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1型神经纤维瘤病合并丛状神经纤维瘤青年患者的言语学习与记忆:与疾病严重程度的关系

Verbal learning and memory in youth with neurofibromatosis type 1 and plexiform neurofibromas: Relationships with disease severity.

作者信息

Loucas Caitlyn, Wolters Pamela, Toledo-Tamula Mary Anne, Rhodes Amanda, Baldwin Andrea, Goodwin Anne, Widemann Brigitte, Martin Staci

机构信息

Pediatric Oncology Branch, National Cancer Institute, Bethesda, MD, United States.

Clinical Research Directorate (CRD), Frederick National Laboratory for Cancer Research, United States.

出版信息

Eur J Paediatr Neurol. 2022 May;38:7-12. doi: 10.1016/j.ejpn.2022.03.004. Epub 2022 Mar 13.

Abstract

AIM

To provide a comprehensive characterization of verbal learning and memory (VLM) abilities in youth with neurofibromatosis type 1 (NF1) and plexiform neurofibromas (PNs) and to evaluate disease severity as a predictor of VLM functioning over time.

METHOD

As part of a longitudinal natural history study, youth with NF1 and PNs were administered repeat neuropsychological assessments, including measures of VLM and ratings of NF1 disease severity completed by a medical professional. This sub-study analyzed data from 89 patients (M age  = 13.1, SD = 4.3 years, range 6-24 years) who had completed tests of VLM abilities and verbal attention at either baseline and/or 36 months.

RESULTS

VLM scores across the sample fell predominantly within the average range of functioning at both time points. However, relative to peers with mild NF1 disease severity, youth with moderate/severe NF1 disease showed lower functioning across multiple VLM domains at 36 months, even after controlling for the effects of verbal attention.

INTERPRETATION

Exclusive use of overall domain scores does not fully characterize VLM functioning in youth with NF1 and PNs. Additionally, children and adolescents with more severe NF1 disease should be monitored more closely for verbal memory challenges and targeted for interventions.

摘要

目的

全面描述1型神经纤维瘤病(NF1)和丛状神经纤维瘤(PNs)青少年的言语学习和记忆(VLM)能力,并评估疾病严重程度作为VLM功能随时间变化的预测指标。

方法

作为一项纵向自然史研究的一部分,对患有NF1和PNs的青少年进行了重复的神经心理学评估,包括VLM测量以及由医学专业人员完成的NF1疾病严重程度评级。本亚研究分析了89例患者(年龄中位数=13.1岁,标准差=4.3岁,范围6 - 24岁)的数据,这些患者在基线和/或36个月时完成了VLM能力测试和言语注意力测试。

结果

在两个时间点,整个样本的VLM分数主要落在功能的平均范围内。然而,与NF1疾病严重程度较轻的同龄人相比,中度/重度NF1疾病的青少年在36个月时,即使在控制了言语注意力的影响后,在多个VLM领域的功能仍较低。

解读

仅使用总体领域分数并不能完全描述患有NF1和PNs的青少年的VLM功能。此外,对于言语记忆挑战,应更密切地监测患有更严重NF1疾病的儿童和青少年,并针对他们进行干预。

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