BACKGROUND Fragile X syndrome (FXS) is a genetic disorder that is highly comorbid with anxiety and autism spectrum disorder (ASD). Elevated negative affect in young children has been associated with increased risk for both anxiety and ASD; however, these relations remain poorly understood in FXS. METHODS The present prospective longitudinal study examined the trajectory of negative affect from infancy through preschool in males and females with FXS and typical development and its relation to anxiety and ASD. RESULTS Results indicate a complex association reflecting group, developmental, and sex effects. Specifically, the group with FXS displayed a trajectory of increasing negative affect across age that was distinct from the typical controls. This atypical trajectory of negative affect in FXS was driven by sex effects in that males showed lower negative affect during infancy followed by steep increases across the toddler and preschool years whereas the females displayed a flatter trajectory. Finally, elevated negative affect predicted anxiety symptoms in males, but not females, with no relationship to ASD in males or females with FXS. CONCLUSIONS The current work addresses the importance of studying the development of psychopathology in a specific neurogenetic population. Temperamental negative affect was shown to be an important early marker for anxiety in young children with FXS, with subtle differences observed between males and females.

中文翻译：

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患有脆性X综合征的男性和女性的早期负面影响：对焦虑和自闭症的影响。

背景技术脆性X综合征（FXS）是与焦虑症和自闭症谱系障碍（ASD）高度并存的遗传病。幼儿负面影响加剧与焦虑和自闭症风险增加有关。但是，这些关系在FXS中仍然知之甚少。方法本项前瞻性纵向研究研究了FXS的男性和女性以及典型发展过程中从婴儿期到学龄前的负面影响的轨迹，以及其与焦虑症和ASD的关系。结果结果表明一个复杂的关联反映了群体，发育和性别效应。具体而言，有FXS的人群表现出的跨年龄增长的负面影响的轨迹与典型对照组不同。FXS中这种负面影响的非典型轨迹是由性别影响引起的，因为男性在婴儿期表现出较低的负面影响，随后在学步期和学龄前期急剧上升，而女性表现出较为平坦的轨迹。最后，升高的负面影响预示了男性而不是女性的焦虑症状，与患有FXS的男性或女性的ASD没有关系。结论当前的工作解决了研究特定神经遗传学人群心理病理学发展的重要性。气质负面影响已被证明是FXS幼儿焦虑的重要早期标志，男女之间的差异微乎其微。