ArticleNeuro-oncology2025
Molecular signatures define BAP1-altered meningioma as a distinct CNS tumor with deregulation of Polycomb repressive complex target genes.
Article in Neuro-oncology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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Who cites it
13 citing papers in PubMed.
- Clinical and Molecular Features of BAP1-Mutated Meningiomas: Case Series.Molecular carcinogenesis · 2026Article
- Modeling meningioma in vitro in the omics era.Human cell · 2026Review
- DNA methylation profiling identifies long-range epigenetic silencing of clustered protocadherins as a key determinant of meningioma progression.Nature communications · 2026Article
- Distinct molecular subgroups in pediatric and young-onset meningiomas require age-adapted risk stratification.Nature communications · 2026Article
- Optic nerve sheath meningioma exhibits neural niche-associated transcriptomic features and rare copy number variation-linked evolution.Brain pathology (Zurich, Switzerland) · 2026Article
- Comprehensive analysis of TEAD inhibition in meningioma identifies MEK and mTOR inhibition as effective combination therapies against resistant lines.bioRxiv : the preprint server for biology · 2026Article
- Splenic hamartoma in two related patients with BAP1 tumour predisposition syndrome caused by a novel germline BAP1 p.(Gly128Arg) missense variant.Familial cancer · 2026Article
- High frequency and unique subtypes of meningioma in patients with BAP1 tumor predisposition syndrome.Journal of neuro-oncology · 2026Article
- MRI and Meningioma Research: Hotspots and Trends via Bibliometric AnalysisCurrent medical imaging · 2026Article
- High Frequency and Unique Subtypes of Meningioma in Patients with BAP1 Tumor Predisposition Syndrome.medRxiv : the preprint server for health sciences · 2025Article
- Subgroup of meningiomas involving FOS and FOSB gene fusions.Nature communications · 2025Article
- Seq-ing answers: exploring meningioma biology utilizing bulk RNA-seq-based reference landscapes.Frontiers in oncology · 2025Review
- A malignant choroid plexus tumor with heterologous differentiation and BAP1 deletion suggesting choroid plexus blastoma.Free neuropathology · 2025Article
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Abstract
backgroundMeningiomas are the most common primary intracranial neoplasms, with highly variable patient outcomes. While most meningiomas are benign, a significant subset recurs postoperatively, presenting substantial treatment challenges. BAP1 gene inactivation has been suggested as a marker for aggressive meningiomas, although its precise molecular and clinical roles remain poorly understood.
methodsTo comprehensively investigate BAP1-altered meningiomas, we used six meningiomas with known BAP1 alterations as a discovery set. Genome-wide DNA methylation profiling of these samples, along 11 151 reference meningiomas, identified a distinct molecular cluster (n = 42) using unsupervised visualization approaches. These tumors were further characterized by DNA/RNA sequencing, histopathological examination, and a retrospective review of clinical data, compared to reference meningioma cohorts, providing a thorough characterization of this rare tumor subtype.
resultsOur integrative analysis revealed BAP1-altered meningiomas as a distinct CNS tumor subtype, characterized by recurrent loss of chromosome 3p21 and driven by various BAP1-inactivating alterations. Although rhabdoid morphology is present in some cases, it is not exclusive and should not be used as a grading criterion. Progression-free survival analysis showed a median of 21 months (95% CI: 12-NA), with a 2-year overall survival rate of 79% (95% CI: 60%-100%), highlighting the aggressive nature of these tumors. Gene expression profiling revealed upregulation of PRC target genes, dysregulated Polycomb signaling, and elevated expression in several cellular and growth factor pathways.
conclusionsBAP1-altered meningiomas represent a distinct and aggressive CNS tumor subtype associated with PRC dysregulation and recurrent 3p chromosome loss. These findings support the designation "meningioma, BAP1-altered."
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