Non-attendance at outpatient clinic appointments by children with cerebral palsy.

AIM: To determine factors that influence non-attendance at outpatient clinics by children with cerebral palsy (CP). METHOD: This was a retrospective cohort study of 1395 children with CP (59.6% male; born 2005 to 2017) identified from the New South Wales (NSW)/Australian Capital Territory CP Register, who had scheduled appointments at outpatient clinics at two NSW tertiary paediatric hospitals between 2012 and 2019. Associations between sociodemographic, clinical, and process-of-care factors and non-attendance were examined using multivariate logistic regression with generalized estimating equations. RESULTS: A total of 5773 (12%) of 50 121 scheduled outpatient days were not attended. Non-attendance increased over time (average increase 5.6% per year, 95% confidence interval [CI]: 3.7-7.3). Older children aged 5 to 9 years (adjusted odds ratio [aOR] 1.11; 95% CI: 1.02-1.22) and 10 to 14 years (aOR 1.17; 95% CI: 1.03-1.34), socioeconomic disadvantage (aOR 1.29; 95% CI: 1.11-1.50), previous non-attendance (aOR 1.38; 95% CI: 1.23-1.53), and recent rescheduled or cancelled appointments (aOR 1.08; 95% CI: 1.01-1.16) were associated with increased likelihood of non-attendance. INTERPRETATION: One in eight outpatient appointments for children with CP were not attended. Non-attendance was associated with increasing age, socioeconomic disadvantage, previous non-attendance, and recent rescheduled or cancelled appointments. Identifying specific barriers and interventions to improve access to outpatient services for these groups is needed. WHAT THIS PAPER ADDS: Twelve per cent of scheduled appointments for children with cerebral palsy are not attended. Proportions of appointments not attended has increased over the last decade. Increasing age and socioeconomic disadvantage increase the likelihood of non-attendance. Previous non-attendance and recent cancelled or rescheduled appointments increase the likelihood of further non-attendance.

A pilot feasibility study of gabapentin for managing pain in children with dystonic cerebral palsy.

BACKGROUND: Gabapentin is often used to manage pain in children with dystonic cerebral palsy, however the evidence for its effectiveness in this population is limited. The primary objective of this feasibility pilot study was to assess the factors which might impact on a future randomised controlled trial including the ability to recruit and retain participants, assess adherence/compliance to the prescribed intervention, and ability to complete all outcome assessments. The secondary objective was to gather preliminary evidence for the effectiveness of gabapentin at reducing pain, improving comfort and reducing dystonia in children with dystonic cerebral palsy. METHODS: This open label pilot study recruited children aged 5-18 years with dystonic cerebral palsy and accompanying pain affecting daily activities from four centres around Australia. Children were prescribed gabapentin for 12 weeks and were assessed at baseline, 6 weeks and 12 weeks. The primary outcome was feasibility of the protocol. Secondary outcomes were pain behaviour, pain intensity, care and comfort, individualised goal setting and dystonia severity. RESULTS: Thirteen children (mean age 10.4 years (SD 2.4yrs), 9 females) were recruited from 71 screened over 15 months. Two children withdrew while eight children experienced side effects. There were issues with adherence to medication dosage regimens and data collection. Improvements were seen in pain behaviour, comfort and pain related goals at 12 weeks. Dystonia was not significantly changed. CONCLUSIONS: Whilst gabapentin has potential to improve pain and comfort in children with dystonic CP, the feasibility of implementing a definitive randomised controlled trial is low. Alternative trials designs are required to further examine the effectiveness of gabapentin in this heterogeneous population. TRIAL REGISTRATION: The trial was registered with the Australian Clinical Trial Registry ( ACTRN12616000366459 ) on 22/03/2016 and the Therapeutic Goods Administration (CT-2016-CTN-00500-1) on 22/06/2016.

National surveillance of oral medication prescription for children with dystonic cerebral palsy.

