Objective assessments of longitudinal outcome in Gilles de la Tourette's syndrome

Autor: Sue Leurgans, Eric J. Pappert, Elan D. Louis, Lucy M. Blasucci, Christopher G. Goetz
Rok vydání: 2003
Předmět:
Zdroj: Neurology. 61:936-940
ISSN: 1526-632X
0028-3878
Popis: Objective: To define the long-term outcome in Gilles de la Tourette syndrome (GTS) using objective rating measures. Background: Previous historical studies suggest spontaneous improvement of tic symptoms after adolescence, but objective longitudinal data are limited. Methods: The authors reviewed all videotapes in their database (1978 through 1991) of children with GTS (ages 8 to 14) who were seen in their tertiary care movement disorder center and underwent a standardized 5-minute filming protocol (n = 56). Through multiple contact methods, they successfully located 36 of these patients, who are now adults (age >20 years), and recruited 31 (28 men and 3 women) to volunteer for a second videotape and in-person assessment. A blinded rater evaluated the 62 tapes and rated five tic domains: body areas involved, motor and phonic tic frequency, and motor and phonic tic severity. Using standardized GTS videotape rating scale and Wilcoxon signed-rank tests with Bonferroni correction for multiple comparisons, the authors compared the two videotapes for each tic domain as well as the composite tic disability score. Results: Ninety percent of adult patients still had tics. Adult patients who considered themselves tic-free were often inaccurate in their self-assessment: 50% had objective evidence of tics. Mean objective tic disability diminished in comparison to childhood (mean composite tic disability score childhood 9.58 vs adulthood 7.52, p = 0.014). All domains improved by adulthood, and significant improvements occurred in motor tic severity (p = 0.008). The improvements in tic disability did not relate to medication use, as only 13% of adults received medications for tics, compared with 81% of children. Conclusions: In GTS syndrome, tics objectively improve over time but most adults have persistent tics.
Databáze: OpenAIRE