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小脑放射性海绵状畸形:两例临床特征

Radiation-Induced Cavernous Malformation in the Cerebellum: Clinical Features of Two Cases.

作者信息

Choi Hyoung Soo, Kim Chae-Yong, Choi Byung Se, Jeon Seung Hyuck, Kim In Ah, Kim Joo-Young, Lee Kyu Sang, Choe Gheeyoung

机构信息

Department of Pediatrics, Seoul National University Bundang Hospital, Seoul National University College of Medicine, Seongnam, Korea.

Department of Neurosurgery, Seoul National University Bundang Hospital, Seoul National University College of Medicine, Seongnam, Korea.

出版信息

Brain Tumor Res Treat. 2025 Apr;13(2):58-64. doi: 10.14791/btrt.2025.0003.

DOI:10.14791/btrt.2025.0003
PMID:40347128
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12070075/
Abstract

Radiation-induced cavernous malformations (RICMs) are rare but significant late complications of high-dose radiation therapy, particularly in young survivors of brain tumors. This report presents two cases of RICMs following aggressive multimodal treatment, including surgery, chemotherapy, and radiation therapy. Case 1 was a 22-year-old male patient with medulloblastoma treated with craniospinal irradiation, tumor bed boost, and tandem autologous peripheral blood stem cell transplantation. Approximately 8 years after treatment completion, routine follow-up imaging revealed a small focal hemorrhage in the right cerebellum, consistent with an RICM. The lesion was asymptomatic and managed conservatively with regular imaging, showing spontaneous resolution over time, with a significant size reduction noted 9 years post-treatment. Case 2 describes a 32-year-old male with an intracranial germinoma treated with whole-ventricular irradiation. Three years after treatment, the patient developed a symptomatic hemorrhagic RICM near a pre-existing developmental venous anomaly. Surgical resection and Gamma Knife Surgery stabilized the lesion; however, residual symptoms, including tremors and gait disturbances, persisted, affecting the patient's daily activities. These cases illustrate the diverse clinical courses of RICMs, ranging from spontaneous resolution to the necessity of surgical intervention, and emphasize the importance of long-term surveillance and tailored management strategies for late-onset complications.

摘要

放射性海绵状血管畸形(RICM)是高剂量放射治疗罕见但严重的晚期并发症,尤其是在脑肿瘤年轻幸存者中。本报告介绍了两例在积极的多模式治疗(包括手术、化疗和放射治疗)后发生的RICM病例。病例1是一名22岁的男性髓母细胞瘤患者,接受了全脑全脊髓照射、瘤床加量照射和串联自体外周血干细胞移植。治疗结束约8年后,常规随访影像学检查发现右小脑有一个小的局灶性出血,符合RICM表现。该病变无症状,通过定期影像学检查进行保守治疗,随着时间推移显示自发消退,治疗后9年时病变大小显著缩小。病例2描述了一名32岁男性颅内生殖细胞瘤患者,接受了全脑室照射。治疗三年后,患者在既往存在的发育性静脉异常附近出现了有症状的出血性RICM。手术切除和伽玛刀手术使病变稳定;然而,包括震颤和步态障碍在内的残留症状仍然存在,影响了患者的日常活动。这些病例说明了RICM的不同临床病程,从自发消退到需要手术干预,并强调了对迟发性并发症进行长期监测和制定个性化管理策略的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/cfde6977a278/btrt-13-58-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/a358f21208cd/btrt-13-58-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/94f4d7ea9571/btrt-13-58-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/b1976a5c2093/btrt-13-58-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/cfde6977a278/btrt-13-58-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/a358f21208cd/btrt-13-58-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/94f4d7ea9571/btrt-13-58-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/b1976a5c2093/btrt-13-58-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3565/12070075/cfde6977a278/btrt-13-58-g004.jpg

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本文引用的文献

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Case report: A second case of cerebral cavernous malformation after high-dose chemotherapy for medulloblastoma.病例报告:髓母细胞瘤大剂量化疗后发生脑海绵状畸形的第二例病例。
Front Oncol. 2024 Oct 30;14:1386468. doi: 10.3389/fonc.2024.1386468. eCollection 2024.
2
Radiation-induced cavernoma in pediatric CNS tumors: a systematic review and treatment paradigm.儿童中枢神经系统肿瘤放疗后引起的海绵状血管畸形:系统评价和治疗模式。
Childs Nerv Syst. 2024 Nov;40(11):3527-3536. doi: 10.1007/s00381-024-06543-0. Epub 2024 Jul 19.
3
Stereotactic radiosurgery for intracranial cavernous malformations of the deep-seated locations: systematic review and meta-analysis.
立体定向放射外科治疗深部颅内海绵状血管畸形:系统评价与Meta分析
Neurosurg Rev. 2024 Apr 24;47(1):186. doi: 10.1007/s10143-024-02434-9.
4
Radiation-Induced Cerebral Cavernous Malformations: A Single-Center Experience and Systematic Literature Review.辐射诱发的脑海绵状畸形:单中心经验及系统文献综述
World Neurosurg. 2023 Nov;179:222-232.e2. doi: 10.1016/j.wneu.2023.08.036. Epub 2023 Aug 17.
5
A Huge Radiation-Induced Cavernous Hemangioma Following Stereotactic Radiosurgery for Meningioma: A Case Report.立体定向放射外科治疗脑膜瘤后出现巨大放射性海绵状血管瘤:一例报告
Brain Tumor Res Treat. 2022 Jul;10(3):190-194. doi: 10.14791/btrt.2022.0020.
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Developmental venous anomaly with radiation-induced arteriovenous malformation.伴放射性动静脉畸形的发育性静脉畸形。
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