AIM: Oral medications are often first-line medical management for children with cerebral palsy who have generalised dystonia; however, evidence for their effectiveness is limited and dosing practices are inconsistent. As a first step to improve consistency, this study aimed to examine current clinical practice of expert doctors for prescribing medications for children with dystonic cerebral palsy including prescribing patterns and combinations of medications used. METHODS: This was a prospective surveillance study of medical doctors working in major Australian centres who manage children with cerebral palsy. Each week over a continuous 6-month period, doctors completed a custom developed online survey for children seen that week with dystonic cerebral palsy for whom they prescribed a new medication to treat dystonia. RESULTS: Twenty-five doctors consented to participate, 16 of whom prescribed new medications for dystonia in children with cerebral palsy over the study period. There were 77 children who were prescribed new medications. Baclofen and gabapentin were prescribed most, followed by levodopa, trihexyphenidyl and diazepam. The most common combinations of medications were baclofen and diazepam or baclofen and gabapentin. Dosage regimens were variable, particularly for trihexyphenidyl and diazepam. CONCLUSION: Inconsistencies in dosing regimens remain for oral medication prescription by Australian doctors when managing dystonia in cerebral palsy. Future studies using the consensus of expert clinicians will be conducted to develop guidelines in an area where the evidence for individual medications is extremely limited.

Understanding the Five-Year Outcomes of Abusive Head Trauma in Children: A Retrospective Cohort Study.

Understanding the long-term medical and developmental outcomes for children who survive abusive head trauma (AHT) is important to ensure necessary supports and services are available. This study examined the retrospective global and specific medical and developmental outcomes of 55 children with AHT who were treated at The Children's Hospital at Westmead. Global outcomes were assessed using the Kings Outcome Scale of Childhood Head Injury (KOSCHI). Five years post-injury, one child had died and two had made a complete recovery. Forty-five children (81.8%) had a moderate or severe disability, an increase from 64.5% at acute discharge. At follow-up, the main impairments were behavioral problems (53%), vision impairment (44%), fine motor difficulties (26%), gross motor problems (26%), communication problems (24%) and 16% had seizures. A Spearman's Rank correlation revealed that only 41% of variance in KOSCHI scores five years post-injury could be accounted for KOSCHI scores at the time of acute discharge (rs(55) = 0.638, p < .001), and many children's presentation was worse at follow-up. Therefore, all children presenting with AHT need long term follow up regardless of early indications of good recovery.

Adverse events associated with paediatric intrathecal baclofen in Australia: 5 years of data collection.

AIM: This study aims to examine the adverse event (AE) rate for intrathecal baclofen (ITB) therapy in an Australian paediatric population and to clarify type and frequency of AEs. METHODS: AE data were extracted from the Australian Paediatric ITB Research Group national database, to include the first 5 years of data collection. Raw data were collated and analysed descriptively. RESULTS: Data were collected for 40 patients. Forty-seven AEs in 23 patients were reported. Ten (25%) patients required surgical intervention related to their AE. Five patients (12.5%) required pump removal. The most frequent ITB-related AEs were catheter dysfunction (24%), drug overdose, withdrawal or sensitivity (19%), seromas and haematomas (15%) and infections (13%). CONCLUSIONS: The AE rate for ITB therapy is high and needs to be considered when counselling patients regarding ITB as a therapeutic option.

Psychiatric comorbidity is common in dystonia and other movement disorders.

OBJECTIVE: To determine rates of psychiatric comorbidity in a clinical sample of childhood movement disorders (MDs). DESIGN: Cohort study. SETTING: Tertiary children's hospital MD clinics in Sydney, Australia and London, UK. PATIENTS: Cases were children with tic MDs (n=158) and non-tic MDs (n=102), including 66 children with dystonia. Comparison was made with emergency department controls (n=100), neurology controls with peripheral neuropathy or epilepsy (n=37), and community controls (n=10 438). INTERVENTIONS: On-line development and well-being assessment which was additionally clinically rated by experienced child psychiatrists. MAIN OUTCOME MEASURES: Diagnostic schedule and manual of mental disorders-5 criteria for psychiatric diagnoses. RESULTS: Psychiatric comorbidity in the non-tic MD cohort (39.2%) was comparable to the tic cohort (41.8%) (not significant). Psychiatric comorbidity in the non-tic MD cohort was greater than the emergency control group (18%, p<0.0001) and the community cohort (9.5%, p<0.00001), but not the neurology controls (29.7%, p=0.31). Almost half of the patients within the tic cohort with psychiatric comorbidity were receiving medical psychiatric treatment (45.5%) or psychology interventions (43.9%), compared with only 22.5% and 15.0%, respectively, of the non-tic MD cohort with psychiatric comorbidity. CONCLUSIONS: Psychiatric comorbidity is common in non-tic MDs such as dystonia. These psychiatric comorbidities appear to be under-recognised and undertreated.

