Intrathecal baclofen versus selective dorsal rhizotomy for children with cerebral palsy who are nonambulant: a systematic review

2020 ◽  
Vol 25 (1) ◽  
pp. 69-77
Author(s):  
Benjamin Davidson ◽  
Nathan Schoen ◽  
Shaina Sedighim ◽  
Renée Haldenby ◽  
Blythe Dalziel ◽  
...  
Keyword(s):  
Motor Function ◽  
Case Series ◽  
Intrathecal Baclofen ◽  
Risk Of Bias ◽  
Inclusion Criteria ◽  
Selective Dorsal Rhizotomy ◽  
Gross Motor ◽  
Gross Motor Function ◽  
Dorsal Rhizotomy ◽  
Gmfcs Level

OBJECTIVECerebral palsy (CP) is the most common childhood physical disability. Historically, children with hypertonia who are nonambulatory (Gross Motor Function Classification System [GMFCS] level IV or V) were considered candidates for intrathecal baclofen (ITB) therapy to facilitate care and mitigate discomfort. Selective dorsal rhizotomy (SDR) was often reserved for ambulant children to improve gait. Recently, case series have suggested SDR as an alternative to ITB in selected children functioning at GMFCS level IV/V. The objective for this study was to systematically review the evidence for ITB and SDR in GMFCS level IV or V children.METHODSMedline, Embase, Web of Science, and Cochrane databases were systematically searched. Articles were screened using the following inclusion criteria: 1) peer-reviewed articles reporting outcomes after SDR or ITB; 2) outcomes reported using a quantifiable scale or standardized outcome measure; 3) patients were < 19 years old at the time of operation; 4) patients had a diagnosis of CP; 5) patients were GMFCS level IV/V or results were reported based on GMFCS status and included some GMFCS level IV/V patients; 6) article and/or abstract in English; and 7) primary indication for surgery was hypertonia. Included studies were assessed with the Risk of Bias in Non-Randomized Studies - of Interventions (ROBINS-I) tool.RESULTSTwenty-seven studies met inclusion criteria. The most commonly reported outcomes were spasticity (on the Mean Ashworth Scale) and gross motor function (using the Gross Motor Function Measure), although other outcomes including frequency of orthopedic procedures and complications were also reported. There is evidence from case series that suggests that both ITB and SDR can lower spasticity and improve gross motor function in this nonambulatory population. Complication rates are decidedly higher after ITB due in part to the ongoing risk of device-related complications. The heterogeneity among study design, patient selection, outcome selection, and follow-up periods was extremely high, preventing meta-analysis. There are no comparative studies, and meaningful health-related quality of life outcomes such as care and comfort are lacking. This review is limited by the high risk of bias among included studies. Studies of SDR or ITB that did not clearly describe patients as being GMFCS level IV/V or nonambulatory were excluded.CONCLUSIONSThere is a lack of evidence comparing the outcomes of ITB and SDR in the nonambulatory CP population. This could be overcome with standardized prospective studies using more robust methodology and relevant outcome measures.

Short- and long-term effects of selective dorsal rhizotomy on gross motor function in ambulatory children with spastic diplegia

2011 ◽  
Vol 7 (5) ◽  
pp. 557-562 ◽  
Author(s):  
Petra E. M. van Schie ◽  
Maaike Schothorst ◽  
Annet J. Dallmeijer ◽  
R. Jeroen Vermeulen ◽  
Willem J. R. van Ouwerkerk ◽  
...  
Keyword(s):  
Botulinum Toxin ◽  
Motor Function ◽  
Botulinum Toxin Type A ◽  
Spastic Diplegia ◽  
Selective Dorsal Rhizotomy ◽  
Gross Motor ◽  
Gross Motor Function ◽  
Dorsal Rhizotomy

