Effects of age and lesion location on motor, behavioral and cognitive outcomes in pediatric posterior fossa tumors

项目总结 60%的儿童脑瘤位于包括小脑在内的后颅窝。 患有发育性小脑损伤的儿童出现一系列不良后果的风险增加， 包括更高的自闭症诊断率以及长期的认知和学习挑战。传统上讲 被认为是一种运动结构的广泛证据现在将小脑与各种认知和行为联系起来 功能，以及不典型的小脑结构和功能已被报道在多发性神经发育和 精神障碍。小脑神经解剖学的一个关键特征是存在功能亚区 支持感应电机和多种非电机功能。这导致了一种假设，即运动、认知和 行为结果应该是基于病变位置的可预测的。而我们之前的损伤-症状 MAP研究表明，成年小脑中风患者就是这种情况，但研究有限。 在儿科人群中将病变的位置与结果联系起来。此外，我们和其他人已经提出， 小脑辅助的学习机制在发育过程中是至关重要的，这导致了这一假说 年龄较小的小脑损伤与更糟糕的长期结果有关，而不是 成人期。根据围产期小脑损伤的早产儿的研究结果，早期小脑损伤 可能导致特定皮质区域的非典型发育，小脑与这些区域相互连接 病变部位依赖方式。为了检验这些假设，我们建议分析先前存在的结构 70例有小脑肿瘤病史儿童的影像和神经心理学资料。我们会 进行基于多变量体素的病变-症状映射以确定病变之间的关系 位置和各种运动、认知和行为结果变量；我们将专门研究 肿瘤诊断和治疗方案的年龄对这些结果的影响；以及，在纵向 数据，我们将检查肿瘤诊断年龄和病变位置对长期预后和 全脑灰质和白质发育。这项研究的目标是改进结果预测。 和儿童小脑患者的教育规划。更好地理解(S) 早期小脑损伤影响终身运动、认知和行为发育可能导致新的 旨在改善这些儿童的行为和神经结果的干预措施。

PROJECT SUMMARY Sixty percent of childhood brain tumors are located in the posterior fossa, which includes the cerebellum. Children with developmental cerebellar damage are at an increased risk for a range of adverse outcomes, including higher rates of autism diagnoses and long term cognitive and academic challenges. Traditionally considered a motor structure, extensive evidence now links the cerebellum to various cognitive and behavioral functions, and atypical cerebellar structure and function has been reported in multiple neurodevelopmental and psychiatric disorders. A key feature of cerebellar neuroanatomy is the existence of functional subregions that support sensorimotor and multiple non-motor functions. This leads to the hypothesis that motor, cognitive, and behavioral outcomes should be predictable based on lesion location. While our previous lesion-symptom mapping studies have shown this to be the case in adult cerebellar stroke patients, there are limited studies linking location of lesion to outcomes in pediatric populations. Further, we and others have proposed that the learning mechanisms subserved by the cerebellum are critical during development, leading to the hypothesis that cerebellar damage at a younger age would be associated with worse long-term outcomes than damage in adulthood. Based on findings from preterm infants with perinatal cerebellar damage, early cerebellar damage could lead to the atypical development of specific cortical regions to which the cerebellum interconnects in a lesion location-dependent manner. To test these hypotheses, we propose to analyze pre-existing structural imaging and neuropsychological data from ~70 children with a history of pediatric cerebellar tumor. We will conduct multivariate voxel-based lesion-symptom mapping to determine the relationship between lesion location and various motor, cognitive, and behavioral outcome variables; we will specifically examine the impact of age at tumor diagnosis and treatment protocols on these outcomes; and, in patients with longitudinal data, we will examine the impact of age of tumor diagnosis and lesion location on long term outcomes and whole-brain grey and white matter development. The goal of this research is to improve outcome prediction and educational planning in pediatric cerebellar patients. A better understanding of the mechanism(s) by which early cerebellar damage impacts lifelong motor, cognitive, and behavioral development may lead to new interventions to enhance behavioral and neural outcomes for these children.