{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T08:04:54Z","timestamp":1761293094244,"version":"build-2065373602"},"reference-count":19,"publisher":"MDPI AG","issue":"11","license":[{"start":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T00:00:00Z","timestamp":1761264000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Current Oncology"],"abstract":"<jats:p>Primary meningeal melanocytoma is an uncommon, pigmented neoplasm that rarely undergoes malignant transformation, and therapeutic guidelines remain undefined. We report a 43-year-old woman who initially presented with a sudden headache and a right temporal intraparenchymal mass. Subtotal resection revealed a melanocytoma (WHO grade I); residual tumor was treated with Gamma Knife. About 15 months later, she deteriorated rapidly due to malignant transformation with cerebral hemorrhage and spinal leptomeningeal metastasis. Pembrolizumab was initiated within four weeks of the malignant diagnosis and produced transient neurological improvement. Due to symptomatic progression, ipilimumab plus nivolumab was commenced and achieved temporary radiographic stabilization, but the patient succumbed to diffuse progression later. Including this case, only five intracranial melanocytomas with malignant transformation treated with immune checkpoint inhibitors have been reported. Our experience supports initiating immunotherapy promptly after malignant transformation and suggests that sequential dual-agent blockade may modestly extend disease control.<\/jats:p>","DOI":"10.3390\/curroncol32110595","type":"journal-article","created":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T07:41:35Z","timestamp":1761291695000},"page":"595","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Primary Intracranial Meningeal Melanocytoma with Malignant Transformation: A Case Report and Comparison of Early Versus Late Immunotherapy Interventions"],"prefix":"10.3390","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-1949-2758","authenticated-orcid":false,"given":"Yi-Qi","family":"Zhang","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, E-Da Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kun-Ming","family":"Rau","sequence":"additional","affiliation":[{"name":"Department of Oncology, E-Da Cancer Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cheng-Loong","family":"Liang","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, E-Da Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yu-Duan","family":"Tsai","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, E-Da Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"He-Tai","family":"Jheng","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, E-Da Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kuo-Wei","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, E-Da Cancer Hospital, Kaohsiung 824005, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,10,24]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"528","DOI":"10.3171\/jns.2004.101.3.0528","article-title":"Transition from meningeal melanocytoma to primary cerebral melanoma: Case report","volume":"101","author":"Roser","year":"2004","journal-title":"J. Neurosurg."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"349","DOI":"10.2176\/nmccrj.cr.2020-0191","article-title":"Primary Meningeal Melanocytoma Located in the Craniovertebral Junction: A Case Report and Literature Review","volume":"8","author":"Uramaru","year":"2021","journal-title":"NMC Case Rep. J."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"360","DOI":"10.1007\/s10354-005-0199-8","article-title":"Spinale und zerebrale Meningeosis eines sekund\u00e4r malignisierten Melanozytoms des Kleinhirnbr\u00fcckenwinkels","volume":"155","author":"Koch","year":"2005","journal-title":"Wien. Med. Wochenschr."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1007\/s11060-010-0311-9","article-title":"Black brain: Transformation of a melanocytoma with diffuse melanocytosis into a primary cerebral melanoma","volume":"102","author":"Gempt","year":"2011","journal-title":"J. Neurooncol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1016\/j.yexmp.2016.12.006","article-title":"Copy number variation analysis and methylome profiling of a GNAQ-mutant primary meningeal melanocytic tumor and its liver metastasis","volume":"102","author":"Kruse","year":"2017","journal-title":"Exp. Mol. Pathol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"991","DOI":"10.1067\/mjd.2000.109282","article-title":"Prognostic value of Ki-67 expression in localized cutaneous malignant melanoma","volume":"43","author":"Henrique","year":"2000","journal-title":"J. Am. Acad. Dermatol."