This month, the OFXW team is busy sharing the results of recent research. We're excited to present posters on five of our latest studies at two conferences. From looking at new ways of measuring behavior and brain function, to seeing how we can help people learn more about taking part in studies, we're covering a lot of ground this month.

A small group of us, Don Bailey, Melissa Raspa, and Anne Wheeler, attended the 50th Annual Gatlinburg Conference on Research and Theory in Intellectual and Developmental Disabilities in San Antonio, TX. Melissa and Anne presented four posters on the results of recent studies:

1. Health Care Decision Making in Context: Decisional Capacity and Informed Consent for Clinical Trials

Melissa presented a poster on the results of a study of health care decision-making for people with fragile X syndrome (FXS). We looked at different factors, such as cognitive and social-emotional skills, that may affect their ability to make a choice about taking part in a clinical trial. We used these results to create an iPad app. The app is a new tool for making informed choices about taking part in research studies.

Next, we tested the iPad app to look at which parts of the informed consent process were the hardest and easiest for people to understand. Abstract concepts about the clinical trial design, such as "placebo" or "randomization," were the hardest. Concepts about clinical trial activities, such as what a participant would do when he visits the doctor's office, were the easiest. Asking multiple-choice questions was the best way to check if participants understood the information presented in the iPad app. Allowing people to go over the concepts multiple times also improved understanding.

Melissa reports back, "We got lots of positive feedback on the decision aid. We're hopeful that other researchers or clinical trial managers will think about using it in future studies."

2. Sensory Difficulties in Individuals with Fragile X Syndrome

Melissa also presented a poster that showed the results of our Sensory Experiences survey. In the study, we tested a new set of survey questions, the Brain-Body Center (BBC) Sensory Scales. The BBC Sensory Scales is a parent-reported measure that assesses visual, auditory, tactile, and eating-related sensitivities. The poster presented data on common sensory challenges in FXS, including:

• Following verbal instructions when lots of noise is present,
• Eating more food than appropriate,
• Resisting certain textures of food,
• Avoiding certain food tastes, and
• Getting fingernails cut.

Now that we have tested the BBC Sensory Scales, we hope to use this new survey in future studies. We plan to look more at differences between people with the full mutation and premutation. We also plan to compare the BBC Sensory Scales to other more commonly used measures of sensory behavior.

3. A Pilot Study of MicroEEG in Fragile X Syndrome

In this poster, Anne shared the results of a study that tested how well a new portable, wireless EEG system to assess brain wave patterns would work for people with fragile X syndrome (FXS). Researchers may be able to use this system in future studies to learn more about brain function in people with FXS.

Assessing brain wave patterns, known as electroencephalography or EEG, can help us understand more about learning and memory in people with FXS. It's a promising method that could even help us in clinical trials to test new drugs for treating FXS. However, the challenge is that standard EEG systems have lots of wires and irritate the skin. This makes them too uncomfortable for many people with FXS. MicroEEG® is a new type of EEG that uses stickers with electrodes, is wireless, and is portable for use anywhere. We wanted to see if this new system would be a better choice for people with FXS than standard systems.

We tested microEEG in a small group of people with FXS. Participants tried microEEG while doing different tasks and gave feedback on how it felt. Most participants were willing to wear it and said it was mostly comfortable.

4. Integrating Technologies for Measuring Change in Behavior in Individuals with Intellectual and Developmental Disabilities

Anne's second poster talked about three new digital tools being created at RTI. These tools help track behavior in people with intellectual and developmental disabilities (IDD) such as fragile X syndrome (FXS). Our vision is to combine these tools to track behaviors and responses to treatment.

1. Wearable, motion-measuring devices: We developed a system to detect repetitive behaviors, such as body rocking or hand flapping. The system uses sensors worn on the wrist and chest to detect movement. The system may help us see how specific treatments affect repetitive behavior. We may also be able to see when repetitive behavior may lead to aggression or self-injury.
2. A smartphone app for parents and teachers to log and measure behaviors in real-time: This app, called MOCHA (Measure Outcomes for CHange), lets parents and teachers log challenging behaviors like hitting or screaming. Users can log these events in real-time via a button worn on their wrist or around their neck. The app then asks them to describe each event. This helps users track information about when and why the behavior occurred.
3. Statistical software to help track and analyze treatment change in individuals: The Patient-Centered Clinical Trials (PACT) software helps researchers and health professionals analyze changes in an individual with IDD as a result of treatment. It can help users see how meaningful the changes are over time.

Later this month, Melissa and another OFXW researcher, Sara Andrews, will present a poster at the Society of Behavioral Medicine 38th Annual Meeting & Scientific Sessions in San Diego, CA.

5. Parents' Opinions about Using their Childs' Electronic Medical Records for Research: A Continuum of Perspectives Across Three Genetic Groups

With the increased use of electronic medical records (EMR) and the desire to use them for research, we wanted to look at preferences for access among three groups of parents: those whose children have a known genetic condition (fragile X syndrome [FXS]), those whose children have a suspected genetic condition (autism spectrum disorder [ASD]), and those with typically developing children (TD).

In this poster, we will share the early results of some recent focus groups. We met with parents of children with FXS and ASD, as well as parents of TD children. All parents had concerns about privacy and security. In general, parents of children with FXS or ASD were more willing to share their child's medical records for research than parents of children who were TD. Parents of children with FXS and ASD wanted to help with research that could help not only their child, but also children in the greater IDD community. We plan to use the findings as the basis for an OFXW survey later this year.

Tell Us What You Think

We'd love to hear your thoughts on the research results we're presenting this month. Please consider sharing your questions or comments with us.

Do any of the studies described here sound like they may be relevant to your life? What other research do you think needs to be done on fragile X? We hope to hear from you!