Impact of brief telehealth interventions on parental stress and challenging behaviors of children with fragile X syndrome

Orphanet J Rare Dis. 2025 Sep 24;20(1):483. doi: 10.1186/s13023-025-04019-1.

ABSTRACT

BACKGROUND: Fragile X syndrome (FXS) is the most common cause of inherited intellectual disability (ID) with comorbid autism and several support requirements. Challenging behaviors are frequently reported as a main concern for parents and caregivers, who also experience increased stress levels. There is little evidence of telehealth parent-implemented intervention (PII) for this population. Our study focused on describing the impact that brief telehealth parent-implemented interventions had on the parental stress levels and challenging behaviors of children with FXS in a Latin American country.

METHODS: Thirteen caregivers were assessed pre- and postintervention with the Parenting Stress Index short form (PSI-SF), Motivation Assessment Scale (MAS), and Fragile-X-specific adaptation of the Aberrant Behavior Checklist-Community questionnaire (ABC-C_FX). Four telehealth sessions were developed with each participant to guide their intervention with their children with FXS. Statistical analysis was performed using paired t tests or Wilcoxon matched-pairs tests, and Pearson’s and Spearman’s correlations were used for comparisons. All the statistical analyses were performed using GraphPad Prism v8.3.0, and a two-tailed p value < 0.05 was considered to indicate statistical significance.

RESULTS: PSI-SF (TS_initial=85(52.5-97) vs. TS_final=55(27.5-90), p = 0.0117) and two MAS subscale frequencies of occurrence (scape_initial=10(4-12.5) vs. scape_final=3(0.5-8.5), p = 0.0146; tangible_initial =11.69 ± 8.27 vs. tangible_final=7.154 ± 6.56, p = 0.0146) significantly decreased. ABC-C_FX did not significantly differ. The LSI-SF was positively correlated with three ABC-C_FX subindexes (lethargy/withdrawal s = 0.719, p = 0.007; hyperactivity r = 0.682, p = 0.01; and irritability s = 0.69, p = 0.011).

CONCLUSIONS: Telehealth parent-implemented interventions decreased parental stress and challenging behavior perception and increased feelings of parental competence. The PII benefits interventions for children with FXS and is a key aspect to consider in situations where movement, transfer and access to specialized professionals are difficult or interfered with in a particular region or because of a major sanitary alert.

PMID:40993733 | DOI:10.1186/s13023-025-04019-1