Telehealth Can Expand Access to Behavioral Treatments for Fragile X

Telehealth, or telemedicine, can expand access to behavioral treatments for children with fragile X syndrome, a clinical study suggests.

An approach called functional communication training, or FCT, delivered via telehealth, was found to ease irritability in boys with the disorder and to lower parenting stress in their caregivers.

The study, “Telehealth-enabled behavioral treatment for problem behaviors in boys with fragile X syndrome: a randomized controlled trial,” was published in the Journal of Neurodevelopmental Disorders.

Disruptive behaviors such as self-injury, aggression, and property destruction create significant health-related problems for children with fragile X, impacting their quality of life and causing significant distress to their families. Access to appropriate treatment is severely limited by factors such as the cost of care and, in many rural populations, the shortage of qualified treatment providers.

To address these issues, researchers at Stanford University evaluated the treatment of disruptive behaviors in fragile X using telehealth — the use of communication technology, such as phones and computers, to facilitate healthcare appointments.

Their clinical trial (NCT03510156) aimed to examine the effect of administering FCT, an established behavior analytic intervention, via telehealth to boys with fragile X. Treatment acceptability and the effect of treatment on parenting stress also were assessed.

In FCT, the focus is to ensure that problem behaviors no longer result in reinforcement, while at the same time teaching the child to engage in alternative and more appropriate forms of communicative behavior with the same goals (such as having attention).

The study included 57 boys, ages 3-10 (mean age 6.8 years), who exhibited at least one problem behavior daily. Problem behaviors were defined as those that could result in tissue damage to the caregiver — for example, by the child hitting, kicking, or throwing items at the caregiver — or to the child’s own body, due to actions such as self-biting and self-hitting. Other problem behaviors included actions that could lead to damage to property, including kicking things, throwing items, and ripping books.

Most of the patients’ primary caregivers, who had a mean age of 39, were married women with college educations, living in a suburban area, and with a household income of $100,000 or less.

The boys were randomly assigned to receive FCT via telehealth (30) or usual treatment (27) over 12 weeks in one-hour sessions. Of the 30 families enrolled, 24 completed the FCT treatment.

Measures used in therapy included in-session observations of problem behaviors, the aberrant behavior checklist community (ABC-C), the treatment acceptability rating form-revised (TARF-R), and the parenting stress index, 4th edition (PSI-4).

Notably, the ABC-C is divided in five subscales: irritability, social withdrawal, stereotypic behavior (repetitive, purposeless movements), hyperactivity/noncompliance, and inappropriate speech. In turn, PSI-4 includes subscales focusing on the sources of stress related to child characteristics — such as hyperactivity, adaptability, demandingness, and mood — and subscales on sources of stress related to parent characteristics, which include competence, isolation, attachment, health, role restriction, depression, and spouse relationship.

Scores on the irritability subscale of the ABC-C — which include aggression, self-injury, tantrums, agitation, and unstable mood — decreased significantly for boys who received FCT via telehealth (42.6%) compared to boys who received treatment as usual (9.13%).

Further analysis supported these findings. The rate of a positive response, measured by improvement of at least 25% on the irritability score, was 66.7% for children in the FCT group and 29.2% in the treatment-as-usual group.

The FCT group also showed significantly greater improvements, or reductions, on stereotypic behavior (2.17 vs. 0.94 points) and hyperactivity or noncompliance (6.66 vs. 1.40 points) compared to the treatment-as-usual group.

In-session observations conducted for those who received treatment showed that levels of problem behaviors decreased by 91.7% from the beginning of treatment.

The mean rate of problem behaviors observed at the beginning of FCT was 2.70 responses per minute, which decreased steadily through the course of treatment and remained just below 0.5 by the end of the 12 weeks.

Notably, increased parent fidelity was significantly associated with greater reductions in children’s problem behaviors.

At the beginning, high scores were obtained on the reasonableness, effectiveness, and willingness subscales of the TARF-R, indicating that FCT was deemed highly reasonable and effective, and that caregivers were willing to use this approach with their child. Treatment acceptability remained high throughout the study.

In addition, FCT procedures were considered neither costly nor disruptive to the family. Side effects were minimal.

At the start of the trial, the highest sources of parental stress were related to characteristics of the child, including distractibility, hyperactivity, and demandingness.

For caregivers, significant benefits were seen on child domain and total stress, as well as on the distractibility/hyperactivity, reinforces parent, demandingness, and mood subscales following FCT treatment. In addition, significant improvements in competence were seen in boys from both groups.

A comparison of the groups indicated that caregivers in the FCT group showed significantly greater improvements on distractibility, hyperactivity and mood subscales, as well as on the child domain of the PSI-4.

“These findings support telehealth-enabled FCT as a framework for expanding access to behavioral treatments for problem behaviors in children with FXS [fragile X syndrome],” the investigators wrote.

“Expanded delivery of behavior analytic treatment via telehealth also has the potential to lower healthcare costs, improve child and family quality of life, and lead to advances in the treatment of problem behavior in the broader population of individuals with neurodevelopmental disorders,” the researchers concluded.