Skye Jax, Bruss Joel, Toescu Sebastian, Aquilina Kristian, Lola Gino Bardi, Boes Aaron D
Department of Neurology, University of Iowa Carver College of Medicine, University of Iowa, Iowa City, Iowa, USA.
Department of Pediatrics, University of Iowa Carver College of Medicine, University of Iowa, Iowa City, Iowa, USA.
medRxiv. 2023 Jan 14:2023.01.12.23284375. doi: 10.1101/2023.01.12.23284375.
Approximately 25% of pediatric patients who undergo cerebellar tumor resection develop cerebellar mutism syndrome (CMS). Our group recently showed that damage to the cerebellar outflow pathway is associated with increased risk of CMS. Here, we tested whether these findings replicate in an independent cohort.
We evaluated the relationship between lesion location and the development of CMS in an observational study of 56 pediatric patients who underwent cerebellar tumor resection. We hypothesized that individuals that developed CMS after surgery (CMS+), relative to those that did not (CMS-) would have lesions that preferentially intersected with: 1) the cerebellar outflow pathway, and 2) a previously generated 'lesion-symptom map' of CMS. Analyses were conducted in accordance with pre-registered hypotheses and analytic methods (https://osf.io/r8yjv/).
We found supporting evidence for both hypotheses. Compared with CMS- patients, CMS+ patients (n=10) had lesions with greater overlap with the cerebellar outflow pathway (Cohen's d=.73, p=.05), and the CMS lesion-symptom map (Cohen's d=1.1, p=.004).
These results strengthen the association of lesion location with risk of developing CMS and demonstrate generalizability across cohorts. These findings may help to inform the optimal surgical approach to pediatric cerebellar tumors.
接受小脑肿瘤切除术的小儿患者中，约25%会发生小脑缄默综合征（CMS）。我们团队最近发现，小脑传出通路受损与CMS风险增加有关。在此，我们测试了这些发现能否在一个独立队列中得到重复验证。
在一项对56例接受小脑肿瘤切除术的小儿患者的观察性研究中，我们评估了病变位置与CMS发生之间的关系。我们假设，术后发生CMS的个体（CMS+）相对于未发生的个体（CMS-），其病变会优先与以下两者相交：1）小脑传出通路，以及2）先前生成的CMS“病变-症状图谱”。分析是根据预先注册的假设和分析方法（https://osf.io/r8yjv/）进行的。
我们为这两个假设都找到了支持证据。与CMS-患者相比，CMS+患者（n = 10）的病变与小脑传出通路的重叠程度更高（Cohen's d = 0.73，p = 0.05），与CMS病变-症状图谱的重叠程度也更高（Cohen's d = 1.1，p = 0.004）。
这些结果强化了病变位置与发生CMS风险之间的关联，并证明了在不同队列中的普遍性。这些发现可能有助于为小儿小脑肿瘤的最佳手术方法提供参考。
