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

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OBJECTIVE

Cerebral 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.

METHODS

Medline, 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.

RESULTS

Twenty-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.

CONCLUSIONS

There 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.

ABBREVIATIONS CP = cerebral palsy; CPCHILD = Caregiver Priorities & Child Health Index of Life with Disabilities; GMFCS = Gross Motor Function Classification System; GMFM = Gross Motor Function Measure; ITB = intrathecal baclofen; MAS = Modified Ashworth Scale; PEDI = Pediatric Evaluation of Disability Inventory; PRISMA = Preferred Reporting Items for Systematic Reviews and Meta-Analyses; PSF = posterior spinal fusion; ROBINS-I = Risk of Bias in Non-Randomized Studies - of Interventions; ROM = range of motion; SDR = selective dorsal rhizotomy.

Supplementary Materials

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Contributor Notes

Correspondence George M. Ibrahim: University of Toronto, ON, Canada. george.ibrahim@sickkids.ca.INCLUDE WHEN CITING Published online October 18, 2019; DOI: 10.3171/2019.8.PEDS19282.Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.
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