Evaluation of functional goal outcomes using the Canadian Occupational Performance Measure (COPM) following Deep Brain Stimulation (DBS) in childhood dystonia

Gimeno, Hortensia; Tustin, Kylee; Lumsden, Daniel E.; Ashkan, Keyoumars; Selway, Richard; Lin, Jean-Pierre

doi:10.1016/j.ejpn.2013.12.010

Cited by 64 publications

(77 citation statements)

References 32 publications

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“…Our study population involves severely affected dyskinetic CP patients with little to no motor skills (GFMCS IV and V). 17,21 A significant difference between groups was found for the dystonia during rest subscale, but not for the dystonia during activity subscale. 9 Standardized questionnaires on quality of life or activities and participation, which were available at the time of the study, do not adequately capture the individual problems in daily life of these patients; attainment of individual goals is therefore the most useful and meaningful way to determine treatment effects.…”

Section: Discussionmentioning

confidence: 83%

The Effect of Intrathecal Baclofen in Dyskinetic Cerebral Palsy: The IDYS Trial

Bonouvrié

Becher²,

Vles

et al. 2019

Annals of Neurology

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Objective Intrathecal baclofen treatment is used for the treatment of dystonia in patients with severe dyskinetic cerebral palsy; however, the current level of evidence for the effect is low. The primary aim of this study was to provide evidence for the effect of intrathecal baclofen treatment on individual goals in patients with severe dyskinetic cerebral palsy. Methods This multicenter, randomized, double‐blind, placebo‐controlled trial was performed at 2 university medical centers in the Netherlands. Patients with severe dyskinetic cerebral palsy (Gross Motor Functioning Classification System level IV–V) aged 4 to 24 years who were eligible for intrathecal baclofen were included. Patients were assigned by block randomization (2:2) for treatment with intrathecal baclofen or placebo for 3 months via an implanted microinfusion pump. The primary outcome was goal attainment scaling of individual treatment goals (GAS T score). A linear regression model was used for statistical analysis with study site as a covariate. Safety analyses were done for number and type of (serious) adverse events. Results Thirty‐six patients were recruited from January 1, 2013, to March 31, 2018. Data for final analysis were available for 17 patients in the intrathecal baclofen group and 16 in the placebo group. Mean (standard deviation) GAS T score at 3 months was 38.9 (13.2) for intrathecal baclofen and 21.0 (4.6) for placebo (regression coefficient = 17.8, 95% confidence interval = 10.4‐25.0, p < 0.001). Number and types of (serious) adverse events were similar between groups. Interpretation Intrathecal baclofen treatment is superior to placebo in achieving treatment goals in patients with severe dyskinetic cerebral palsy. ANN NEUROL 2019

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Section: Discussionmentioning

confidence: 83%

The Effect of Intrathecal Baclofen in Dyskinetic Cerebral Palsy: The IDYS Trial

Bonouvrié

Becher²,

Vles

et al. 2019

Annals of Neurology

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“…Most studies fail to report what matters most to children with dystonia and their carers: reduction in pain, improvements in activities of daily living, and manual function (67, 68). This is a particularly important failing since dystonia worsens in approximately 2/3 of cases and remains severe in a further 1/3 of cases referred for further management after conventional medication (1).…”

Section: Measuring What Matters Most To Children With Dystonia Is Alsmentioning

confidence: 99%

Recognizing the Common Origins of Dystonia and the Development of Human Movement: A Manifesto of Unmet Needs in Isolated Childhood Dystonias

Lin

Nardocci

2016

Front. Neurol.

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Dystonia in childhood may be severely disabling and often unremitting and unrecognized. Considered a rare disorder, dystonic symptoms in childhood are pervasive in many conditions including disorders of developmental delay, cerebral palsy (CP), autism, neurometabolic, neuroinflammatory, and neurogenetic disorders. Collectively, there is a need to recognize the role of early postures and movements which characterize phases of normal fetal, infant, and child development as a backdrop to the many facets of dystonia in early childhood neurological disorders and to be aware of the developmental context of dystonic symptoms. The role of cocontraction is explored throughout infancy, childhood, young adulthood, and in the elderly. Under-recognition of pervasive dystonic disorders of childhood, including within CP is reviewed. Original descriptions of CP by Gowers are reviewed and contemporary physiological demonstrations are used to illustrate support for an interpretation of the tonic labyrinthine response as a manifestation of dystonia. Early recognition and molecular diagnosis of childhood dystonia where possible are desirable for appropriate clinical stratification and future precision medicine and functional neurosurgery where appropriate. A developmental neurobiological perspective could also be useful in exploring new clinical strategies for adult-onset dystonia disorders focusing on environmental and molecular interactions and systems behaviors.

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“…Although the Barry–Albright Dystonia Scale was designed to apply more easily to children, it does not seem to be as sensitive to changes after DBS, most likely because the combination of provoking and severity factors in the Burke–Fahn–Marsden Scale leads to a more fine‐grained outcome scale. Functional or goal attainment scales may have greater sensitivity to the goals of individual patients . The variability and difficulty in quantifying dystonia, the presence of other movement disorders such as spasticity or weakness, and the developmental and cognitive effects of long‐standing motor deficits make full evaluation of the effects of DBS difficult .…”

Section: Discussionmentioning

confidence: 99%

Deep brain stimulation for cerebral palsy: where are we now?

Sanger

2019

Develop Med Child Neuro

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DBS Deep brain stimulation GPiInternal globus pallidusCerebral palsy (CP) is a complex disorder and children frequently have multiple impairments. Dystonia is a particularly frustrating impairment that interferes with rehabilitation and function and is difficult to treat. Of the available treatments, deep brain stimulation (DBS) has emerged as an option with the potential for large effect size in a subgroup of children. While brain stimulation has been used in CP for more than 40 years, modern devices and targeting methods are improving both the safety and efficacy of the procedure. Successful use of DBS depends on appropriate selection of patients, identification of effective neuroanatomical targets in each patient, careful neurosurgical procedure, and detailed follow-up evaluation and programming. The use of functional neurosurgery for neuromodulation in CP remains a technology in its infancy, but improving experience and knowledge are likely to make this one of the safest and most effective interventions for children with moderate-to-severe motor disorders. This review summarizes the current procedures for patient and target selection, and surgical implantation of DBS electrodes for CP. The history of DBS and future directions when used in secondary dystonia are also examined.

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Evaluation of functional goal outcomes using the Canadian Occupational Performance Measure (COPM) following Deep Brain Stimulation (DBS) in childhood dystonia

Cited by 64 publications

References 32 publications

The Effect of Intrathecal Baclofen in Dyskinetic Cerebral Palsy: The IDYS Trial

The Effect of Intrathecal Baclofen in Dyskinetic Cerebral Palsy: The IDYS Trial

Recognizing the Common Origins of Dystonia and the Development of Human Movement: A Manifesto of Unmet Needs in Isolated Childhood Dystonias

Deep brain stimulation for cerebral palsy: where are we now?

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