Neurocognitive monitoring in congenital central hypoventilation syndrome with the NIH Toolbox®.
Neurocognitive monitoring in congenital central hypoventilation syndrome with the NIH Toolbox®.
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DOI：
10.1002/ppul.25973
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发表时间：
2022-09
影响因子：
3.1
通讯作者：
Zelko, Frank A.
中科院分区：
文献类型：
--
作者：
Welbel, Remi Z.;Rand, Casey M.;Zhou, Amy;Fadl-Alla, Allaa;Chen, Maida Lynn;Weese-Mayer, Debra E.;Zelko, Frank A.
关键词：
先天性中枢低通气综合征(CCHS)是一种罕见的神经营养不良，由类同源盒基因PHOX2B突变引起，该基因改变了对呼吸和自主神经系统的调节，需要人工呼吸机作为生命维持。据报道，CCHS有广泛的神经认知表现，包括一系列认知缺陷。我们将NIH工具箱®认知电池(NTCB)这项新技术应用到CCHS队列中，研究了它相对于传统临床神经认知测量的收敛和发散有效性。NTCB由七个任务组成，通过交互式计算机平板应用程序呈现。对51名CCHS参与者进行了NTCB测试，其中24人也接受了传统的临床神经认知测试(韦氏智力量表)。将来自总样本和次队列的年龄校正的NTCB得分与人群正常值进行比较。结果表明，与总体常模相比，NTCB测试结果表明，测量新学习和信息处理速度的流体认知减少(p <； 0.001)，但测量过去学习的晶体认知没有变化。经年龄校正的NTCB液体和结晶指数与可比韦克斯勒指数之间存在中到强的相关性(r >； 0.60)，支持NTCB的收敛和判别效度。结果揭示了CCHS个体的流体认知缺陷，并表明NTCB是一种有效和敏感的认知结果测量工具。我们的发现表明，NTCB可能在追踪CCHS的神经认知方面起到有用的作用。
Congenital central hypoventilation syndrome (CCHS) is a rare neurocristopathy, caused by mutations in the paired‐like homeobox gene PHOX2B, which alters control of breathing and autonomic nervous system regulation, necessitating artificial ventilation as life‐support. A broad range of neurocognitive performance has been reported in CCHS, including an array of cognitive deficits. We administered the NIH Toolbox® Cognition Battery (NTCB), a novel technology comprised of seven tasks presented via an interactive computer tablet application, to a CCHS cohort and studied its convergent and divergent validity relative to traditional clinical neurocognitive measures. The NTCB was administered to 51 CCHS participants, including a subcohort of 24 who also received traditional clinical neurocognitive testing (Wechsler Intelligence Scales). Age‐corrected NTCB scores from the overall sample and subcohort were compared to population norms. Associations between NTCB indices and Wechsler Intelligence scores were studied to determine the convergent and divergent validity of the NTCB. NTCB test results indicated reduced Fluid Cognition, which measures new learning and speeded information processing (p < 0.001), but intact Crystallized Cognition, which measures past learning, in CCHS relative to population norms. Moderate to strong associations (r > 0.60) were found between age‐corrected NTCB Fluid and Crystallized indices and comparable Wechsler indices, supporting the convergent and discriminant validity of the NTCB. Results reveal deficits of Fluid Cognition in individuals with CCHS and indicate that the NTCB is a valid and sensitive measure of cognitive outcomes in this population. Our findings suggest that the NTCB may play a useful role in tracking neurocognition in CCHS.
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影响因子：
5.1
作者：
SILVESTRI, JM;WEESEMAYER, DE;NELSON, MN
通讯作者：
NELSON, MN
影响因子：
3.6
作者：
Kumar, Rajesh;Woo, Marlyn S.;Macey, Paul M.;Woo, Mary A.;Harper, Ronald M.
通讯作者：
Harper, Ronald M.
影响因子：
2.7
作者：
Carlozzi NE;Tulsky DS;Wolf TJ;Goodnight S;Heaton RK;Casaletto KB;Wong AWK;Baum CM;Gershon RC;Heinemann AW
通讯作者：
Heinemann AW
DOI：
10.1017/s1355617719000614
发表时间：
2019-09-01
影响因子：
2.6
作者：
Scott, Emmi P.;Sorrell, Anne;Benitez, Andreana
通讯作者：
Benitez, Andreana
影响因子：
3.7
作者：
Macey PM;Richard CA;Kumar R;Woo MA;Ogren JA;Avedissian C;Thompson PM;Harper RM
通讯作者：
Harper RM