Short term outcomes of children with abusive head trauma two years post injury: A retrospective study.

PURPOSE: Abusive head trauma (AHT) can have debilitating sequelae for children who survive. A retrospective medical record review was used to describe short-term developmental outcomes of children with AHT and identify predictors of poorer outcomes. METHOD: Children with AHT who received follow up by the hospital's rehabilitation department for 12 to 24 months post-injury were included in this review. Data for 85 children were collected on hearing, vision, gross motor, fine motor, speech and language, cognition, play, adaptive functioning, behaviour and personal-social skills. RESULTS: Global assessment found 42% of children had a good recovery, 34% had a moderate disability and 24% had a severe disability. For whom there was data, more than half had abnormal cognition, behaviour and personal-social skills, whilst more than a third had abnormal speech and language, neurological signs on last assessment, vision, play skills, and gross and fine motor skills. Factors that predicted poorer prognosis across all developmental domains included paediatric intensive care unit admission, longer length of hospital stay, breathing difficulty and lower Glasgow Coma Scale on presentation. CONCLUSION: This study highlights the substantial number of children who have abnormal development in the short-term post-AHT and assists in identifying those who require extensive long-term follow up.

Transition to adult care for young people with cerebral palsy.

Cerebral palsy (CP) is associated with a high burden of comorbid respiratory disease subsequent to multiple risk factors associated with increasing levels of disability. Correspondingly, respiratory disease is the leading cause of death in CP, including amongst young people who are transitioning or who have just transitioned between paediatric and adult healthcare services. Therefore, consideration of both preventive and therapeutic respiratory management is integral to transition in patients with CP, as summarised in this review.

Orthopaedic Surgery in Dystonic Cerebral Palsy.

BACKGROUND: Outcomes after orthopaedic interventions in patients with dystonic cerebral palsy (DCP) are historically regarded as unpredictable. This study aims to evaluate the overall outcome of orthopaedic surgery in children with DCP. METHOD: Children with DCP who underwent lower limb orthopaedic surgery with a minimum follow-up of 12 months were included. Data collected included age at time of surgery, surgical procedures performed, Gross Motor Function Classification System (GMFCS) level, and Barry Albright Dystonia Scale (BADS) score. The cohort was divided into 2 groups. Group 1 (GMFCS levels I to III), mean age 12 years 7 months and group 2 (GMFCS levels IV to V), mean age 10 years 7 months. Group 1 had surgery aimed at deformity correction to improve gait and mobility, and group 2 for the management or prevention of hip displacement. Outcome measures analyzed were: the incidence of unpredictable results related to surgery and early recurrence of deformity in both groups. Functional mobility scale scores were evaluated for group 1 and hip migration percentage for group 2. Linear mixed models were used to take into account repeated measures over time and correlations between measurements from the same patient. RESULTS: Group 1 (n=18); had low BADS scores and were considered to have mild dystonia. Three children experienced unpredictable results, 2 had early recurrence of deformity, 3 had a decline, and 1 child improved in the functional mobility scale.Group 2 (n=19); had high BADS scores and were considered to have moderate to severe dystonia. Nine surgical events involved bony procedures and 15 were soft tissue surgery only. One surgical event lead to unpredictable results and 2 children had early recurrence of deformity. Postoperatively, a linear trend of increasing migration percentage [0.49% (95% confidence interval, 0.23-0.74; P=0.0002)] was seen up to 21 months. There was no significant change after 21 months [-0.08% (95% confidence interval, -0.24 to +0.041; P=0.18)]. CONCLUSIONS: This study suggests that unpredictable results and early recurrence of deformity following orthopaedic surgery in children with DCP are not as common as previously regarded. Furthermore, functional mobility and hip morphology can be improved. LEVEL OF EVIDENCE: Level IV-this is a case-series.

