Les trajectoires de l'activité cardiaque de la petite enfance au début de l'enfance prédisent différemment les symptômes d'autisme et d'anxiété dans le syndrome de l'X fragile

Titre original en anglais : Trajectories of Heart Activity Across Infancy to Early Childhood Differentially Predict Autism and Anxiety Symptoms in Fragile X Syndrome

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Hogan, A., Hunt, E., Smith, K., Black, C., Bangert, K., Klusek, J., & Roberts, J. (2021). Trajectories of Heart Activity Across Infancy to Early Childhood Differentially Predict Autism and Anxiety Symptoms in Fragile X Syndrome. Frontiers in Psychiatry, 12, 727559-727559. https://doi.org/10.3389/fpsyt.2021.727559

Date de publication: 06/10/2021 Ajout dans AutiHub: 26/09/2026 Type: Article Langue de l’article: Anglais
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Auteurs

Auteur·ices des publications
7
Auteur·ices de la publication identifié·es comme autistes
1 / 7 (14,3 %)
  1. Abigail Hogan
  2. Erin Hunt
  3. Kayla Smith *
  4. Conner Black
  1. Katherine Bangert
  2. Jessica Klusek
  3. Jane Roberts

Résumé

Contexte : Le syndrome de l'X fragile (FXS) est un trouble monogénique caractérisé par des taux élevés de trouble du spectre de l'autisme (TSA) et d'anxiété. Une « hypothèse d'hyperéveil » de longue date dans le FXS soutient qu'un dysfonctionnement du système nerveux autonome (SNA) sous-tend de nombreux symptômes du FXS. Toutefois, le début développemental et la trajectoire du dysfonctionnement du SNA, ainsi que les conséquences du dysfonctionnement du SNA sur les symptômes psychiatriques ultérieurs, restent mal compris dans le FXS. Une meilleure compréhension de l'émergence, de la trajectoire et des conséquences du dysfonctionnement du SNA au cours du développement précoce dans le FXS a des implications cruciales pour la prévention, l'intervention et des résultats optimaux tant dans le développement typique qu'atypique. Cette étude longitudinale a examiné si et quand les garçons atteints de FXS présentent un fonctionnement atypique du SNA, de la petite enfance au début de l'enfance, et comment les trajectoires du fonctionnement du SNA au cours de la petite enfance et du début de l'enfance prédisent ultérieurement la sévérité des symptômes de TSA et d'anxiété au cours du développement. Méthodes : Les participants comprenaient 73 garçons atteints de FXS et 79 garçons au développement typique (TD) appariés selon l'âge. L'activité cardiaque de base a été enregistrée lors de plusieurs évaluations entre 3 et 83 mois d'âge, aboutissant à 372 observations. L'éveil général et l'activité parasympathique ont été indexés respectivement par l'intervalle interbattement (IBI) et l'arythmie sinusale respiratoire (RSA). Les symptômes de TSA et d'anxiété ont été évalués à l'âge de 36 mois ou plus tard chez un sous-groupe de participants (FXS n = 28 ; TD n = 25). Résultats : Les garçons atteints de FXS ont présenté des profils atypiques de changement développemental du fonctionnement du SNA au cours de la petite enfance et du début de l'enfance. Par conséquent, le dysfonctionnement du SNA est devenu progressivement plus divergent au fil du temps, le groupe FXS présentant un IBI significativement plus court et une RSA plus faible à 29 et 24 mois d'âge, respectivement. Un IBI plus court à 24 mois et une pente d'IBI plus plate au cours du développement prédisaient des symptômes d'anxiété élevés, mais non des symptômes de TSA, plus tard durant l'enfance chez les garçons FXS et TD. Une RSA réduite à 24 mois prédisait des symptômes de TSA élevés, mais non des symptômes d'anxiété, dans les deux groupes. Le changement développemental de la RSA au cours du développement précoce ne prédisait pas les symptômes ultérieurs d'anxiété ou de TSA. Conclusion : Il s'agit de la première étude longitudinale à examiner l'« hypothèse d'hyperéveil » chez les nourrissons et les jeunes enfants atteints de FXS. Les résultats suggèrent que l'hyperéveil, c'est-à-dire un IBI plus court et une RSA plus faible, est manifeste chez les garçons atteints de FXS dès l'âge de 24 à 29 mois. Il est intéressant de noter que des aspects distincts du fonctionnement précoce du SNA sont différemment liés aux symptômes ultérieurs de TSA et d'anxiété. L'éveil général, indexé par un IBI plus court qui devient progressivement plus divergent de celui des témoins TD, prédit les symptômes ultérieurs d'anxiété. En revanche, les facteurs liés au parasympathique, indexés par des niveaux plus faibles de RSA, prédisent les symptômes de TSA. Ces résultats soutiennent l'« hypothèse d'hyperéveil » dans le FXS, en ce sens qu'un dysfonctionnement du SNA manifeste précocement au cours du développement prédit des symptômes de TSA et d'anxiété apparaissant ultérieurement. Cette étude a également des implications importantes pour le développement de traitements et d'interventions ciblés qui pourraient potentiellement atténuer les effets à long terme de l'hyperéveil dans le FXS.

