Movement Disorders - Childhood Rating Scale 4-18 revised in children with dyskinetic cerebral palsy

Battini, Roberta; Sgandurra, Giuseppina; Menici, Valentina; Scalise, Roberta; Pietro, Roberta Di; Lucibello, Simona; Giannini, Maria Teresa; Cioni, Giovanni

doi:10.23736/s1973-9087.20.06079-7

Cited by 14 publications

(17 citation statements)

References 29 publications

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“…There are several rating scale assessments and diagnostic tools available for people with DCP. 1,[23][24][25][26][27][28] These rating scales and tools are valuable, but each has limitations, including lengthiness, poor clinical sensitivity, deficient differentiation of dystonia from other dyskinesias or tone patterns, and variable measurements of dystonia characteristics (e.g., amplitude, duration, and severity). Reliable assessments for dystonia are valuable both clinically and as clinical trial outcome measures.…”

Section: Current State Of the Research/research Gapsmentioning

confidence: 99%

Top 10 Research Themes for Dystonia in Cerebral Palsy

et al. 2022

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Dystonia in cerebral palsy (DCP) is a common, debilitating, but under-studied condition. The CP community (people with CP and caregivers) is uniquely equipped to help determine the research questions that best address their needs. We developed a community-driven DCP research agenda using the well-established James Lind Alliance methodology. CP community members, researchers, and clinicians were recruited via multiple advocacy, research, and professional organizations. To ensure shared baseline knowledge, participants watched webinars outlining our current knowledge on DCP prepared by a Steering Group of field experts ( cprn.org/research-cp-dystonia-edition). Participants next submitted their remaining uncertainties about DCP. These were vetted by the Steering Group and consolidated to eliminate redundancy to generate a list of unique uncertainties which were then prioritized by the participants. The top prioritized uncertainties were aggregated into themes via iterative consensus-building discussions within the Steering Group. 166 webinar viewers generated 67 unique uncertainties. 29 uncertainties (17 generated by community members) were prioritized higher than their randomly matched pairs. These were coalesced into the following Top 10 DCP Research Themes:1) Develop new treatments;2) Assess rehabilitation, psychological, and environmental management approaches;3) Compare effectiveness of current treatments;4) Improve diagnosis and severity assessments;5) Assess the impact of mixed tone (spasticity and dystonia) in outcomes and approaches;6) Assess predictors of treatment responsiveness;7) Determine pathophysiologic mechanisms;8) Describe the natural history;9) Develop dystonia pain treatments;10) Increase family awareness.This community-driven research agenda reflects the concerns most important to the community, both in perception and in practice. We therefore encourage future DCP research to center around these themes. Furthermore, noting that community members (not clinicians or researchers) generated the majority of top-prioritized uncertainties, our results highlight the important contributions community members can make to research agendas, even beyond DCP.

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Section: Current State Of the Research/research Gapsmentioning

confidence: 99%

Top 10 Research Themes for Dystonia in Cerebral Palsy

et al. 2022

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“…Class 1 included a 0–0.2 index (healthy), class 2 a 0.2–0.4 index (mildly affected), class 3 a 0.4–0.6 index (moderately affected), class 4 a 0.6–0.8 index (severely affected), and class 5 a 0.8–1 index. The MD-CRS has successfully been used in children with various movement disorders due to different etiologies, demonstrating to be a sensitive tool to detect different profiles of drug response, quantifying the rate of changes in these patients [ 11 , 12 ]. By providing a general assessment and a movement-disorder severity scoring, our child initially reported a final Global Index (Class II, mildly affected).…”

Section: Case Reportmentioning

confidence: 99%

Treatment of Dystonia Using Trihexyphenidyl in Costello Syndrome

et al. 2020

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Costello syndrome (CS), a rare syndrome with multisystemic involvement inherited as a dominant trait, is characterized by developmental delay, coarse facial appearance, cardiac defects including hypertrophic cardiomyopathy, skin abnormalities, brain complications, and a predisposition to certain malignancies. The musculoskeletal system is particularly affected in CS, with peculiar orthopedic anomalies that impact posture and gait. Dystonia has been recently documented to contribute to abnormal postures and musculoskeletal anomalies characterizing CS, suggesting the possible use of pharmacological treatments to treat these complications. We report the case of a child affected by CS displaying a particularly severe musculoskeletal involvement with dystonic posture especially in the arms and legs. The Movement Disorder-Childhood Rating Scale (MD-CRS) and a gait analysis were used to assess clinical patterns of hyperkinetic movement disorder and dystonia. The child was further treated with trihexyphenidyl for six months with a final dosage of 14 mg. MD-CRS and gait analysis assessments provided evidence for a significant improvement of posture and the related musculoskeletal problems with no side effects. Our preliminary study report provides first evidence that pharmacological anti-dystonia treatment significantly improves movement and posture disorders in patients with CS. Further studies enrolling larger cohorts of patients should be performed to validate these preliminary observations.

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“…To fill this gap, in 2008, the Childhood Movement Disorders Rating Scale (MD-CRS) was proposed and has been recently updated in a revised form (MD-CRS 4-18 R). The scale aimed to define the functional impairment and the severity produced by movement disorder (e.g., dystonia and choreoathetosis) in the pediatric population (11,(13)(14)(15). The psychometric properties in DCP of MD-CRS 4-18 R have been recently published (15).…”

Section: Introductionmentioning

confidence: 99%

A Retrospective Longitudinal Study in a Cohort of Children With Dyskinetic Cerebral Palsy Treated With Tetrabenazine

et al. 2021

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Tetrabenazine has been studied with a variety of hyperkinetic movement disorders, but there is limited and empirical literature on the potential efficacy of tetrabenazine in children with dyskinetic cerebral palsy (DCP). The purpose of this study was to evaluate the efficacy of tetrabenazine in a sample of children with DCP using the Movement Disorders—Childhood Rating Scale 4–18 Revised (MD-CRS 4–18 R). The study is a multicenter retrospective longitudinal study in which the participants were selected from the databases of each Center involved, according to detailed inclusion criteria. The study was performed on 23 children and adolescents (19 male and 4 females; mean age 8.28 years, SD 3.59) with DCP having been evaluated before starting the treatment (baseline), after 6 and 12 months of treatment and in a sub-cohort after >2 years follow-up. A linear mixed model was used to evaluate the effects of the different timings on each MD-CRS 4–18 R Index (Index I, Index II, and Global Index) adding age and type of movement disorder as random effect. A significant clinical improvement related to a reduction of MD-CRS 4-18 R Indexes was detected between the baseline and after 6 and 12 months of treatment. Findings support the efficacy of tetrabenazine in children with DCP through a standardized outcome measure (MD-CRS 4–18 R) and confirm the use of this scale as a suitable tool to detect changes in further randomized clinical trials.

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Movement Disorders - Childhood Rating Scale 4-18 revised in children with dyskinetic cerebral palsy

Cited by 14 publications

References 29 publications

Top 10 Research Themes for Dystonia in Cerebral Palsy

Top 10 Research Themes for Dystonia in Cerebral Palsy

Treatment of Dystonia Using Trihexyphenidyl in Costello Syndrome

A Retrospective Longitudinal Study in a Cohort of Children With Dyskinetic Cerebral Palsy Treated With Tetrabenazine

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