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儿童低级别胶质瘤(LGG)幸存者的长期认知缺陷反映了治疗前的状况——来自德国LGG研究的报告

Long-term cognitive deficits in pediatric low-grade glioma (LGG) survivors reflect pretreatment conditions-report from the German LGG studies.

作者信息

Traunwieser Thomas, Kandels Daniela, Pauls Franz, Pietsch Torsten, Warmuth-Metz Monika, Bison Brigitte, Krauss Juergen, Kortmann Rolf-Dieter, Timmermann Beate, Thomale Ulrich-Wilhelm, Luettich Peggy, Neumann-Holbeck Anne, Tischler Tanja, Hernáiz Driever Pablo, Witt Olaf, Gnekow Astrid K

机构信息

Paediatrics and Adolescent Medicine, Medical Faculty, University of Augsburg, Augsburg, Germany.

Department of Clinical Psychology, Helmut Schmidt University, Hamburg, Germany.

出版信息

Neurooncol Adv. 2020 Aug 8;2(1):vdaa094. doi: 10.1093/noajnl/vdaa094. eCollection 2020 Jan-Dec.

Abstract

BACKGROUND

Disease and treatment contribute to cognitive late effects following pediatric low-grade glioma (LGG). We analyzed prospectively collected neuropsychological data of German pediatric LGG survivors and focused on the impact of hydrocephalus at diagnosis, neurofibromatosis type 1 (NF1) status, and extent of surgery.

METHODS

We used the Neuropsychological Basic Diagnostic screening tool based on the Cattell-Horn-Carroll model for intelligence and the concept of cross-battery assessment at 2 and 5 years from diagnosis for 316 patients from the German pediatric LGG study and LGG registry (7.1 years median age; 45 NF1; cerebral hemispheres 16%, supratentorial midline 39%, infratentorial 45%). Hydrocephalus was classified radiologically in 137 non-NF1 patients with infratentorial tumors (95/137 complete/subtotal resection).

RESULTS

Patients with NF1 versus non-NF1 exhibited inferior verbal short-term memory and visual processing ( < .001-.021). In non-NF1 patients, infratentorial tumor site and complete/subtotal resection were associated with sequelae in visual processing, psychomotor speed, and processing speed ( < .001-.008). Non-NF1 patients without surgical tumor reduction and/or nonsurgical treatment experienced similar deficits. Degree of hydrocephalus at diagnosis had no further impact. Psychomotor and processing speed were impaired comparably following chemo-/radiotherapy ( < .001-.021). Pretreatment factors such as NF1 or tumor site were relevant at multivariate analysis.

CONCLUSIONS

All pediatric LGG survivors are at risk to experience long-term cognitive impairments in various domains. Even surgical only management of cerebellar LGG or no treatment at all, that is, biopsy only/radiological diagnosis did not protect cognitive function. Since pattern and extent of deficits are crucial to tailor rehabilitation, neuropsychological and quality of survival assessments should be mandatory in future LGG trials.

摘要

背景

疾病和治疗会导致小儿低级别胶质瘤(LGG)出现认知后期效应。我们对前瞻性收集的德国小儿LGG幸存者的神经心理学数据进行了分析,并重点关注诊断时脑积水的影响、1型神经纤维瘤病(NF1)状态以及手术范围。

方法

我们使用基于卡特尔-霍恩-卡罗尔智力模型的神经心理学基本诊断筛查工具,并采用跨电池评估概念,对来自德国小儿LGG研究和LGG登记处的316例患者在诊断后2年和5年进行评估(中位年龄7.1岁;45例NF1;大脑半球16%,幕上中线39%,幕下45%)。对137例幕下肿瘤的非NF1患者进行放射学脑积水分类(95/137例完全/次全切除)。

结果

NF1患者与非NF1患者相比,言语短期记忆和视觉处理能力较差(P<0.001 - 0.021)。在非NF1患者中,幕下肿瘤部位以及完全/次全切除与视觉处理、心理运动速度和处理速度方面的后遗症相关(P<0.001 - 0.008)。未进行手术肿瘤切除和/或非手术治疗的非NF1患者有类似的缺陷。诊断时脑积水程度没有进一步影响。化疗/放疗后心理运动和处理速度受到类似损害(P<0.001 - 0.021)。多因素分析显示,NF1或肿瘤部位等治疗前因素具有相关性。

结论

所有小儿LGG幸存者都有在各个领域出现长期认知障碍的风险。即使仅对小脑LGG进行手术治疗或根本不进行治疗,即仅进行活检/放射学诊断,也不能保护认知功能。由于缺陷的模式和程度对于定制康复至关重要,未来LGG试验应强制进行神经心理学和生存质量评估。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aaca/7497816/a751c7b63b7c/vdaa094f0001.jpg

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