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Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal em...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9498703/ https://www.ncbi.nlm.nih.gov/pubmed/36140827 http://dx.doi.org/10.3390/genes13091660 |
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author | Lozano, Reymundo Thompson, Talia Dixon-Weber, Jayne Erickson, Craig A. Berry-Kravis, Elizabeth Williams, Sara Smith, Elizabeth Frazier, Jean A. Rosselot, Hilary Farmer, Cristan Hessl, David |
author_facet | Lozano, Reymundo Thompson, Talia Dixon-Weber, Jayne Erickson, Craig A. Berry-Kravis, Elizabeth Williams, Sara Smith, Elizabeth Frazier, Jean A. Rosselot, Hilary Farmer, Cristan Hessl, David |
author_sort | Lozano, Reymundo |
collection | PubMed |
description | Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal emotional or body states. A comprehensive survey of the presence, frequency, and duration of anxiety-related symptoms and questions to elicit open-ended responses was completed by caregivers of 456 individuals with FXS, ages 2–81 years (87 female, 369 male) and 24 female and 2 male FXS self-advocates ages 15–66 years. Caregivers reported classic behavioral indicators of anxiety, such as avoidance, irritability, motor agitation, and physiological symptoms, as well as behavioral features in FXS such as repetitive behavior, aggression, and self-injury. Self-advocate accounts largely paralleled caregiver data. Factor analyses yielded four factors: (1) increased irritability, aggression, and self-injury; (2) increased physical movement, nervous activity, and restlessness; (3) physical and physiological features of anxiety; and (4) internalizing and gastrointestinal symptoms. Caregivers are capable of observing and reporting behaviors that are valid indicators of anxious states that are usually reported in self-report standardized assessments. These results support the development of an anxiety measure for FXS that minimizes problems with rater inference. |
format | Online Article Text |
id | pubmed-9498703 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-94987032022-09-23 Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives Lozano, Reymundo Thompson, Talia Dixon-Weber, Jayne Erickson, Craig A. Berry-Kravis, Elizabeth Williams, Sara Smith, Elizabeth Frazier, Jean A. Rosselot, Hilary Farmer, Cristan Hessl, David Genes (Basel) Article Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal emotional or body states. A comprehensive survey of the presence, frequency, and duration of anxiety-related symptoms and questions to elicit open-ended responses was completed by caregivers of 456 individuals with FXS, ages 2–81 years (87 female, 369 male) and 24 female and 2 male FXS self-advocates ages 15–66 years. Caregivers reported classic behavioral indicators of anxiety, such as avoidance, irritability, motor agitation, and physiological symptoms, as well as behavioral features in FXS such as repetitive behavior, aggression, and self-injury. Self-advocate accounts largely paralleled caregiver data. Factor analyses yielded four factors: (1) increased irritability, aggression, and self-injury; (2) increased physical movement, nervous activity, and restlessness; (3) physical and physiological features of anxiety; and (4) internalizing and gastrointestinal symptoms. Caregivers are capable of observing and reporting behaviors that are valid indicators of anxious states that are usually reported in self-report standardized assessments. These results support the development of an anxiety measure for FXS that minimizes problems with rater inference. MDPI 2022-09-16 /pmc/articles/PMC9498703/ /pubmed/36140827 http://dx.doi.org/10.3390/genes13091660 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Article Lozano, Reymundo Thompson, Talia Dixon-Weber, Jayne Erickson, Craig A. Berry-Kravis, Elizabeth Williams, Sara Smith, Elizabeth Frazier, Jean A. Rosselot, Hilary Farmer, Cristan Hessl, David Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title | Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title_full | Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title_fullStr | Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title_full_unstemmed | Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title_short | Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives |
title_sort | observable symptoms of anxiety in individuals with fragile x syndrome: parent and caregiver perspectives |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9498703/ https://www.ncbi.nlm.nih.gov/pubmed/36140827 http://dx.doi.org/10.3390/genes13091660 |
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