Harvey S Singer1, Shreenath Rajendran2, H Richard Waranch2, E Mark Mahone3. 1. Johns Hopkins University School of Medicine, Baltimore, Maryland. Electronic address: hsinger@jhmi.edu. 2. Johns Hopkins University School of Medicine, Baltimore, Maryland. 3. Johns Hopkins University School of Medicine, Baltimore, Maryland; Kennedy Krieger Institute, Baltimore, Maryland.
Abstract
BACKGROUND: Complex motor stereotypies (CMS) typically begin before age three years and include rhythmic, repetitive, fixed movements that last for seconds to minutes and can be interrupted with distraction. OBJECTIVE: We evaluated the effectiveness of a home-based, parent-provided therapy accompanied by scheduled telephone calls with a therapist, in five- to seven-year old children with primary CMS. METHODS: Eligible families received an instructional digital versatile disk (DVD) written instructions, and scheduled telephone contacts with a therapist at baseline (DVD receipt), one, three, and eight weeks later. At each call, parents completed outcome measures and received feedback. Outcome scales Stereotypy Severity Scale (SSS) Motor and Impairment scales and a Stereotypy Linear Analogue Scale (SLAS) were also completed via the Iinternet (REDCap)-at screening, one and two months post-baseline call. At study conclusion, participants were divided into an intent-to-treat (ITT; had at least one call) or a lost-to-follow-up (LTF) group. RESULTS: Thirty-eight children (mean = 6 years ± 11 months) were enrolled. The LTF group (n = 14) had significantly higher scores than the ITT (n = 24) group on all attention-deficit/hyperactivity disorder ratings (P < 0.01), but not stereotypy severity. Primary outcome scores, acquired by telephone and REDCap, showed a significant reduction in SSS Motor and Impairment scores between the initial and the last completed evaluation (P ≤ 0.001). Calculated change ratios were SSS Motor -0.23/-0.30 (cal/REDCap); SSS Impairment -0.31/-0.32; and SLAS -0.54 (REDCap). Clinical improvement was further supported by results from a parent improvement scale and end of study questionnaires. CONCLUSIONS: Home-based, parent-administered behavioral therapy supplemented by telephone contact with a therapist is effective in reducing complex motor stereotypies in children.
BACKGROUND: Complex motor stereotypies (CMS) typically begin before age three years and include rhythmic, repetitive, fixed movements that last for seconds to minutes and can be interrupted with distraction. OBJECTIVE: We evaluated the effectiveness of a home-based, parent-provided therapy accompanied by scheduled telephone calls with a therapist, in five- to seven-year old children with primary CMS. METHODS: Eligible families received an instructional digital versatile disk (DVD) written instructions, and scheduled telephone contacts with a therapist at baseline (DVD receipt), one, three, and eight weeks later. At each call, parents completed outcome measures and received feedback. Outcome scales Stereotypy Severity Scale (SSS) Motor and Impairment scales and a Stereotypy Linear Analogue Scale (SLAS) were also completed via the Iinternet (REDCap)-at screening, one and two months post-baseline call. At study conclusion, participants were divided into an intent-to-treat (ITT; had at least one call) or a lost-to-follow-up (LTF) group. RESULTS: Thirty-eight children (mean = 6 years ± 11 months) were enrolled. The LTF group (n = 14) had significantly higher scores than the ITT (n = 24) group on all attention-deficit/hyperactivity disorder ratings (P < 0.01), but not stereotypy severity. Primary outcome scores, acquired by telephone and REDCap, showed a significant reduction in SSS Motor and Impairment scores between the initial and the last completed evaluation (P ≤ 0.001). Calculated change ratios were SSS Motor -0.23/-0.30 (cal/REDCap); SSS Impairment -0.31/-0.32; and SLAS -0.54 (REDCap). Clinical improvement was further supported by results from a parent improvement scale and end of study questionnaires. CONCLUSIONS: Home-based, parent-administered behavioral therapy supplemented by telephone contact with a therapist is effective in reducing complex motor stereotypies in children.
Authors: Christopher Oakley; E Mark Mahone; Christina Morris-Berry; Tina Kline; Harvey S Singer Journal: Pediatr Neurol Date: 2014-12-19 Impact factor: 3.372
Authors: E Mark Mahone; Deana Crocetti; Laura Tochen; Tina Kline; Stewart H Mostofsky; Harvey S Singer Journal: Pediatr Neurol Date: 2016-09-08 Impact factor: 3.372
Authors: A D Harris; H S Singer; A Horska; T Kline; M Ryan; R A E Edden; E M Mahone Journal: AJNR Am J Neuroradiol Date: 2015-11-05 Impact factor: 3.825
Authors: Shannon L Dean; Laura Tochen; Farhan Augustine; Syed F Ali; Deana Crocetti; Shreenath Rajendran; Mary E Blue; E Mark Mahone; Stewart H Mostofsky; Harvey S Singer Journal: Cerebellum Date: 2021-08-14 Impact factor: 3.847