/neup.12510","type":"journal-article","created":{"date-parts":[[2018,8,30]],"date-time":"2018-08-30T06:28:56Z","timestamp":1535610536000},"page":"609-618","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Case of primary central nervous system histiocytic sarcoma with prominent proliferation of histiocytic cells between the trabeculae of reactive glial cells"],"prefix":"10.1111","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8284-567X","authenticated-orcid":false,"given":"Emiko","family":"Takahashi","sequence":"first","affiliation":[{"name":"Department of Surgical Pathology Aichi Medical University Hospital  Nagakute Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ayako","family":"Sakakibara","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine Nagoya University Hospital  Nagoya Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toyonori","family":"Tsuzuki","sequence":"additional","affiliation":[{"name":"Department of Surgical Pathology Aichi Medical University Hospital  Nagakute Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shigeo","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine Nagoya University Hospital  Nagoya Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2018,8,28]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.3960\/jslrt.53.1"},{"key":"e_1_2_8_3_1","first-page":"468","volume-title":"World Health Organization Classification of Tumours of Haematopoietic and Lymphoid Tissues","author":"Weiss 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