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However, additional cancers are found more rarely in\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS patients. The aim of this study was to investigate the association, clinical, and pathologic characteristics of meningioma in\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      We conducted a retrospective chart review of meningiomas in two independent cohorts of patients with germline\n                      <jats:italic>BAP1<\/jats:italic>\n                      pathogenic or likely pathogenic (P\/LP) variants at The Ohio State University Wexner Medical Center and at the Memorial Sloan Kettering Cancer Center from October 1st, 2010 date to April 21st, 2025. Additionally, we conducted a literature review of meningioma case studies for individuals with germline\n                      <jats:italic>BAP1<\/jats:italic>\n                      (P\/LP) variants.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      In a cohort of 237 subjects with\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS, we identified 6.8% (16\/237) with a history of meningiomas. The average age of diagnosis was 44.5 years (17\u201371). For patients with available pathology, 61.5% (8\/13) of the tumors were grade 2\/3. Patients with available tumor tissue 83.3% (5\/6) showed evidence of\n                      <jats:italic>BAP1<\/jats:italic>\n                      biallelic inactivation. Family history of meningioma was reported in 18.8% (3\/16) of patients. Four cases of meningioma were identified during meningioma surveillance imaging, and five cases had recurrences after treatment. Published cases were consistent with the early age of onset, high-grade tumors, and clinical phenotype of tumors.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      This study provides additional evidence that high-grade brain and spinal meningiomas are part of the clinical spectrum of\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS. Craniospinal imaging surveillance in the\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS population should be considered starting around puberty, enabling early detection and management for individuals with\n                      <jats:italic>BAP1<\/jats:italic>\n                      -TPDS.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1007\/s11060-026-05445-2","type":"journal-article","created":{"date-parts":[[2026,2,11]],"date-time":"2026-02-11T10:59:43Z","timestamp":1770807583000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["High frequency and unique subtypes of meningioma in patients with BAP1 tumor predisposition syndrome"],"prefix":"10.1007","volume":"176","author":[{"given":"Kaylee A.","family":"Ramsey","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lindsey","family":"Byrne","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivia B.","family":"Taylor","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amr","family":"Soliman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emma","family":"Schreiner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabella","family":"Gray","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alicia","family":"Latham","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rania","family":"Sheikh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saman S.","family":"Ahmadian","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Russell R.","family":"Lonser","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua D.","family":"Palmer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria I.","family":"Carlo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Colleen M.","family":"Cebulla","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohamed H.","family":"Abdel-Rahman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,2,11]]},"reference":[{"key":"5445_CR1","doi-asserted-by":"publisher","first-page":"606","DOI":"10.1038\/s41418-020-00709-4","volume":"28","author":"L Masclef","year":"2021","unstructured":"Masclef L, Ahmed O, Estavoyer B et al (2021) Roles and mechanisms of BAP1 deubiquitinase in tumor suppression. 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