We are excited to share journal publications like this one resulting from FORWARD data. There are many more papers currently in development, and the future for Fragile X syndrome research is bright as more data is gathered.
Fragile X syndrome (FXS) is characterized by variable physical, neurologic, and behavioral manifestations. This variability represents a challenge when assessing outcomes in clinical practice and interventional research studies, as well as determining an accurate prognosis.
Taking advantage of the extensive FORWARD database, we analyzed a pediatric cross-sectional sample (1,072 males, 338 females) in order to identify neurobehavioral subtypes using latent class analysis. As input, we included co-occurring behavioral conditions, sleep and sensory problems, and three standardized instruments collected in FORWARD (autistic behavior: Social Communication Questionnaire [SCQ], Social Responsiveness Scale — Second Edition [SRS-2]; problem behavior: Aberrant Behavior Checklist revised for FXS [ABCFX]).
Among the different models resulting from the latent class analysis, a 5-class solution yielded the most clinically meaningful pharmacotherapy-independent behavioral subtypes. Each group had distinctive SCQ, SRS-2, and ABCFX profiles, and adequate non-overlap (> 71%). They were termed
- “Mild” (31%),
- “Moderate without Social Impairment” (32%),
- “Moderate with Social Impairment” (7%),
- “Moderate with Disruptive Behavior” (20%), and
- “Severe” (9%).
Our findings support the notion of subtyping individuals with FXS, for improving their clinical management and the development of new treatments.
Kaufmann WE, Raspa M, Bann CM, et al. Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome [published online ahead of print, 2022 Nov 28]. J Autism Dev Disord. 2022;10.1007/s10803-022-05821-7. doi:10.1007/s10803-022-05821-7
About the FORWARD Registry & Database
FORWARD is made possible by all the participating families sharing their life experiences for research. By giving families, doctors, scientific researchers, and policymakers an inside look into how Fragile X syndrome presents itself across the human lifespan, you’re involvement is a huge contribution toward more positive health outcomes and better care and services for future generations.
Below are more journal publications resulting from FORWARD data.
more from forward
Importance of Fragile X Specialty Clinics
Research Summary // Appropriate clinical management of affected individuals and their family members have become critical in addressing medical needs to improve quality of life.