Global mapping for the epidemiology of paediatric spinal cord damage: towards a living data repository.

STUDY DESIGN: Literature review. OBJECTIVES: Globally map key paediatric spinal cord damage epidemiological measures and provide a framework for an ongoing repository of data. SETTING: Worldwide, initiative of ISCoS Prevention Committee. METHODS: Literature search of Medline (1946-March 2017) and Embase (1974-March 2017). Relevant articles in any language regarding children with spinal cord damage included. Stratification of data quality into Green/Yellow /Red 'zones' facilitated comparison between countries. RESULTS: A total of 862 abstracts were reviewed and data from 25 articles were included from 14 countries in 6 of the 21 Global Regions. Fourteen studies involved paediatric traumatic spinal cord injury (SCI) and seven were regarding non-traumatic spinal cord dysfunction (SCDys). An additional four articles provided both paediatric SCI and SCDys data. The median SCI incidence rates in Global Regions were: Asia, East 5.4/million population/year; Australasia 9.9/million population/year; Western Europe 3.3/million population/year and North America, High Income 13.2 million population/year. The median SCDys incidence rates in Global Regions were: Australasia 6.5/million population/year; Western Europe 6.2/million population/year and North America, High Income 2.1/million population/year. SCI was mostly due to land transport (46-74%), falls (12-35%) and sport/recreation (10-25%) and SCDys was mostly caused by tumours (30-63%) and inflammatory/autoimmune causes (28-35%). CONCLUSIONS: There is a scarcity of quality epidemiology studies of paediatric SCD regarding incidence, prevalence, aetiology and survival. Recent ISCoS frameworks provide guidance for researchers to use established classification of SCDys and age group cut-off levels in future studies, thereby improving the ability to compare and combine data.

Neurocognitive outcomes in children following immersion: a long-term study.

OBJECTIVE: To investigate long-term neurocognitive outcomes after a near-drowning incident in children who were deemed neurologically intact on discharge from hospital. DESIGN: A prospective cohort study of near-drowning children. SETTING: 95 drowning and near-drowning admissions, 0-16 years of age, from January 2009 to December 2013, to The Children's Hospital at Westmead, Sydney, NSW, Australia. PARTICIPANTS: 23 children both met the criteria and had parental consent for the study. MAIN OUTCOME MEASURES: Identification of the long-term deficits in behaviour, executive function, motor skills, communicative skills and well-being over a 5-year period. Assessment was undertaken at 3-6 months, 1 year, 3 years and 5 years after near-drowning at clinic visits. Physical developmental screening and executive function screening were done using Behavior Rating Inventory of Executive Function-Preschool version (BRIEF-P) and BRIEF. RESULT: 95 drowning and near-drowning episodes occurred during the study period. 10 (11%) children died, 28 were admitted to the paediatric intensive care unit and 64 directly to a ward. 3 children died in emergency department, 7 children had severe neurological deficit on discharge from the hospital. 23 were subsequently recruited into the study; 5 (22%) of these children had abnormalities in behaviour and/or executive function at some during their follow-up. CONCLUSION: Children admitted to hospital following a near-drowning event warrant long-term follow-up to identify any subtle sequelae which might be amenable to intervention to ensure optimal patient outcome.

Evidence- and Consensus-Based Guidelines for the Management of Communication and Swallowing Disorders Following Pediatric Traumatic Brain Injury.

OBJECTIVE: Evidence-based management guidelines for communication and swallowing disorders following pediatric traumatic brain injury (TBI) are scarcely available, potentially resulting in suboptimal outcomes. To improve clinical care of this population, a multidisciplinary guideline development committee was formed to develop evidence-based recommendation (EBR) and consensus-based recommendation (CBR) for the management of speech, language, and swallowing disorders during the first year of recovery. METHODS: A 3-round Delphi survey was completed by the committee to reach agreement (80% consensus) for the CBRs. Systematic review evidence guided development of EBRs, devised using the National Health and Medical Research Council statement form. RESULTS: Altogether, 30 recommendations (5 EBRs and 25 CBRs) were developed to guide management of speech, language, and swallowing disorders, including prediction of these disorders; health team required, optimal timing of assessment; assessment tools; intervention strategies and commencement of treatment; and key information to support parents. CONCLUSION: The developed recommendations provide a basis for the systematic management of communication and swallowing disorders to be refined as new evidence emerges. Key recommendations include screening of children with moderate/severe TBI for these disorders acutely using specified measures. Patients with severe TBI and prolonged ventilation are a particular at-risk group and should be considered for early referral to speech-language pathology to support timely diagnosis and management. No evidence was identified to support an EBR for treatment, highlighting a key area for research.