Object The primary aim of this prospective cohort study was to evaluate the short-term (1 year) and long-term (mean 6 years) effects of selective dorsal rhizotomy (SDR) on gross motor function and spasticity in ambulatory children with spastic diplegia. Secondary aims were to investigate side effects, additional treatment during follow-up (botulinum toxin type A injections or orthopedic surgery), and parental satisfaction. Methods Thirty-three children who had undergone SDR at a mean age of 6 years and 7 months (± 2 years) were included. There were 7 children at Gross Motor Function Classification System (GMFCS) Level I, 7 at Level II, and 19 at Level III. Gross motor function was assessed with the Gross Motor Function Measure–66 (GMFM-66). Spasticity was measured according to a modified Tardieu scale. Side effects, additional treatment, and parental satisfaction were recorded using a parental questionnaire and medical records. Results At 1-year follow-up, mean GMFM-66 scores improved significantly by 4.3 ± 4.1 points. Children at GMFCS Levels I and II showed significantly more improvement (7.2 points) on the GMFM-66 compared with children at GMFCS Level III (2.9 points). On long-term follow-up (mean 6 years ± 22 months), mean GMFM-66 scores improved significantly by 6.5 ± 5.9 points, without a difference between children at GMFCS Levels I and II and Level III. No relapse of spasticity was noted. Ten children (30%) needed orthopedic surgery and 13 children (39%) received botulinum toxin type A treatment after SDR. Twenty (91%) of the 22 parents who answered the questionnaire at long-term follow-up believed that their child's functioning had improved after SDR. Conclusions Selective dorsal rhizotomy resulted in short- and long-term improvements in gross motor function, without relapse of spasticity. However, the majority of the children still needed additional surgery or botulinum toxin A treatment.

scholarly journals The effect of GMFCS level, age, sex, and dystonia on multi-dimensional outcomes after selective dorsal rhizotomy: prospective observational study

2021 ◽  
Vol 37 (5) ◽  
pp. 1729-1740
Author(s):  
Conor Scott Gillespie ◽  
◽  
Alan Matthew George ◽  
Benjamin Hall ◽  
Steven Toh ◽  
...  
Keyword(s):  
Motor Function ◽  
Self Care ◽  
Pediatric Neurosurgery ◽  
Care Quality ◽  
Fine Motor ◽  
Selective Dorsal Rhizotomy ◽  
Dorsal Rhizotomy ◽  
Significant Difference ◽  
Age Category ◽  
Gmfcs Level

Abstract Purpose Investigate the effect of age category (1–9 years vs 10–18 years), sex, Gross Motor Function Classification System (GMFCS) level, and presence of dystonia on changes in eight function test parameters 24 months after selective dorsal rhizotomy (SDR). Methods Prospective, single-center study of all children aged 3–18 years with bilateral cerebral palsy with spasticity who underwent SDR at a tertiary pediatric neurosurgery center between 2012 and 2019. A linear mixed effects model was used to assess longitudinal changes. Results From 2012 to 2019, 42 children had follow-up available at 24 months. Mean GMFM-66 scores increased after SDR (mean difference 5.1 units: 95% CI 3.05–7.13, p < 0.001). Statistically significant improvements were observed in CPQoL, PEDI Self-care and Mobility, 6MWT, Gillette, and MAS scores. There was no significant difference in the improvements seen for age category, sex, GMFCS level, and presence of dystonia for most of the parameters tested (5/8, 6/8, 5/8, and 6/8 respectively). Conclusion SDR may improve gross and fine motor function, mobility and self-care, quality of life, and overall outcome based on extensive scoring parameter testing at 24 months. Atypical patient populations may benefit from SDR if appropriately selected. Multi-center, prospective registries investigating the effect of SDR are required.

Is Physical Activity-Related Self-Efficacy Associated with Moderate to Vigorous Physical Activity and Sedentary Behaviour among Ambulatory Children with Cerebral Palsy?

2021 ◽  
pp. e20200064
Author(s):  
Felipe Ganz ◽  
Virginia Wright ◽  
Patricia J. Manns ◽  
Lesley Pritchard
Keyword(s):  
Physical Activity ◽  
Sedentary Behaviour ◽  
Motor Function ◽  
Self Efficacy ◽  
Sedentary Time ◽  
Vigorous Physical Activity ◽  
Linear Regression Models ◽  
Gross Motor ◽  
Gross Motor Function ◽  
Gmfcs Level

Purpose: To determine how physical activity-related self-efficacy is associated with physical activity and sedentary behaviour time among ambulatory children with cerebral palsy (CP). Method: Children with CP, Gross Motor Function Classification System (GMFCS) Levels I-III ( N = 26; aged 9–18 y), completed the task self-efficacy component of a self-efficacy scale and wore Actigraph GT3X+ accelerometers for 5 days. Correlations (Pearson and Spearman’s rank-order; a = 0.050) were conducted to evaluate the relationships among age, GMFCS level, self-efficacy, and both daily moderate-to-vigorous physical activity (MVPA) and sedentary time. Linear regression models were used to determine the relationships among the independent variables and MVPA and sedentary time. Results: Self-efficacy was positively associated with MVPA time ( r = 0.428, p = 0.015) and negatively correlated with sedentary time ( r = –0.332, p = 0.049). In our linear regression models, gross motor function (β = –0.462, p = 0.006), age (β = –0.344, p = 0.033), and self-efficacy (β = 0.281, p = 0.080) were associated with MVPA time ( R2 = 0.508), while GMFCS level (β = 0.439, p = 0.003) and age (β = 0.605, p < 0.001) were associated with sedentary time ( R2 = 0.584). Conclusions: This research suggests that self-efficacy, age, and gross motor function are associated with MVPA in children with CP. Additional research is needed to confirm these findings and further explore the influence of self-efficacy on sedentary behaviour.

