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3 "Changhee Park"
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Sarcoma
Case Series of Soft Tissue Sarcoma Patients with Brain Metastasis with Implications from Genomic and Transcriptomic Analysis
Changhee Park, Rokhyun Kim, Jaeyong Choi, Miso Kim, Tae Min Kim, Ilkyu Han, Jong-Il Kim, Han-Soo Kim
Cancer Res Treat. 2024;56(2):665-674.   Published online September 27, 2023
DOI: https://doi.org/10.4143/crt.2023.864
AbstractAbstract PDFSupplementary MaterialPubReaderePub
Purpose
Brain metastasis rarely occurs in soft tissue sarcoma (STS). Here, we present five cases of STS with brain metastases with genetic profiles.
Materials and Methods
We included five patients from Seoul National University Hospital who were diagnosed with STS with metastasis to the brain. Tissue from the brain metastasis along with that from the primary site or other metastases were used for DNA and RNA sequencing to identify genetic profiles. Gene expression profiles were compared with sarcoma samples from The Cancer Genome Atlas.
Results
The overall survival after diagnosis of brain metastasis ranged from 2.2 to 34.3 months. Comparison of mutational profiles between brain metastases and matched primary or other metastatic samples showed similar profiles. In two patients, copy number variation profiles between brain metastasis and other tumors showed several differences including MYCL, JUN, MYC, and DDR2 amplification. Gene ontology analysis showed that the group of genes significantly highly expressed in the brain metastasis samples was enriched in the G-protein coupled receptor activity, structural constituent of chromatin, protein heterodimerization activity, and binding of DNA, RNA, and protein. Gene set enrichment analysis showed enrichment in the pathway of neuroactive ligand-receptor interaction and systemic lupus erythematosus.
Conclusion
The five patients had variable ranges of clinical courses and outcomes. Genomic and transcriptomic analysis of STS with brain metastasis implicates possible involvement of complex expression modification and epigenetic changes rather than the addition of single driver gene alteration.
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Real-World Clinical Outcomes and Prognostic Factors for Patients with Advanced Angiosarcoma who Received Systemic Treatment
Changhee Park, Miso Kim, Yoonjin Kwak, Kyung Chul Moon, Se Hyun Kim, Bhumsuk Keam, Yu Jung Kim, Tae Min Kim, Dong-Wan Kim
Cancer Res Treat. 2021;53(4):1195-1203.   Published online February 1, 2021
DOI: https://doi.org/10.4143/crt.2020.1337
AbstractAbstract PDFSupplementary MaterialPubReaderePub
Purpose
Angiosarcoma is a highly aggressive mesenchymal tumor. Although systemic chemotherapy is often considered for the inoperable or metastatic angiosarcoma, the outcome of such treatment is unsatisfactory and poorly delineated.
Materials and Methods
We reviewed electronic medical records of 75 patients with angiosarcoma who were treated with systemic chemotherapy for inoperable or metastatic disease. Patients were classified as having liver involvement if they had either primary or metastatic hepatic lesions.
Results
Among the patients evaluated, 51 patients were male (68%) and 24 patients (32%) had primary cutaneous angiosarcoma. Liver involvement was present in 28 patients (37.3%). A total of 59 patients received first-line weekly paclitaxel (wPac) and showed an objective response rate (ORR) of 23.7% (n=14), a median progression free survival (mPFS) of 4.0 months (95% confidence interval [CI] 3.0–6.1), and a median overall survival (mOS) of 10.2 months (95% CI 7.0–14.6). Among patients without liver involvement, patients receiving wPac (n=35) had significantly prolonged mPFS (5.8 vs. 3.2 months, respectively, p=0.014) with a tendency for prolonged mOS (13.8 vs. 11.6 months, respectively, p=0.13) than those receiving other regimens (n=12). A total of 24 patients received second- or later-line pazopanib monotherapy and showed an ORR of 16.7% (n=4), a mPFS of 2.4 months (95% CI 1.8–4.3) and a mOS of 5.4 months (95% CI 3.5–NA).
Conclusion
Treatment with first-line wPac and later-line pazopanib seems to provide survival benefit, especially for patients with advanced angiosarcoma without liver involvement.

