The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): Test development and reliability
The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): Test development and reliability
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DOI：
10.1016/j.nmd.2009.11.014
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发表时间：
2010-03-01
影响因子：
2.8
通讯作者：
Finkel, R. S.
中科院分区：
文献类型：
--
作者：
Glanzman, A. M.;Mazzone, E.;Finkel, R. S.
脊髓性肌萎缩症I型（SMA-I）患者的运动技能非常有限。很难量化这些患者的运动能力，因此，目前没有经过验证的运动功能指标可用作SMA-I临床试验的结局指标。我们开发了费城儿童医院婴儿神经肌肉疾病测试（“CHOP INTEND”），以评估SMA-I患者的运动技能。该测试是在对26名平均年龄为11.5个月（1.4-37.9个月）的SMA-I婴儿进行婴儿运动性能测试和费城儿童医院SMA力量测试（一种新设计的SMA运动评估）评估后开发的。CHOP INTEND的项目由专家小组根据项目平均值和标准差、项目频率分布和Chronbach α选择。通过对9名SMA-I婴儿在2个月内进行重测，建立了所得测试的评定者内可靠性;组内相关系数（ICC）（3，1）= 0.96。评价者间可靠性通过4名评价者对患有神经肌肉疾病的混合婴儿组进行视频分析得出; ICC（3，4）= 0.98，在8名典型发育婴儿组中，ICC（3，5）= 0.93。CHOP INTEND的表面效度得到了项目选择中专家小组的支持;然而，需要进一步验证。CHOP INTEND是SMA-I和婴儿期神经肌肉疾病患者运动技能的可靠指标。(c)2009 Elsevier B. V.保留所有权利。
The motor skills of patients with spinal muscular atrophy, type I (SMA-I) are very limited. It is difficult to quantify the motor abilities of these patients and as a result there is currently no validated measure of motor function that can be utilized as an outcome measure in clinical trials of SMA-I. We have developed the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders ("CHOP INTEND") to evaluate the motor skills of patients with SMA-I. The test was developed following the evaluation of 26 infants with SMA-I mean age 11.5 months (1.4-37.9 months) with the Test of Infant Motor Performance and The Children's Hospital of Philadelphia Test of Strength in SMA, a newly devised motor assessment for SMA. Items for the CHOP INTEND were selected by an expert panel based on item mean and standard deviation, item frequency distribution, and Chronbach's alpha. Intra-rater reliability of the resulting test was established by test-retest of 9 infants with SMA-I over a 2 month period; Intraclass correlation coefficient (ICC) (3,1) = 0.96. Interrater reliability was by video analysis of a mixed group of infants with neuromuscular disease by 4 evaluators; ICC (3,4) = 0.98 and in a group of 8 typically developing infants by 5 evaluators ICC (3,5) = 0.93. The face validity of the CHOP INTEND is supported by the use of an expert panel in item selection; however, further validation is needed. The CHOP INTEND is a reliable measure of motor skills in patients with SMA-I and neuromuscular disorders presenting in infancy. (c) 2009 Elsevier B.V. All rights reserved.