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Long-term follow-up of surgical intervention pattern in pediatric low-grade gliomas: report from the German SIOP-LGG 2004 cohort
Journal of Neurosurgery: Pediatrics ( IF 2.1 ) Pub Date : 2022-07-22 , DOI: 10.3171/2022.6.peds22108
Ulrich-Wilhelm Thomale 1 , Astrid K Gnekow 2 , Daniela Kandels 2 , Brigitte Bison 3, 4 , Pablo Hernáiz Driever 5 , Olaf Witt 6 , Torsten Pietsch 7 , Arend Koch 8 , David Capper 8 , Rolf-Dieter Kortmann 9 , Beate Timmermann 10 , Semi Harrabi 11 , Michèle Simon 5 , Ahmed El Damaty 12 , Juergen Krauss 13 , Martin U Schuhmann 14 , Annette Aigner 15
Affiliation  

OBJECTIVE

Neurosurgical treatment is an integral part of the treatment algorithms for pediatric low-grade glioma (LGG), yet patterns of surgical procedures are rarely challenged. The objective of this study was to evaluate surgical treatment patterns in pediatric LGG.

METHODS

The German Societé Internationale d’Oncologie Pédiatrique (SIOP)–LGG 2004 cohort was analyzed to identify relevant patient and tumor characteristics associated with time to death, next surgery, number of resections, and radiological outcome.

RESULTS

A total of 1271 patients underwent 1713 neurosurgical interventions (1 intervention in 947, 2 in 230, 3 in 70, and 4–6 in 24). The median age of the study population was 8.57 years at first surgery, and 46.1% were female. Neurofibromatosis type 1 (NF1) was found in 4.4%, and 5.4% had tumor dissemination. Three hundred fifty-four patients (27.9%) had chemotherapy and/or radiotherapy. The cumulative incidence of second surgery at 10 years was 26%, and was higher for infants, those with spinal and supratentorial midline (SML) tumors, and those with pilomyxoid astrocytomas. The hazard ratio for subsequent surgery was higher given dissemination and noncomplete initial resection, and lower for caudal brainstem and SML tumors. Among 1225 patients with fully documented surgical records and radiological outcome, 613 reached complete remission during the observation period, and 50 patients died. Patients with pilocytic astrocytoma had higher chances for a final complete remission, whereas patients with initial partial or subtotal tumor resection, dissemination, NF1, or primary tumor sites in the spinal cord and SML had lower chances.

CONCLUSIONS

Neurosurgery is a key element of pediatric LGG treatment. In almost 50% of the patients, however, at least some tumor burden will remain during long-term follow-up. This study found that most of these patients reached a stable disease status without further surgeries. Multidisciplinary team decisions must balance the goal of complete resection, risk factors, repeated surgeries, and possible treatment alternatives in a wide range of heterogeneous entities. Procedural details and neurological outcome should be recorded to better assess their impact on long-term outcome.



中文翻译:


儿童低级别胶质瘤手术干预模式的长期随访:来自德国 SIOP-LGG 2004 年队列的报告


 客观的


神经外科治疗是儿科低级别胶质瘤 (LGG) 治疗方案中不可或缺的一部分,但外科手术的模式很少受到挑战。本研究的目的是评估儿科 LGG 的手术治疗模式。

 方法


对德国国际儿科肿瘤学会 (SIOP)-LGG 2004 年队列进行了分析,以确定与死亡时间、下次手术、切除次数和放射学结果相关的患者和肿瘤特征。

 结果


共有 1,271 名患者接受了 1,713 次神经外科干预(其中 947 例接受 1 次干预,230 例接受 2 次干预,70 例接受 3 次干预,24 例接受 4-6 次干预)。研究人群首次手术时的中位年龄为 8.57 岁,其中 46.1% 为女性。 4.4% 的患者患有 1 型神经纤维瘤病 (NF1),5.4% 的患者出现肿瘤播散。三百五十四名患者(27.9%)接受了化疗和/或放疗。 10 岁时第二次手术的累积发生率为 26%,婴儿、脊柱和幕上中线 (SML) 肿瘤以及毛粘液样星形细胞瘤患者的第二次手术累积发生率较高。由于扩散和初次切除不完整,后续手术的风险比较高,而尾部脑干和 SML 肿瘤的风险比较低。在 1225 名有完整手术记录和放射学结果记录的患者中,613 名患者在观察期间达到完全缓解,50 名患者死亡。毛细胞型星形细胞瘤患者最终完全缓解的机会较高,而最初接受部分或次全肿瘤切除、扩散、NF1或脊髓原发肿瘤部位和 SML 的患者获得最终完全缓解的机会较低。

 结论


神经外科是儿科 LGG 治疗的关键要素。然而,在近 50% 的患者中,在长期随访期间至少会保留一些肿瘤负荷。这项研究发现,大多数患者的病情已达到稳定状态,无需进一步手术。多学科团队的决策必须平衡完全切除的目标、风险因素、重复手术以及各种异质实体中可能的治疗方案。应记录程序细节和神经学结果,以更好地评估其对长期结果的影响。

更新日期:2022-07-22
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