scholarly journals Improvement in Fine Manual Dexterity in Children with Spinal Muscular Atrophy Type 2 after Nusinersen Injection: A Case Series

Children ◽  
2021 ◽  
Vol 8 (11) ◽  
pp. 1039
Author(s):  
Minsu Gu ◽  
Hyun-Ho Kong
Keyword(s):  
Spinal Muscular Atrophy ◽  
Motor Function ◽  
Daily Living ◽  
Muscular Atrophy ◽  
Case Series ◽  
Manual Dexterity ◽  
Gross Motor ◽  
Spinal Muscular Atrophy Type ◽  
Gross Motor Function

Although nusinersen has been demonstrated to improve motor function in patients with spinal muscular atrophy (SMA), no studies have investigated its effect on fine manual dexterity. The present study aimed to investigate the ability of nusinersen to improve fine manual dexterity in patients with SMA type 2. A total of five patients with SMA type 2 were included. The Hammersmith Functional Motor Scale (expanded version) (HFMSE) and Purdue Pegboard (PP) tests were used to evaluate gross motor function and fine manual dexterity, respectively, until 18 months after nusinersen administration. HFMSE scores improved by 3–10 points (+13–53%) in all patients following nusinersen administration. PP scores also improved in all patients, from 4 to 9 points (+80–225%) in the preferred hand and from 3 to 7 points (+60–500%) in the non-preferred hand. These results suggest that nusinersen treatment improved both gross motor function and fine manual dexterity in children with SMA type 2. Addition of the PP test may aid in evaluating the fine manual dexterity essential for activities of daily living in these patients.

scholarly journals No Increased Risk for Scoliosis After Early Selective Dorsal Rhizotomy - A Longitudinal Population-Based Controlled Registry Study from four to 25 years of Age

2020 ◽  
Author(s):  
Annika Lundkvist Josenby ◽  
Lena Westbom
Keyword(s):  
Spinal Pain ◽  
Control Group ◽  
Spastic Diplegia ◽  
Selective Dorsal Rhizotomy ◽  
Gross Motor ◽  
Dorsal Rhizotomy ◽  
Severe Spasticity ◽  
Increased Risk ◽  
Group Frequency ◽  
Gmfcs Level

Abstract Spasticity interfering with gross motor development in cerebral palsy (CP) can be reduced with selective dorsal rhizotomy (SDR). Although reported, it is unknown if SDR surgery increases the risk for later spine problems. Using CP-registry data from a geographically defined population with the same health care and habilitation services, the objectives were to compare reported scoliosis and spinal pain up to adult age in all SDR-operated with all non-SDR-operated individuals with same medical history, functional abilities, and level of spasticity at four years of age. Method In the total population with CP spastic diplegia in Skåne and Blekinge, born 1990-2006, 149 individuals had moderate to severe spasticity and no medical contraindications against SDR at four years of age and were included; 36 persons had undergone SDR at a median age of 4.0 years (range 2.5 – 6.6 years), and 113 individuals constituting the control group, had not. Data on scoliosis and spinal pain at 10, 15, 20 and 25 years of age were analyzed using Kaplan-Meier survival curves and Fisher’s exact test. Gross motor function classification (GMFCS) levels at four years of age (or pre-operatively) were used for stratification.Result Presence of scoliosis at 15, 20, and 25 years of age was the same in the SDR group as in the control group (p=0.734, 0.735 and 1.0). In severe functional disability (GMFCS IV), the SDR group had later onset and lower occurrence of scoliosis (p=0.004) than the control group. Frequency of reported spinal pain did not differ between the groups. Conclusion Neither scoliosis, nor spinal pain was more frequent after SDR than expected by natural history. On the contrary, in severe CP (GMFCS level IV), scoliosis was less frequently reported and had a later onset in the SDR group than in the same GMFCS-level control group.

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