Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome.
Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome.
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潜在类别分析可识别脆性 X 综合征儿童的独特行为亚型。
DOI：
10.1007/s10803-022-05821-7
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发表时间：
2024
影响因子：
3.9
通讯作者：
FORWARDConsortium
中科院分区：
文献类型：
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作者：
Kaufmann,WalterE;Raspa,Melissa;Bann,CarlaM;Gable,JuliaM;Harris,HollyK;Budimirovic,DejanB;Lozano,Reymundo;FORWARDConsortium
脆性X综合征（FXS）的特点是多变的神经行为异常，这导致了开发和评估治疗以及确定准确预后的困难。我们采用了一个儿科横断面样本（1,072名男性，338名女性）从FORWARD，一个基于临床的自然史研究，以确定行为亚型的潜在类别分析。输入包括共同发生的行为条件，睡眠和感觉问题，自闭症行为量表（SCQ，SRS-2），和异常行为检查表修订FXS（ABCFX）。5级解决方案产生了最有临床意义的、药物治疗独立的行为组，具有独特的SCQ、SRS-2和ABCFX特征，并且充分不重叠（≥ 71%）：“轻度”（31%）、“中度无社交障碍”（32%）、“中度伴社交障碍”（7%）、“中度伴破坏性行为”（20%）和“重度”（9%）。我们的研究结果支持FXS亚型，以改善临床管理和治疗开发。
Fragile X syndrome (FXS) is characterized by variable neurobehavioral abnormalities, which leads to difficulties in developing and evaluating treatments and in determining accurate prognosis. We employed a pediatric cross-sectional sample (1,072 males, 338 females) from FORWARD, a clinic-based natural history study, to identify behavioral subtypes by latent class analysis. Input included co-occurring behavioral conditions, sleep and sensory problems, autistic behavior scales (SCQ, SRS-2), and the Aberrant Behavior Checklist revised for FXS (ABCFX). A 5-class solution yielded the most clinically meaningful, pharmacotherapy independent behavioral groups with distinctive SCQ, SRS-2, and ABCFXprofiles, and adequate non-overlap (≥ 71%): “Mild” (31%), “Moderate without Social Impairment” (32%), “Moderate with Social Impairment” (7%), “Moderate with Disruptive Behavior” (20%), and “Severe” (9%). Our findings support FXS subtyping, for improving clinical management and therapeutic development.