Background: Fragile X syndrome (FXS) is a monogenic disorder characterized by high rates of autism spectrum disorder (ASD) and anxiety. A longstanding “hyperarousal hypothesis” in FXS has argued that ANS dysfunction underpins many symptoms of FXS. However, the developmental onset and trajectory of ANS dysfunction, as well as the consequences of ANS dysfunction on later psychiatric symptoms, remain poorly understood in FXS. Insight into the emergence, trajectory, and consequences of ANS dysfunction across early development in FXS has critical implications for prevention, intervention, and optimal outcomes in both typical and atypical development. This longitudinal study investigated whether and when males with FXS evidence atypical ANS function from infancy through early childhood, and how trajectories of ANS function across infancy and early childhood predict ASD and anxiety symptom severity later in development. Methods: Participants included 73 males with FXS and 79 age-matched typically developing (TD) males. Baseline heart activity was recorded at multiple assessments between 3 and 83 months of age, resulting in 372 observations. General arousal and parasympathetic activity were indexed via interbeat interval (IBI) and respiratory sinus arrhythmia (RSA), respectively. ASD and anxiety symptoms were assessed at 36 months of age or later in a subgroup of participants (FXS n = 28; TD n = 25). Results: Males with FXS exhibited atypical patterns of developmental change in ANS function across infancy and early childhood. As a result, ANS dysfunction became progressively more discrepant across time, with the FXS group exhibiting significantly shorter IBI and lower RSA by 29 and 24 months of age, respectively. Shorter IBI at 24 months and a flatter IBI slope across development predicted elevated anxiety symptoms, but not ASD symptoms, later in childhood in both FXS and TD males. Reduced RSA at 24 months predicted elevated ASD symptoms, but not anxiety symptoms, in both groups. Developmental change in RSA across early development did not predict later anxiety or ASD symptoms. Conclusion: This is the first longitudinal study to examine the “hyperarousal hypothesis” in infants and young children with FXS. Findings suggest that hyperarousal (i.e., shorter IBI, lower RSA) is evident in males with FXS by 24–29 months of age. Interestingly, unique aspects of early ANS function differentially relate to later ASD and anxiety symptoms. General arousal, indexed by shorter IBI that becomes progressively more discrepant from TD controls, predicts later anxiety symptoms. In contrast, parasympathetic-related factors, indexed by lower levels of RSA, predict ASD symptoms. These findings support the “hyperarousal hypothesis” in FXS, in that ANS dysfunction evident early in development predicts later-emerging symptoms of ASD and anxiety. This study also have important implications for the development of targeted treatments and interventions that could potentially mitigate the long-term effects of hyperarousal in FXS.

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7
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337
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0
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57 / 337 (16,9 %) noms sont associés à un·e auteurice déjà présent·e dans AutiHub. 280 / 337 (83,1 %) noms ne sont pas encore associés.
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Calculé sur l’ensemble des noms d’auteurices détectés dans la bibliographie citée. Parmi les noms associés à un·e auteurice déjà présent·e dans AutiHub : 1 / 57 (1,8 %). Personnes distinctes identifiées comme autistes : 1 / 266 (0,4 %).
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