Research PaperPreventive care services and health behaviors in children with fragile X syndrome
Section snippets
Methods
Data for this analysis were derived from version two of the Fragile X Online Registry with Accessible Research Database (FORWARD), a multisite observational study which began collecting baseline data in 2012 from 25 fragile X clinics across the United States. FORWARD has two primary components: the registry and the longitudinal database. The registry form collects demographic data, and is open to individuals with FXS and their family members. The longitudinal database includes a parent form, a
Results
Of the 898 individuals in the baseline longitudinal dataset, 592 had a registry form, parent form, and clinician form on file (Fig. 1). Of those, we excluded 69 21 years or older, and an additional 117 because they had missing or invalid data for key variables. Table 1 summarizes the differences between those with complete data (n = 406) and those with missing or invalid data (n = 117). The two groups differed significantly in the distributions of household income (P = 0.04), type of health
Discussion
Our study examined receipt of four preventive care outcomes; dental care, immunization status, influenza vaccination, and PA, all based on parent report. Table 3 compares our results to population-based studies examining these measures among children with special health care needs (CSHCN), children without special health care needs, and the general pediatric population. Approximately three-quarters of our clinic based cohort had seen a dentist within the past year, compared to 52–81% of all
Conclusion
Our aim was to describe the current state of preventive care among children with fragile X syndrome, which has not been previously examined. We found that receipt of key preventive care services and practicing health behaviors among children and young adults with FXS appears to be similar to, or greater than children with special health care needs and the general population of children. However, the proportion meeting a number of these guidelines remains suboptimal, and there is uneven access
Disclaimer
The findings and conclusions in this publication are those of the authors and do not necessarily represent the official position of the Centers for Disease Control and Prevention. Elizabeth Berry Kravis, MD, PhD, Milen Velinov, MD, PhD, Nicole Tartaglia, MD, Craig Erikson, MD, and Howard Andrews, PhD were PIs on the study.
Funding
The FORWARD project was supported by the Cooperative Agreement Numbers DD000753, DD001185, DD001186, DD001190, DD001188, and DD001189 funded by the Centers for Disease Control and Prevention.
Declarations of interest
None.
Previous presentation
Poster at the NFXF International Fragile X Conference.
Acknowledgments
We thank the National Fragile X Foundation, collaborating fragile X clinics (listed below), the national coordinator Amie Milunovich, current and former members of the FORWARD Steering Committee: Elizabeth Berry Kravis (PI), MD, PhD, Milen Velinov (PI), MD, PhD, Nicole Tartaglia (PI), MD, Craig Erikson (PI), MD, Walter Kauffman, MD, Stephanie Sherman PhD, Julie Bolen, PhD, MPH, Ted Brown, MD, PhD, Catharine Riley, PhD, MPH, and Tony Ferlenda, MPA for their ongoing partnership and support. We
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