Home-Based, Computerized Working Memory Programs Viable for Neurofibromatosis Type 1

Participants in the pilot study saw improvements in short-term memory, attention, and executive functioning.

A recent study concluded that computerized, home-based working memory (WM) programs may be an effective treatment method for children with neurofibromatosis type 1 (NF1) and other cognitive impairments. 

A total of 31 children were screened ahead of the pilot, open-label, prospective, pre-post-test study, and all participants were between the ages of 8 and 15 years, with a median age of 10.97 years (standard deviation [SD], 2.51). CogmedRM, the training program utilized, was completed by 27 eligible children (87%) over the course of 9 weeks. 

When evaluating for safety and feasibility for children with NF1, it was found that patients who completed cognitive training and follow-up testing (n = 20) saw significant improvements in short-term memory, attention, and executive functioning. Improvements were seen on both the Weschler Intelligence Scale for Children (WISC-IV) Digit Span (t = 2.49; P <.05) and WISC-IV Digit Span Forwards (t = 3.16; P <.01) from baseline to post-intervention. When evaluating Reliable Change Index metrics, investigators found that no participants declined in Digit Span Forwards scores and 25.9% of participants showed clinically significant improvements. 

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“Given the findings of the current pilot study, at-home cognitive training programs seems to be beneficial for children with NF1; however, continued empirical support for the intervention is still needed,” corresponding author Kristina K. Hardy, PhD, pediatric psychiatrist, division of neuropsychology, Children’s National Hospital; and associate professor, departments of psychiatry and behavioral sciences and pediatrics, the George Washington University School of Medicine, Washington, DC, et al wrote. “Future research should continue to explore the efficacy, generalizability, and maintenance of such programs.”

Improvements were also seen on in Cogstate tasks, specifically on identification speed (t = 3.49; P <.01) and accuracy (t = 2.48; P <.05). Participants further demonstrated improvements in one card learning speed (t = 2.44; P <.05) and Groton Maze Learning Errors (t = -2.74; P <.05). 

On average, participants completed 19.65 (SD, 7.18) CogmedRM sessions, with 18 participants (69%) completing 20 out of 25 sessions (80%), deeming them adherent to training guidelines. Investigators noted that 11 participants (42%) completed all 25 training sessions and that high adherence rates were similar to that of other studies utilizing computerized cognitive intervention. 

The CogmedRM program utilizes game-like tasks, with participants expected to take about 30 to 45 minutes to complete 8 exercises during a single session. The computer-based program also integrated phone-based coaching for participants and families with trained study staff to provide support and foster both motivation and efficiency.

Over the course of the study, no serious adverse events were reported. Parents and participants also completed a feasibility and acceptability interview following the intervention, with 90% of respondents rating their child’s experience as “somewhat” or “very’ satisfactory. 

The study was limited due to its small sample size and potential limited generalizability due to children being from one specialty clinic. Investigators note future studies should be larger, further exploring the interaction between pharmacological interventions and cognitive interventions, as many children with NF1 often take stimulant medications.

“If cognitive training programs, such as CogmedRM, are found to be efficacious, either on their own or—especially—in conjunction with other medications, this intervention could and should be rapidly translated into clinical practice,” Hardy et al wrote. “In addition, the availability of home-based interventions has recently become more salient in the context of the COVID-19 pandemic; thus, interventions like CogmedRM that can be accessed remotely may be increasingly accessible and acceptable to patients and families. Such an efficacious intervention for children and adolescents at high risk for neurocognitive deficits could be included with standard of care practices for at-risk individuals, with the hope of remediating deficits and improving quality of life and everyday functioning.”

REFERENCE
Hardy KK, Berger C, Griffin D, et al. Computerized working memory training for children with neurofibromatosis type 1 (NF1): A pilot study. J Child Neurol. Published online September 2, 2021. doi:10.1177/08830738211038083