Sensory Symptoms and Signs of Hyperarousal in Individuals with Fragile X Syndrome: Findings from the FORWARD Registry and Database Multisite Study.

Autor: Lachiewicz AM; Department of Pediatrics, Duke University Health System, Durham, NC, USA. ave.lachiewicz@duke.edu., Stackhouse TM; Developmental FX, Denver, CO, USA., Burgess K; North Carolina Fragile X Foundation, Durham, NC, USA., Burgess D; Department of Pediatrics, Duke University Health System, Durham, NC, USA., Andrews HF; Departments of Psychiatry and Biostatistics, Mailman School of Public Health, Columbia University, Irving Medical Center, New York, NY, USA., Choo TH; Division of Mental Health Data Science, New York State Psychiatric Institute, New York, NY, USA., Kaufmann WE; Department of Human Genetics, Emory University School of Medicine, Atlanta, GA, USA., Kidd SA; Department of Pediatrics, University of California San Francisco, San Francisco, CA, USA.
Jazyk: angličtina
Zdroj: Journal of autism and developmental disorders [J Autism Dev Disord] 2024 Nov; Vol. 54 (11), pp. 4259-4277. Date of Electronic Publication: 2023 Oct 16.
DOI: 10.1007/s10803-023-06135-y
Abstrakt: This study was designed to increase our understanding about characteristics and the impact of sensory symptoms (SS) and signs of hyperarousal (HA) in individuals with fragile X syndrome (FXS) from childhood through early adulthood and by gender. Data derived from the Fragile X Online Registry With Accessible Research Database (FORWARD), a natural history study of FXS, were analyzed using descriptive statistics and multivariate linear and logistic regression models to examine SS and signs of HA, their impact on behavioral regulation and limitations on the subject/family. The sample (N = 933) consisted of 720 males and 213 females. More males were affected with SS (87% vs. 68%) and signs of HA (92% vs. 79%). Subjects who were endorsed as having a strong sensory response had more comorbidities, including behavioral problems. The predominant SS was difficulty with eye gaze that increased with age in both genders. As individuals age, there was less use of non-medication therapies, such as occupational therapy (OT)/physical therapy (PT), but there was more use of psychopharmacological medications and investigational drugs for behaviors. Multiple regression models suggested that endorsing SS and signs of HA was associated with statistically significantly increased ABC-C-I subscale scores and limited participation in everyday activities. This study improves our understanding of SS and signs of HA as well as their impact in FXS. It supports the need for more research regarding these clinical symptoms, especially to understand how they contribute to well-known behavioral concerns.
(© 2023. This is a U.S. Government work and not under copyright protection in the US; foreign copyright protection may apply.)
Databáze: MEDLINE
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