Byung-Rae Cho, Young Suk Lee, Dong-Sup Chung, Wan-Soo Yoon
Multicentric gliomas are rare clinical entities, and cases exhibiting distinct histological lineages despite sharing a common molecular origin are exceptionally scarce. We report a unique case of synchronous multicentric gliomas in the bilateral frontal lobes of a 59-yearold female that shared an ancestral IDH1-R132H mutation but diverged into distinct molecular trajectories. According to the 2021 World Health Organization (WHO) classification, the left frontal tumor was diagnosed as a central nervous system (CNS) WHO grade 3 oligodendroglioma characterized by 1p/19q co-deletion and CDKN2A mutation, while the right frontal tumor was identified as a CNS WHO grade 3 astrocytoma harboring ATRX and TP53 mutations. This case provides compelling evidence for the stem cell origin theory, suggesting that a common progenitor undergoes independent clonal evolution in different anatomical locations through disparate secondary genetic alterations. Our findings underscore that multicentric lesions cannot be assumed to be molecularly identical, necessitating comprehensive and individual molecular evaluation of each discrete lesion to ensure accurate prognostic stratification and the formulation of personalized therapeutic strategies in the era of integrated diagnostics.