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. 2019 Feb;61(2):128-134.
doi: 10.1111/dmcn.14005. Epub 2018 Sep 6.

Effect of continuous intrathecal baclofen therapy in children: a systematic review

Annemieke I Buizer  1 , Brian H M Martens  2 , Casey Grandbois van Ravenhorst  1 , Linda J Schoonmade  3 , Jules G Becher  1 , R Jeroen Vermeulen  2
Affiliations

Effect of continuous intrathecal baclofen therapy in children: a systematic review

Annemieke I Buizer et al. Dev Med Child Neurol. 2019 Feb.

Abstract
in English, Spanish, Portuguese

Aim: To investigate the effects of continuous intrathecal baclofen (ITB) therapy in children with cerebral palsy (CP) and other neurological conditions.

Method: This systematic review was conducted using standardized methodology, searching four electronic databases (PubMed, Embase, CINAHL, Cochrane Library) for relevant literature published between inception and September 2017. Included studies involved continuous ITB as an intervention and outcome measures relating to all International Classification of Functioning, Disability and Health: Children and Youth (ICF-CY) components.

Results: Thirty-three studies were identified, of which one, including 17 children with spastic CP, produced level II evidence, and the others, mainly non-controlled cohort studies, level IV and V. Outcomes at body function level were most frequently reported. Results suggest continuous ITB may be effective in reducing spasticity and dystonia in CP, as well as other neurological conditions, and may improve the ease of care and quality of life of children with CP, but the level of evidence is low.

Interpretation: Despite three decades of applying ITB in children and a relatively large number of studies investigating the treatment effects, a direct link has not yet been demonstrated because of the low scientific quality of the primary studies. Further investigation into the effects of continuous ITB at all levels of the ICF-CY is warranted. Although large, controlled trials may be difficult to realize, national and international collaborations may provide opportunities. Also, multicentre prospective cohort studies with a long-term follow-up, employing harmonized outcome measures, can offer prospects to expand our knowledge of the effects of continuous ITB therapy in children.

What this paper adds: There is low-level evidence for continuous intrathecal baclofen (ITB) in children with cerebral palsy. Continuous ITB is effective in reducing spasticity and dystonia in non-controlled cohort studies. Evaluation of individual goals and systematic assessment of long-term effects in large cohort studies are required.

Objetivo: Investigar los efectos de la terapia con baclofeno intratecal continuo (BIT) en niños con parálisis cerebral (PC) y otras condiciones neurológicas.

Metodo: Esta revisión sistemática utilizo metodología estandarizada, buscando en cuatro bases de datos electrónicas (PubMed, Embase, CINAHL, Cochrane Library) literatura relevante publicada entre el comienzo de BIT y septiembre del 2017. Los estudios incluidos involucraron BIT continuo como una intervención y la medición de resultados incluía todos los componentes relacionados a la Clasificación Internacional del Funcionamiento, de la Discapacidad y de la Salud para niños y jóvenes (ICF‐CY en inglés).

Resultados: Treinta y tres estudios fueron identificados, de los cuales uno, incluyo 17 niños con PC espástica, produciendo un nivel II de evidencia, y los otros, principalmente estudios de cohorte no controlados nivel IV y V. Los resultados sobre el nivel de función corporal, fue reportado con mayor frecuencia. Los resultados sugieren que la BIT continua puede ser efectiva en reducir la espasticidad y la distonía en PC, así también en otras condiciones neurológicas, y puede facilitar el cuidado y la calidad de vida de los niños con PC, pero el nivel de evidencia es bajo.

