Can children with spina bifida and muscular dystrophy participate in their own health status evaluations?

Abstract

The outcomes movement has emphasized the importance of the patient in evaluating treatment outcome. However, concern has been raised about the ability of children, particularly those with multisystem disease, to evaluate their disability. To determine whether children with spina bifida and muscular dystrophy and their parents agree when asked about the relative difficulty of daily activities and the severity of symptoms experienced by the child. First, a list of symptoms and activity restrictions was generated from the literature, clinicians, and interviews with families with spina bifida and muscular dystrophy. Second, another group of parents and children with spina bifida (with hip dislocation or scoliosis) and with muscular dystrophy (with scoliosis), including those before and after surgery, independently rated the severity and importance of their objective and subjective complaints. The correlation between parents and their children was high for both objective (median Spearman's = 0.70; standard deviation [SD] = ±0.17; range = -0.05-1.00) and subjective (median Spearman's = 0.76; SD = ±0.14; range = 0.13-1.00) complaints, with an overall excellent level of agreement (Kappa = 0.75; 95% confidence interval [CI]: 0.73, 0.76). Children with spina bifida and muscular dystrophy are capable of understanding and assessing their disability.