Citations

Citations to this article as recorded by  
  • A systematic review and meta-analysis of pazopanib efficacy and adverse effects in sarcomas
    Fernanda Picozzi, Alessandro Ottaiano, Antonella Lucia Marretta, Mariachiara Santorsola, Lucia Cannella, Antonio Pizzolorusso, Francesco Caraglia, Giuseppina Della Vittoria Scarpati, Alessandra Bracigliano, Ottavia Clemente, Ines Simeone, Michele Caraglia
    Journal of Translational Medicine.2026;[Epub]     CrossRef
  • Radiation-Induced Angiosarcoma Following Breast Surgery: A Case Report
    Mutsumi Hayashi, Yukino Kobayashi, Ryoko Semba, Kanako Ogura, Fumi Murakami, Kiyomi Kimura
    Surgical Case Reports.2026; 12(1): n/a.     CrossRef
  • Combination of Immune Checkpoint Inhibitors and Tyrosine Kinase Inhibitors for the Treatment of Late Stage Diffuse Primary Hepatic Angiosarcoma: a Case Report and Literature Review
    Ruolin Lyu, Hongyang Shu, Xiaofan Wu, Yiwen Liu, Ming Wang
    SN Comprehensive Clinical Medicine.2026;[Epub]     CrossRef
  • Angiosarcoma in a soft tissue sarcoma cohort: real-world patterns and outcomes
    Kübra Canaslan, Özge Yetginoğlu, Hasan Oğuz Çetinayak, Emine Burçin Tuna, Tuğba Yavuzşen
    Frontiers in Oncology.2026;[Epub]     CrossRef
  • Punch Biopsy as a Diagnostic Keystone for Metastatic Cardiac Angiosarcoma Treated With Anthracycline-Based Chemotherapy: A Case Report
    Aonghus Joyce, Gráinne Murphy, Cynthia C Heffron, David Aherne, Richard M Bambury
    Cureus.2025;[Epub]     CrossRef
  • Clinicopathological features, treatment outcomes, and prognostic factors of angiosarcoma: a 21-year experience at one center
    Chenyan Fang, Xiaoting Zeng, Qing Ji, Tao Zhu, Meiyu Fang, Jun Cao
    Orphanet Journal of Rare Diseases.2025;[Epub]     CrossRef
  • Secondary Publication of Japanese Dermatological Association Guidelines: Clinical Questions of Guidelines for Cutaneous Angiosarcoma 2025
    Yasuhiro Fujisawa, Yasuo Yoshioka, Koji Yoshino, Taku Fujimura, Yoichi Naito, Mamiko Masuzawa, Kohei Oashi, Eiji Nakano, Hiroshi Kato, Yusuke Muto, Hiroshi Koga, Tomomitsu Miyagaki, Hiroshi Uchi, Yasuhiro Nakamura, Kenji Asagoe
    The Journal of Dermatology.2025;[Epub]     CrossRef
  • Primary Jejunal Angiosarcoma
    Michelle Mai, Sapana Gupta, Kanhai Farrakhan, Shaolei Lu, Paul Akerman, May Min
    ACG Case Reports Journal.2025;[Epub]     CrossRef
  • 改訂版 皮膚がん診療ガイドラインと皮膚がん取扱い規約 皮膚血管肉腫
    康弘 藤澤
    Skin Cancer.2025; 40(2): 80.     CrossRef
  • Chest Wall Angiosarcoma with an Initial Presentation of Multiple Lung Metastases: a Case Report
    Hitoshi Suzuki, Mari Shinoda, Daisuke Ito, Shin Shomura, Kentaro Inoue, Kazuo Fukutome, Akira Shimamoto, Hisamichi Yuda
    Haigan.2024; 64(7): 917.     CrossRef
  • Hepatic Angiosarcoma with Peliosis Hepatis
    Kensuke Kitsugi, Kazuhito Kawata, Moe Matsumoto, Masahiro Umemura, Tomohiko Hanaoka, Maho Yamashita, Shingo Takatori, Jun Ito, Kazuyoshi Ohta, Takeshi Chida, Hidenao Noritake, Takafumi Suda
    Internal Medicine.2023; 62(8): 1157.     CrossRef
  • Conversion surgery for recurrent hepatic angiosarcoma after systemic chemotherapy with paclitaxel
    Yuta Ushida, Takafumi Sato, Tomotaka Kato, Yasuyuki Shigematsu, Hiromichi Ito, Takeshi Suzuki, Yosuke Inoue, Yoshihiro Ono, Atsushi Oba, Yu Takahashi
    Clinical Journal of Gastroenterology.2022; 15(2): 427.     CrossRef
  • Management of Cutaneous Angiosarcoma: an Update Review
    Siwei Bi, Ai Zhong, Xiya Yin, Jingyi Li, Ying Cen, Junjie Chen
    Current Treatment Options in Oncology.2022; 23(2): 137.     CrossRef
  • Results of NC-6300 (Nanoparticle Epirubicin) in an Expansion Cohort of Patients with Angiosarcoma
    Richard F Riedel, Victoria Chua, Ania Moradkhani, Natalie Krkyan, Amir Ahari, Atsushi Osada, Sant P Chawla
    The Oncologist.2022; 27(10): 809.     CrossRef
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  • 191 Download
  • 10 Web of Science
  • 14 Crossref
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CNS cancer
Clinical and Genomic Characteristics of Adult Diffuse Midline Glioma
Changhee Park, Tae Min Kim, Jeong Mo Bae, Hongseok Yun, Jin Wook Kim, Seung Hong Choi, Soon-Tae Lee, Joo Ho Lee, Sung-Hye Park, Chul-Kee Park
Cancer Res Treat. 2021;53(2):389-398.   Published online November 9, 2020
DOI: https://doi.org/10.4143/crt.2020.694
AbstractAbstract PDFSupplementary MaterialPubReaderePub
Purpose
The treatment outcomes and genomic profiles of diffuse midline glioma (DMG) in adult patients are rarely characterized. We performed a retrospective study to evaluate the clinicogenomic profiles of adult patients with brain DMG.
