ABSTRACT
IMPORTANCE: People with Barth syndrome (BTHS) present with sensory and motor deficits that affect their ability to swallow medications in solid form. Inability to swallow pills can reduce opportunities for people with BTHS to participate in clinical trial research. OBJECTIVE: To evaluate the effectiveness of a brief, multifaceted pill-swallowing program that used evidence-based training methods. DESIGN: Pretest-posttest with 6-mo follow-up. SETTING: Community setting. PARTICIPANTS: A convenience sample of children, adolescents, and adults with a genetically confirmed diagnosis of BTHS. INTERVENTION: Possible intervention strategies included behavioral approaches (e.g., shaping), adaptive approaches, and positioning approaches. Interventions were tailored to each participant's needs and were carried out by occupational therapy practitioners with advanced training in feeding and eating. OUTCOMES AND MEASURES: Pill-swallowing milestones were ranked on a scale ranging from 0 to 12; participants were scored pretraining, immediately posttraining, and at 6-mo posttraining. RESULTS: Sixteen participants with BTHS, ages 6-34 yr, participated in the training. Fourteen of the 16 participants demonstrated improvement in their pill-swallowing ability. Overall, there was a statistically significant change in pill-swallowing ability from pretraining to posttraining, and these changes were maintained after 6 mo. CONCLUSION: and Relevance: This study suggests that a brief multifaceted training approach, led by trained professionals, may be effective for helping people with sensory and motor deficits learn to swallow pills independently. WHAT THIS ARTICLE ADDS: Very little has been published in the occupational therapy literature describing how to teach the skill of pill swallowing to clients or how to measure progress toward this goal. This study tested the outcomes of a multifaceted approach to pill swallowing that can be conducted by occupational therapy practitioners; a novel measurement approach is also introduced that can be used with clients in clinical practice.
