Is intrathecal baclofen an effective treatment for children with hereditary spastic paraparesis?

Pointon, Ross Simon; Whelan, Hannah; Raza, Rushna; Lodh, Rajib

doi:10.1136/archdischild-2020-319878

Cited by 4 publications

(2 citation statements)

References 14 publications

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“…Nevertheless, there is evidence for the symptomatic relief of spasticity in children using ITB in other diseases. The doses used in ITB therapy are symptom and not disease specific, as such data on the safety of ITB in other diseases can be used to guide HSP treatment [25]. However, the use of ITB in children entails additional risks, since children may be at higher risk from over-dosing on baclofen, having infections and requiring repeated surgeries due to catheter problems [25].…”

Section: Discussionmentioning

confidence: 99%

Intrathecal baclofen for the management of hereditary spastic paraparesis: a systematic review

Viana Pinto,

Romeiro,

Gouveia

et al. 2024

International Journal of Rehabilitation Research

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This systematic review aims to evaluate the use of intrathecal baclofen (ITB) for hereditary spastic paraparesis (HSP) treatment. An extensive search in two electronical databases was performed. We identified articles published between 1990 and 2022 (PubMed, Scopus), and applied the following inclusion criteria: diagnosis of HSP at the time of the intervention, either familial or sporadic; report on the effect of ITB in patients with HSP; test trial via either bolus injections or continuous infusion tests; and ITB pump implantation. A data extraction sheet based on the Cochrane Consumers and Communication Review Group’s data extraction template was created and adapted to collect relevant data. A qualitative analysis was performed to present the results in narrative summary fashion. A total of 6 studies met our inclusion criteria. 51 patients with HSP had a pre-implantation ITB trial. The time since the diagnosis until the pump implantation ranged from 5 to 30 years. The initial bolus ranged from 20 to 50 μg and the mean doses used at steady state ranged from 65 to 705 μg. An improvement in spasticity was observed on the modified Ashworth Scale in patients treated with ITB. Although all studies reported a subjective gait improvement, not all found an objective improvement in gait. The most common side effect reported was catheter-related problems. The findings of this review support the use of ITB as an effective and a viable option for the treatment of spasticity in HSP refractory to conservative therapies.

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Section: Discussionmentioning

confidence: 99%

Intrathecal baclofen for the management of hereditary spastic paraparesis: a systematic review

Viana Pinto,

Romeiro,

Gouveia

et al. 2024

International Journal of Rehabilitation Research

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“…A recent systematic review concluded that there was a lack of evidence comparing outcomes of ITB and SDR in the management of non-ambulant CP, with a need for robustly designed prospective studies 31. Similarly, while evidence is limited to reported cases/case series, positive outcomes of ITB in the management of ambulant HSP cases have been reported 32…”

Section: Introductionmentioning

confidence: 99%

Neurosurgical management of elevated tone in childhood: interventions, indications and uncertainties

Lumsden

2023

Arch Dis Child

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Elevated tone (hypertonia) is a common problem in children with physical disabilities. Medications intended to reduce tone often have limited efficacy, with use further limited by a significant side effect profile. Consequently, there has been growing interest in the application of Neurosurgical Interventions for the Management of Posture and Tone (NIMPTs). Three main procedures are now commonly used: selective dorsal rhizotomy (SDR), intrathecal baclofen (ITB) and deep brain stimulation (DBS). This review compares these interventions, along with discussion on the potential role of lesioning surgery. These interventions variably target spasticity and dystonia, acting at different points in the distributed motor network. SDR, an intervention for reducing spasticity, is most widely used in carefully selected ambulant children with cerebral palsy. ITB is more commonly used for children with more severe disability, typically non-ambulant, and can improve both dystonia and spasticity. DBS is an intervention which may improve dystonia. In children with certain forms of genetic dystonia DBS may dramatically improve dystonia. For other causes of dystonia, and in particular dystonia due to acquired brain injury, improvements following surgery are more modest and variable. These three interventions vary in terms of their side-effect profile and reversibility. There are currently populations of children for who it is unclear which intervention should be considered (SDR vs ITB, or ITB vs DBS). Concerns have been raised as to the equity of access to NIMPTs for children across the UK, and whether the number of surgeries performed each year meets the clinical need.

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The use of intrathecal baclofen for management of spasticity in hereditary spastic paraparesis: A case series

Pointon

Whelan

Raza

et al. 2022

European Journal of Paediatric Neurology

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Is intrathecal baclofen an effective treatment for children with hereditary spastic paraparesis?

Cited by 4 publications

References 14 publications

Intrathecal baclofen for the management of hereditary spastic paraparesis: a systematic review

Intrathecal baclofen for the management of hereditary spastic paraparesis: a systematic review

Neurosurgical management of elevated tone in childhood: interventions, indications and uncertainties

The use of intrathecal baclofen for management of spasticity in hereditary spastic paraparesis: A case series

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