A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences

Abstract Background Isocitrate dehydrogenase (IDH) mutations are associated with better prognosis and longer survival in gliomas. In contrast, multifocal glioma is a rare intracranial tumor, primarily affecting middle-aged and elderly individuals. While recurrence is common due to the complex tumor...

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Main Authors: Wen Li, Yuanshuai Zhou, Shaheryar Shafi, Jun Dong, Minxuan Sun
Format: Article
Language:English
Published: BMC 2025-07-01
Series:World Journal of Surgical Oncology
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Online Access:https://doi.org/10.1186/s12957-025-03913-z
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author Wen Li
Yuanshuai Zhou
Shaheryar Shafi
Jun Dong
Minxuan Sun
author_facet Wen Li
Yuanshuai Zhou
Shaheryar Shafi
Jun Dong
Minxuan Sun
author_sort Wen Li
collection DOAJ
description Abstract Background Isocitrate dehydrogenase (IDH) mutations are associated with better prognosis and longer survival in gliomas. In contrast, multifocal glioma is a rare intracranial tumor, primarily affecting middle-aged and elderly individuals. While recurrence is common due to the complex tumor microenvironment, cellular heterogeneity, and clonal evolution, multifocal recurrence originating from an initially unifocal, low-grade glioma has been rarely reported. Case presentation We present the case of a 36-year-old male initially diagnosed with oligodendroglioma, IDH-mutant and 1p/19q-codeleted, CNS WHO grade 3. After gross total resection and standard chemoradiotherapy, the patient developed multifocal recurrence after three years, with new lesions in the left temporal and frontal lobes. Histopathological analysis revealed divergent progression: the distal temporal lesion demonstrated glioblastoma-like features, whereas the in situ frontal recurrence retained characteristics of oligodendroglioma. To investigate the mechanism of multifocal tumor recurrence, we performed single-cell sequencing analysis on both foci, revealing distinct cellular compositions and hidden lineage relationships among malignant cells. Discussion and conclusion We describe a rare case of multifocal glioma recurrence in a middle-aged adult, highlighting the cellular heterogeneity and lineage relationships between distinct tumor foci. This case contributes to the limited literature on multifocal glioma recurrence and provides a well-characterized specimen for investigating the molecular distinctions between IDH-mutant and IDH-wildtype gliomas.
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spelling doaj-art-befa27f278d04dd4a2bd9b8106f3cd8e2025-08-20T03:03:37ZengBMCWorld Journal of Surgical Oncology1477-78192025-07-012311610.1186/s12957-025-03913-zA rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrencesWen Li0Yuanshuai Zhou1Shaheryar Shafi2Jun Dong3Minxuan Sun4School of Biomedical Engineering (Suzhou), Division of Life Sciences and Sciences and Medicine, University of Science and Technology of ChinaSchool of Biomedical Engineering (Suzhou), Division of Life Sciences and Sciences and Medicine, University of Science and Technology of ChinaSchool of Biomedical Engineering (Suzhou), Division of Life Sciences and Sciences and Medicine, University of Science and Technology of ChinaDepartment of Neurosurgery, The Second Affiliated Hospital of Soochow UniversitySchool of Biomedical Engineering (Suzhou), Division of Life Sciences and Sciences and Medicine, University of Science and Technology of ChinaAbstract Background Isocitrate dehydrogenase (IDH) mutations are associated with better prognosis and longer survival in gliomas. In contrast, multifocal glioma is a rare intracranial tumor, primarily affecting middle-aged and elderly individuals. While recurrence is common due to the complex tumor microenvironment, cellular heterogeneity, and clonal evolution, multifocal recurrence originating from an initially unifocal, low-grade glioma has been rarely reported. Case presentation We present the case of a 36-year-old male initially diagnosed with oligodendroglioma, IDH-mutant and 1p/19q-codeleted, CNS WHO grade 3. After gross total resection and standard chemoradiotherapy, the patient developed multifocal recurrence after three years, with new lesions in the left temporal and frontal lobes. Histopathological analysis revealed divergent progression: the distal temporal lesion demonstrated glioblastoma-like features, whereas the in situ frontal recurrence retained characteristics of oligodendroglioma. To investigate the mechanism of multifocal tumor recurrence, we performed single-cell sequencing analysis on both foci, revealing distinct cellular compositions and hidden lineage relationships among malignant cells. Discussion and conclusion We describe a rare case of multifocal glioma recurrence in a middle-aged adult, highlighting the cellular heterogeneity and lineage relationships between distinct tumor foci. This case contributes to the limited literature on multifocal glioma recurrence and provides a well-characterized specimen for investigating the molecular distinctions between IDH-mutant and IDH-wildtype gliomas.https://doi.org/10.1186/s12957-025-03913-zRecurrent gliomaMultifocal gliomascRNA-seqOligodendrocyte precursor cellsOligodendrocytes
spellingShingle Wen Li
Yuanshuai Zhou
Shaheryar Shafi
Jun Dong
Minxuan Sun
A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
World Journal of Surgical Oncology
Recurrent glioma
Multifocal glioma
scRNA-seq
Oligodendrocyte precursor cells
Oligodendrocytes
title A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
title_full A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
title_fullStr A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
title_full_unstemmed A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
title_short A rare IDH-Mutant glioma case: insights from single-cell transcriptomic analysis of tumor recurrences
title_sort rare idh mutant glioma case insights from single cell transcriptomic analysis of tumor recurrences
topic Recurrent glioma
Multifocal glioma
scRNA-seq
Oligodendrocyte precursor cells
Oligodendrocytes
url https://doi.org/10.1186/s12957-025-03913-z
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