Department of Experimental Psychology, University of Oxford, United Kingdom.
Department of Experimental Psychology, University of Oxford, United Kingdom; Turner Institute for Brain and Mental Health, Monash University, Melbourne, Australia.
Res Dev Disabil. 2020 Sep;104:103692. doi: 10.1016/j.ridd.2020.103692. Epub 2020 May 13.
Even genetic disorders associated with monogenic aetiologies are characterized by complex and variable risk for poor outcomes, highlighting the need to follow trajectories longitudinally. Here, we investigated the longitudinal relationships between attention deficit hyperactivity disorder (ADHD) and autism spectrum disorder (ASD) symptoms in a population at high risk for both: boys with fragile X syndrome. 59 boys with fragile X syndrome aged 3-10 years old at entry participated in this study, and were followed up one and two years after their first visit. As expected, we found strong relationships over three timepoints for ADHD symptoms (as measured by the parent-rated Conners scale) and ASD symptoms (as measured by the Social Communication Questionnaire [SCQ]). In addition, using structural equation modeling (SEM) we found that ADHD symptoms at time 2 predicted ASD symptoms at time 3, suggestive of a causal relationship. Importantly, these relationships hold when including chronological age at entry to the study, as well as when including severity of impairment as measured by IQ, and their effects on both ASD and ADHD symptoms do not reach significance. This result highlights the need to study outcomes longitudinally and it informs the comorbidity of the two symptom domains in FXS as well as their potential directionality, both of which have been little researched. In addition, our findings may suggest a future need to study how ADHD symptoms and their treatment impact individuals with ASD.
即使是与单基因病因相关的遗传疾病，其不良预后的风险也具有复杂性和可变性，这突出表明需要进行纵向追踪研究。在这里，我们研究了脆性 X 综合征高风险人群中注意缺陷多动障碍（ADHD）和自闭症谱系障碍（ASD）症状的纵向关系：患有脆性 X 综合征的男孩。本研究纳入了 59 名年龄在 3-10 岁的脆性 X 综合征男孩，在首次就诊后的 1 年和 2 年进行了随访。正如预期的那样，我们在三个时间点上发现了 ADHD 症状（通过父母评定的 Conners 量表测量）和 ASD 症状（通过社会沟通问卷 [SCQ] 测量）之间的强相关性。此外，使用结构方程模型（SEM），我们发现第 2 次就诊时的 ADHD 症状预测了第 3 次就诊时的 ASD 症状，提示存在因果关系。重要的是，当将进入研究时的实际年龄以及智商测量的损伤严重程度纳入考虑时，这些关系仍然成立，并且它们对 ASD 和 ADHD 症状的影响并不显著。这一结果强调了需要进行纵向研究来观察结局，这也为 FXS 中两个症状领域的共病及其潜在的方向性提供了信息，而这两方面的研究都很少。此外，我们的发现可能表明未来需要研究 ADHD 症状及其治疗如何影响 ASD 患者。
