{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T05:11:30Z","timestamp":1792732290934,"version":"4.5.0"},"reference-count":85,"publisher":"MDPI AG","issue":"14","license":[{"start":{"date-parts":[[2024,7,10]],"date-time":"2024-07-10T00:00:00Z","timestamp":1720569600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cancers"],"abstract":"<jats:p>Background: Primary meningeal melanocytic tumors are ultra-rare entities with distinct histological and molecular features compared with other melanocytic or pigmented lesions, such as brain and leptomeningeal metastases from metastatic melanoma. Methods: The European Network for Rare Cancers (EURACAN) Task Force on Ultra-Rare Brain Tumors (domain 10, subdomain 10) performed a literature review from January 1985 to December 2023 regarding the epidemiologic and clinical characteristics, histological and molecular features, radiological findings, and efficacy of local treatments (surgery and radiotherapy) and systemic treatments for these entities. Results: Molecular analysis can detect specific mutations, including GNAQ, GNA11, SF3B1, EIF1AX, BAP1, that are typically found in circumscribed primary meningeal melanocytic tumors and not in other melanocytic lesions, whereas NRAS and BRAF mutations are typical for diffuse primary meningeal melanocytic tumors. The neuroimaging of the whole neuroaxis suggests a melanocytic nature of a lesion, depicts its circumscribed or diffuse nature, but cannot predict the tumor\u2019s aggressiveness. Gross-total resection is the first choice in the case of circumscribed meningeal melanocytoma and melanoma; conversely, meningeal biopsy may be reserved for patients with diffuse and multinodular leptomeningeal spread to achieve a definitive diagnosis. High-dose radiotherapy is rarely indicated in diffuse melanocytic tumors except as palliative treatment to alleviate symptoms. Last, a definitive advantage of a specific systemic treatment could not be concluded, as most of the data available derive from case reports or small cohorts. Conclusions: As primary meningeal melanocytic tumors are extremely rare, the correlations between the clinical characteristics, molecular profile, radiological findings at diagnosis and progression are weak, and poor evidence on the best therapeutic approach is available. There is a need to develop shared platforms and registries to capture more knowledge regarding these ultra-rare entities.<\/jats:p>","DOI":"10.3390\/cancers16142508","type":"journal-article","created":{"date-parts":[[2024,7,10]],"date-time":"2024-07-10T13:34:38Z","timestamp":1720618478000},"page":"2508","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":23,"title":["Primary Meningeal Melanocytic Tumors of the Central Nervous System: A Review from the Ultra-Rare Brain Tumors Task Force of the European Network for Rare Cancers (EURACAN)"],"prefix":"10.3390","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3243-0059","authenticated-orcid":false,"given":"Alessia","family":"Pellerino","sequence":"first","affiliation":[{"name":"Division of Neuro-Oncology, Department of Neuroscience \u201cRita Levi Montalcini\u201d, University and City of Health and Science Hospital, 10126 Torino, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1437-214X","authenticated-orcid":false,"given":"Robert M.","family":"Verdijk","sequence":"additional","affiliation":[{"name":"Department of Pathology, Section Ophthalmic Pathology, Erasmus MC University Medical Center, 3015 Rotterdam, The Netherlands"},{"name":"Department of Pathology, Leiden University Medical Center, 2333 Leiden, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0006-0934-5365","authenticated-orcid":false,"given":"Lucia","family":"Nichelli","sequence":"additional","affiliation":[{"name":"Department of Neuroradiology, Assistance Publique-H\u00f4pitaux de Paris, Sorbonne Universit\u00e9, Groupe Hospitalier Piti\u00e9-Salp\u00eatri\u00e8re-Charles Foix, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolaus H.","family":"Andratschke","sequence":"additional","affiliation":[{"name":"Department of Radiation Oncology, University of Zurich, University Hospital Zurich, 8091 Zurich, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5290-1204","authenticated-orcid":false,"given":"Ahmed","family":"Idbaih","sequence":"additional","affiliation":[{"name":"CNRS, Inserm, DMU Neurosciences, Service de Neuro-Oncologie-Institut de Neurologie, Sorbonne Universit\u00e9, H\u00f4pitaux Universitaires La Piti\u00e9 Salp\u00eatri\u00e8re-Charles Foix, F-75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roland","family":"Goldbrunner","sequence":"additional","affiliation":[{"name":"Center for Neurosurgery, Department of General Neurosurgery, University of Cologne, 50923 Cologne, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,7,10]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"2303","DOI":"10.1111\/ene.14437","article-title":"Primary melanotic tumors of the nervous system: A consecutive case series","volume":"27","author":"Grosshans","year":"2020","journal-title":"Eur. 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