},{"key":"ref_7","doi-asserted-by":"crossref","unstructured":"Liu, Q., Peng, Z., Shen, L., and Shen, L. (2021). Prognostic and Clinicopathological Value of Ki-67 in Melanoma: A Meta-Analysis. Front. Oncol., 11.","DOI":"10.3389\/fonc.2021.737760"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"2153","DOI":"10.4103\/0028-3886.359256","article-title":"Meningeal Melanocytoma\u2014A Clinicohistological Masquerader","volume":"70","author":"Chhabra","year":"2022","journal-title":"Neurol. India"},{"key":"ref_9","first-page":"11698","article-title":"A case report on intermediate-grade malignant meningeal melanocytoma with elevated cell proliferation index","volume":"8","author":"Sun","year":"2015","journal-title":"Int. J. Clin. Exp. Pathol."},{"key":"ref_10","doi-asserted-by":"crossref","unstructured":"Pellerino, A., Verdijk, R.M., Nichelli, L., Andratschke, N.H., Idbaih, A., and Goldbrunner, R. (2024). 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). Cancers, 16.","DOI":"10.3390\/cancers16142508"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"209","DOI":"10.1111\/bpa.12241","article-title":"Primary melanocytic tumors of the central nervous system: A review with focus on molecular aspects","volume":"25","author":"Grunsven","year":"2015","journal-title":"Brain Pathol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1186\/s42047-024-00171-3","article-title":"PRAME immunohistochemistry distinguishes nodal nevi from metastatic melanoma","volume":"7","author":"Junior","year":"2024","journal-title":"Surg. Exp. Pathol."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1097\/PAS.0000000000001393","article-title":"Immunohistochemistry for PRAME in the Distinction of Nodal Nevi From Metastatic Melanoma","volume":"44","author":"Lezcano","year":"2020","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"155162","DOI":"10.1016\/j.prp.2024.155162","article-title":"Beyond typical histology of BAP1-inactivated melanocytoma","volume":"259","author":"Donati","year":"2024","journal-title":"Pathol. Res. Pract."},{"key":"ref_15","doi-asserted-by":"crossref","unstructured":"Wang, J.-W., Feng, Y.-F., and Liu, J.-H. (2025). CTLA-4 and PD-1 combined blockade therapy for malignant melanoma brain metastases: Mechanisms, challenges, and prospects. Front. Immunol., 16.","DOI":"10.3389\/fimmu.2025.1629879"},{"key":"ref_16","doi-asserted-by":"crossref","unstructured":"Pilch, J., Mizera, J., Tota, M., and Donizy, P. (2024). GNAQ\/GNA11-Related Benign and Malignant Entities\u2014A Common Histoembriologic Origin or a Tissue-Dependent Coincidence. Cancers, 16.","DOI":"10.3390\/cancers16213672"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"1327","DOI":"10.1038\/ng.3677","article-title":"Recurrent SERPINB3 and SERPINB4 mutations in patients who respond to anti-CTLA4 immunotherapy","volume":"48","author":"Riaz","year":"2016","journal-title":"Nat. Genet."},{"key":"ref_18","first-page":"204","article-title":"Intrathecal immunotherapy in patients who have melanoma with leptomeningeal disease","volume":"23","author":"Oliva","year":"2025","journal-title":"Clin. Adv. Hematol. Oncol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"898","DOI":"10.1038\/s41591-022-02170-x","article-title":"Concurrent intrathecal and intravenous nivolumab in leptomeningeal disease: Phase 1 trial interim results","volume":"29","author":"Oliva","year":"2023","journal-title":"Nat. Med."}],"container-title":["Current Oncology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1718-7729\/32\/11\/595\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T08:01:27Z","timestamp":1761292887000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1718-7729\/32\/11\/595"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10,24]]},"references-count":19,"journal-issue":{"issue":"11","published-online":{"date-parts":[[2025,11]]}},"alternative-id":["curroncol32110595"],"URL":"https:\/\/doi.org\/10.3390\/curroncol32110595","relation":{},"ISSN":["1718-7729"],"issn-type":[{"value":"1718-7729","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,10,24]]}}}