Goal-oriented Therapy With Brain Stimulation Improves Boy’s Walking
An intervention combining goal-oriented physical therapy with electrical stimulation of the brain via a device placed on the tongue led to marked gains in walking ability and other areas for a child with Angelman syndrome, according to a new report.
The study, “Improvement in Gait and Participation in a Child with Angelman Syndrome after Translingual Neurostimulation Associated with Goal-Oriented Therapy: A Case Report,” was published in Children.
Scientists in France described the case of a 7-year-old boy with Angelman syndrome caused by a maternal deletion in the 15q11.2-13.1 chromosomal region, the most common type of Angelman-causing mutation. The boy had severe seizures as an infant, but had been seizure-free since age 5 with a combination of anti-seizure medications (clonazepam and sodium valproate).
The child exhibited marked delays in motor development; he was not able to walk independently until age 5, and then only on flat, firm surfaces without obstacles. His gait was jerky, with an atypical forward lean and large arm movements.
The boy had severe difficulty sleeping and showed intellectual and behavioral abnormalities. He met criteria for autism spectrum disorder and displayed unusual repetitive behaviors and hyperactivity. He had no capacity for speech, and his nonverbal communication skills were also very limited. He was entirely dependent on caregivers for daily activities like dressing, feeding, and bathing.
The boy underwent a physical therapy intervention to improve his ability to walk. Sessions were performed with the supervision of a physical therapist two hours in the morning and two hours in the afternoon, five days per week, for four weeks.
During these sessions, the boy also was given translingual neurostimulation (TLNS) — a technique that uses a device on the tongue to administer gentle electrical stimulation to the brain. It’s thought to help promote synaptic plasticity, the brain’s ability to “rewire itself” in response to new information.
The researchers reported that the child showed no issues with the TLNS device; the only change noted was that he salivated more while it was in his mouth.
“The child showed no signs of discomfort during or after the stimulation sessions. … He accepted having the device placed in his mouth and participated well in rehabilitation while the stimulation was switched on,” the research team wrote.
Before the intervention, the boy underwent standardized walking assessments, using cameras and computer algorithms to objectively measure various aspects of his gait. These assessments were repeated immediately after the four-week intervention, and again six months later.
Results showed marked improvements in numerous gait-related parameters from the start to the end of the intervention. For example, the average length of the child’s steps nearly doubled (from 0.45 to 0.74 meters) and his average walking speed increased from 0.47 to 0.78 m/s. At six months post-intervention, many of these improvements remained to some extent, though some worsening was noted.
The child’s parents “reported a dramatic improvement in their son’s motor ability” over the four-week intervention, the researchers reported. These had a profound effect on day-to-day life — being able to stand upright longer made it easier for him to get dressed.
“He could walk 1 to 2 km [about a mile], including on different surfaces (snow, grass and dirt paths), and arm swing was spontaneous; he was thus able to take part in family walks and was less dependent during outings. Furthermore, he showed signs of great pleasure in walking independently,” the scientists wrote. “To our knowledge, this is the first report of a rapid (four weeks) and considerable improvement in gait ability and other voluntary and involuntary motor skills in a child with Angelman syndrome following a rehabilitation intervention.”
The parents also reported many welcome changes following the intervention that were not directly related to motor function. The child’s communication improved; he was able to consistently nod to answer “yes” or “no” questions and “suddenly attributed single syllables to around 10 specific objects and people,” the researchers said.
The child also developed the ability to eat independently with a fork and slept better at night. He also seemed less distractible and was better able to engage in activities and play.
Six months after the intervention, the parents said walking improvements and ability to eat had persisted, but other changes — particularly those concerning speech, sleep, and behavior — had disappeared over time.
“Further studies are required to understand the mechanisms underlying this improvement, how to maintain the improvement, and to determine if such results are generalizable across all children with Angelman syndrome and other syndromes that affect synaptic plasticity,” the scientists said.