探讨儿童胚胎肿瘤多层玫瑰结的维持治疗。

IF 0.8 4区 医学 Q4 HEMATOLOGY Journal of Pediatric Hematology/Oncology Pub Date : 2025-07-01 Epub Date: 2025-04-07 DOI:10.1097/MPH.0000000000003033
Mohamed M Ahmed, Fernando Carceller, Leslie R Bridges, Conor Mallucci, Navneet Singh, Sucheta Vaidya
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引用次数: 0

摘要

具有多层玫瑰花结的胚胎肿瘤(ETMR)代表了一种具有侵袭性行为的独特实体。历史回顾性分析表明,1年的总生存率从0%到14%不等。然而,Khan及其同事的一份当代报告显示,2年和4年的总体生存率分别为29%和27%。我们提出了一个18个月大的女孩诊断为ETMR的病例,通过19号染色体microRNA集群扩增证实了最初出现局灶性癫痫发作。患者接受了手术干预、大剂量化疗联合干细胞抢救和质子治疗,在完成标准治疗后达到无病状态。随后,给予为期12个月的维持方案,包括鞘内拓扑替康、口服丙戊酸钠和口服顺式维甲酸。维持治疗耐受性良好,不良反应可控。患者在维持治疗后32个月无进展(从初次就诊算起50个月)。本研究探讨ETMR维持治疗的可行性和安全性。未来的研究可能会探索这种方法,以确定其对儿童ETMR的疗效。
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Exploring Maintenance Therapy in Pediatric Embryonal Tumor With Multilayered Rosettes.

Embryonal tumors with multilayered rosettes (ETMR) represent a distinct entity characterized by aggressive behavior. Historical retrospective analyses have documented dire overall survival rates ranging from 0% to 14% at 1 year. However, a contemporary report by Khan and colleagues shows overall survival rates reaching 29% at 2 years and 27% at 4 years. We present the case of an 18-month-old girl diagnosed with ETMR, confirmed by chromosome 19 microRNA cluster amplification following initial presentation with focal seizures. The patient underwent a combination of surgical interventions, high-dose chemotherapy with stem cell rescue, and proton therapy, achieving a disease-free status after completing standard treatment. Subsequently, a 12-month maintenance regimen comprising intrathecal topotecan, oral sodium valproate, and oral cis-retinoic acid was administered. The maintenance therapy was well tolerated, with manageable adverse effects. The patient remains progression-free for 32 months postmaintenance therapy (50 months from initial presentation). This study explores the feasibility and safety profile of maintenance therapy in ETMR. Future studies may explore this approach to determine its efficacy in children with ETMR.

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来源期刊
CiteScore
1.90
自引率
8.30%
发文量
415
审稿时长
2.5 months
期刊介绍: ​Journal of Pediatric Hematology/Oncology (JPHO) reports on major advances in the diagnosis and treatment of cancer and blood diseases in children. The journal publishes original research, commentaries, historical insights, and clinical and laboratory observations.
期刊最新文献
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