Materials and Methods
Patients aged ≥ 18 years diagnosed with brain DMG at Seoul National University Hospital were included. The clinicopathological parameters, treatment outcomes, survival, and genomic profiles using 82-gene targeted next-generation sequencing (NGS) were analyzed. The 6-month progression-free survival (PFS6) after radiotherapy and overall survival (OS) were evaluated.
Results
Thirty-three patients with H3-mutant brain DMG were identified. The median OS from diagnosis was 21.8 months (95% confidence interval [CI], 13.2 to not available [NA]) and involvement of the ponto-medullary area tended to have poor OS (median OS, 20.4 months [95% CI, 9.3 to NA] vs. 43.6 months [95% CI, 18.2 to NA]; p=0.07). Twenty-four patients (72.7%) received radiotherapy with or without temozolomide. The PFS6 rate was 83.3% (n=20). Patients without progression at 6 months showed significantly prolonged OS compared with those with progression at 6 months (median OS, 24.9 months [95% CI, 20.4 to NA] vs. 10.8 months [95% CI, 4.0 to NA]; p=0.02, respectively). Targeted NGS was performed in 13 patients with DMG, among whom nine (69.2%) harbored concurrent TP53 mutation. Two patients (DMG14 and DMG23) with PIK3CAR38S+E545K and KRASG12A mutations received matched therapies. Patient DMG14 received sirolimus with a PFS of 8.4 months.
Conclusion
PFS6 after radiotherapy was associated with prolonged survival in adult patients with DMG. Genome-based matched therapy may be an encouraging approach for progressive adult patients with DMG.

Citations

Citations to this article as recorded by  
  • Clinical, molecular characteristics, and telomere-maintaining mechanism of spinal cord glioma with or without H3 K27M mutation: a prognostic and experimental study
    Haoxuan Huang, Yue Gou, Yirui Kuang, Chunbo Liu, Changwu Wu, Jun Tan
    International Journal of Surgery.2026; 112(2): 2858.     CrossRef
  • Prevalence and Clinicoradiopathological Characterization of H3 K27-Altered Diffuse Midline Gliomas in Adults—A Retrospective Observational Study
    Kristof Babarczy, Bence L. Radics, Lili Kiss, Alexandra Graczer, Bence Nagy, Sandor Dosa, Gyongyi Kelemen, Marton Balazsfi, Pal Barzo, Andras Voros, Peter Klivenyi, Levente Szalardy
    Pathophysiology.2026; 33(1): 21.     CrossRef
  • Clinical and molecular characteristics of thalamic gliomas in adults: prognostic impact of TERT promoter mutation
    Shunsuke Tsuzuki, Yoshihiro Muragaki, Masayuki Nitta, Kenta Masui, Takashi Komori, Taiichi Saito, Takashi Maruyama, Shunichi Koriyama, Buntou Ro, Takakazu Kawamata
    Neurosurgical Review.2026;[Epub]     CrossRef
  • Choosing Wisely between Radiotherapy Dose-Fractionation Schedules: The Molecular Graded Prognostic Assessment for Elderly Glioblastoma Patients
    Hye In Lee, Jina Kim, In Ah Kim, Joo Ho Lee, Jaeho Cho, Rifaquat Rahman, Geoffrey Fell, Chan Woo Wee, Hong In Yoon
    Cancer Research and Treatment.2025; 57(2): 378.     CrossRef
  • H3F3B p.K27I-mutant diffuse midline glioma is a distinct subtype of H3K27-altered diffuse midline glioma
    Lei Cheng, Min Zhou, Tao Luo, Rongfang Dong, Ni Chen, Xinyong Cai, Xingwen Wang, Hao Wu, Zan Chen, Zuowei Wang, Xueling Qi, Dehong Lu, Lianghong Teng, Fengzeng Jian, Leiming Wang
    Acta Neuropathologica Communications.2025;[Epub]     CrossRef
  • Prognostic modeling for diffuse midline glioma: development and validation of a risk stratification nomogram using SEER and institutional cohorts
    Ge Zhang, Huandi Zhou, Wanyue Han, Lei Lou, Xiaoying Xue
    Frontiers in Oncology.2025;[Epub]     CrossRef