Interpretación: A pesar de tres décadas de aplicación de BIT en niños, con un relativo largo número de estudios investigando los efectos del tratamiento, el impacto terapéutico aún no ha sido bien demostrada debido a la baja calidad científica de los estudios primarios. Investigaciones posteriores sobre el efecto de la BIT continúa en todos los niveles de la ICF‐CY son necesarios. A pesar de que estudios controlados prolongados pueden ser difíciles de realizar, colaboración nacional e internacional puede proveer oportunidades para su realización. Además, estudios de cohortes prospectivos con un seguimiento a largo plazo, utilizando mediciones de resultados estandarizadas, pueden ofrecer perspectivas que expandan nuestro conocimiento sobre los efectos de la terapia continua con BIT en niños.

Objetivo: Investigar os efeitos da terapia com baclofeno intratecal (BIT) contínuo em crianças com paralisia cerebral (PC) e outras condições neurológicas.

Método: Esta revisão sistemática foi realizada usando metodologia padronizada, buscando quatro bases de dados (PubMed, Embase, CINAHL, Cochrane Library) pela literatura relevante publicada desde o início até setembro de 2017. Os estudos incluídos envolveram BIT contínuo como intervenção, e medidas de resultado relacionadas a todos os components da Classificação Internacional de Funcionalidade, Incapacidade e Saúde: crianças e jovens (CIF‐CJ).

Resultados: Trinta e três estudos foram identificados, dos quais um, incluindo 17 crianças com PC espástica, produziu evidência de nível II e os outros, principalmente estudos de coorte não controlados, níveis IV e V. Os resultados no nível de função corporal foram os mais frequentemente relatados. Os resultados sugerem que o BIT contínuo pode ser efetivo para reduzir espasticidade e distonia em PC, assim como outras condição neurológicas, e pode melhorar a facilidade do cuidado e qualidade de vida de crianças com PC, mas o nível de evidência é baixo.

Interpretação: Apesar de três décadas de aplicação de BIT em crianças, e do número relativamente alto de estudos investigando os efeitos do tratamento, uma relação direta não foi demonstrada ainda devido à baixa qualidade dos estudos primários. Futuros estudos investigando os efeitos do BIT contínuo em todos os níveis da CIF‐CJ são necessários. Embora estudos grandes e controlados sejam difíceis de realizar, colaborações nacionais e internacionais podem dar esta oportunidade. Ainda, esudos multicêntricos de coorte prospectivos com acompanhamento de longo prazo, utilizando medidas de resultados harmonizadas, podem oferecer perspectivas para expandir nosso conhecimento sobre os efeitos da terapia com BIT contínuo em crianças.

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Figures

Figure 1
Figure 1
Formula for calculating effect sizes (d), where x¯ represents the mean of the outcome before (pre) and after (post) intervention, SD is standard deviation (pooled), and n is the number of patients.
Figure 2
Figure 2
Formula for calculating confidence intervals (CIs) of effect sizes (d), where x¯ represents the mean of the outcome before (pre) and after (post) intervention, SD is standard deviation (pooled), and n is the number of patients.
Figure 3
Figure 3
Effect sizes of intrathecal baclofen (ITB) on different domains of the International Classification of Functioning Disability and Health: Children and Youth (ICF‐CY) mode in patients with cerebral palsy. MAS, Modified Ashworth Scale; UE, upper extremity; LE, lower extremity; BADS, Barry‐Albright Dystonia Scale; BFM, Burke–Fahn–Marsden scale; VAS, visual analogue scale; GGI, Gillette Gait Index; FAQ, Functional Assessment Questionnaire; MUUL, Melbourne Assessment of Unilateral Upper Limb Function; GMFM, Gross Motor Function Measure; PF50, Parent Form 50; CHQ‐PF50, Child Health Questionnaire Parent Form 50; CP‐CHILD, Caregiver Priorities and Child Health Index of Life with Disabilities.
Figure 4
Figure 4
Effect sizes of intrathecal baclofen (ITB) on different domains of the International Classification of Functioning Disability and Health: Children and Youth (ICF‐CY) mode in other neurological conditions. MAS, Modified Ashworth Scale; UE, upper extremity; LE, lower extremity; AI, anoxic injury; ND, near‐drowning; HI, head injury.
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