  • The role of adjuvant chemotherapy in patients with H3K27 altered diffuse midline gliomas: a multicentric retrospective study
    Vincenzo Di Nunno, Giuseppe Lombardi, Matteo Simonelli, Giuseppe Minniti, Angela Mastronuzzi, Valentina Di Ruscio, Martina Corrà, Marta Padovan, Marta Maccari, Mario Caccese, Giorgia Simonetti, Arianna Berlendis, Mariangela Farinotti, Bianca Pollo, Manila
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  • Neuroradiological, genetic and clinical characteristics of histone H3 K27-mutant diffuse midline gliomas in the Kansai Molecular Diagnosis Network for CNS Tumors (Kansai Network): multicenter retrospective cohort
    Nobuhide Hayashi, Junya Fukai, Hirokazu Nakatogawa, Hiroshi Kawaji, Ema Yoshioka, Yoshinori Kodama, Kosuke Nakajo, Takehiro Uda, Kentaro Naito, Noriyuki Kijima, Yoshiko Okita, Naoki Kagawa, Yoshinobu Takahashi, Naoya Hashimoto, Hideyuki Arita, Koji Takano
    Acta Neuropathologica Communications.2024;[Epub]     CrossRef
  • Genetic alteration analysis of non-pediatric diffuse midline glioma, H3 K27-altered
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    Vincenzo Di Nunno, Enrico Franceschi, Lidia Gatto, Alicia Tosoni, Stefania Bartolini, Alba Ariela Brandes
    Expert Review of Clinical Pharmacology.2023; 16(1): 17.     CrossRef
  • Genomic profiles of IDH-mutant gliomas: MYCN-amplified IDH-mutant astrocytoma had the worst prognosis
    Kwanghoon Lee, Seong-Ik Kim, Eric Eunshik Kim, Yu-Mi Shim, Jae-Kyung Won, Chul-Kee Park, Seung Hong Choi, Hongseok Yun, Hyunju Lee, Sung-Hye Park
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  • Radiotherapy and radio‐sensitization in H3K27M‐mutated diffuse midline gliomas
    Chao Liu, Shuwen Kuang, Lei Wu, Quan Cheng, Xuan Gong, Jun Wu, Longbo Zhang
    CNS Neuroscience & Therapeutics.2023; 29(7): 1721.     CrossRef
  • Volumetric response and pattern of failure of histone altered high grade glioma in adults following management with radiation therapy
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    Xuan Gong, Shuwen Kuang, Dongfeng Deng, Jun Wu, Longbo Zhang, Chao Liu
    CNS Neuroscience & Therapeutics.2023; 29(12): 3863.     CrossRef
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  • H3 K27M-Altered Diffuse Midline Gliomas: A Review
    Karol Wiśniewski, Andrew Ghaly, Kate Drummond, Andreas Fahlstrӧm
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  • Adult diffuse midline gliomas H3 K27-altered: review of a redefined entity
    Carlos Axel López-Pérez, Xochitl Franco-Mojica, Ricardo Villanueva-Gaona, Alexandra Díaz-Alba, Marco Antonio Rodríguez-Florido, Victor Garcia Navarro
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  • H3K27M-Altered Diffuse Midline Gliomas Among Adult Patients: A Systematic Review of Clinical Features and Survival Analysis
    Othman Bin-Alamer, Adrian E. Jimenez, Tej D. Azad, Chetan Bettegowda, Debraj Mukherjee
    World Neurosurgery.2022; 165: e251.     CrossRef
  • Molecular profile and clinical features of patients with gliomas using a broad targeted next generation‑sequencing panel
    Ourania Romanidou, Paraskevi Apostolou, Kyriakos Kouvelakis, Kyriakos Tsangaras, Alexia Eliades, Achilleas Achilleos, Charalambos Loizides, Christos Lemesios, Marios Ioannides, Elena Kypri, George Koumbaris, Kyriaki Papadopoulou, Athanasios Papathanasiou,
    Oncology Letters.2022;[Epub]     CrossRef
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  • 257 Download
  • 21 Web of Science
  • 20 Crossref
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