A series of n-of-1 trials of stimulants in brain injured children.

BACKGROUND: There is controversy about whether central nervous system stimulant (CNS) medication is an effective method of treating acquired attention deficits in children with acquired brain injury (ABI). OBJECTIVE: The primary objective was to determine the effectiveness of stimulants on attention, concentration and executive function in children with ABI. METHODS: Randomised, double-blind, placebo-controlled, multi-centre n-of-1 trials of stimulants assessing effect on attention, concentration and executive function in 53 children and adolescents with ABI who were outpatients of three tertiary hospitals in Australia. Trials consisted of 3 two-week cycles, each cycle consisting of stimulant medication at doses titrated by physician (1 week) and placebo (1 week) in random order. The effect on parent and teacher Conners' 3 and Behaviour Rating Inventory of Executive Function (BRIEF) was analysed using hierarchical Bayesian methods. RESULTS: Overall, Teacher Conners' Hyperactivity/Impulsivity and Teacher BRIEF Global Executive scales showed important improvement (T-score mean change 2.6; 95% credible interval (CI): 0.4, 4.9; posterior probability of mean change >0 : 0.99; T-score mean change 3.1; 95% CI: -0.1, 6.4; posterior probability of mean change >0 : 0.97). There were no important improvements in parent/guardian-reported primary outcomes. There was heterogeneity in response identified through individual results of the N-of-1 trials. CONCLUSIONS: N-of-1 trials have a clear role in identifying those children/adolescents with ABI and secondary Attention Deficit Hyperactivity Disorder (ADHD) who have important improvements, or worsening on stimulants. The results can only be generalized to children/adolescents who have an apparent pre-trial clinical effect from stimulants.

Aggregated n-of-1 trials of central nervous system stimulants versus placebo for paediatric traumatic brain injury--a pilot study.

BACKGROUND: In 2006 there were 432,700 people in Australia who had acquired brain injury (ABI) with some limitation of activities; 90% of these were traumatic brain injuries (TBIs) and nearly a third sustained injury below age 15 years. One to four years post injury, 20% to 46% of children with traumatic brain injury (TBI) have clinically significant disorders of attention. There is controversy as to whether central nervous system (CNS) stimulants can be an effective method of treating these.Objectives were to determine the efficacy of CNS stimulants for children with TBI, and to calculate the sample size for a larger trial using the Conners' 3 Parent Rating Scales Score as the primary endpoint. METHODS: Pilot series of aggregated prospective randomised, double-blind, n-of-1 trials of stimulant versus placebo within individual patients. SETTING: tertiary children's public hospital. PARTICIPANTS: ten children aged 6 to 16 years more than 12 months post TBI with attention, concentration and behavioral difficulties on stimulants. INTERVENTIONS: Three cycles of methylphenidate or dexamphetamine orally at doses titrated by physician compared to placebo. MAIN OUTCOME MEASURES: Conners 3 Parent (Conners 3-P) and Teacher (Conners 3-T) Rating Scales (Global Index), Behaviour Rating Inventory of Executive Function (BRIEF) and Eyberg Child Behaviour Inventory (ECBI). RESULTS: Five of ten patients completed the study. Data from 18 completed cycles from seven patients were analysed. The posterior mean difference between stimulant and placebo scores for the Conners 3-PS (Global Index) was 2.3 (SD 6.2; 95% credible region -1.0 to 6.1; posterior probability that this mean difference was greater than zero was 0.92), and for the Conners 3-T (Global Index) the posterior mean difference was 5.9 (SD 4.5; 95% credible region -3.1 to 14.9; posterior probability 0.93). Posterior mean differences suggest improvement in behaviour and executive function and a decrease in number and intensity of child behaviour problems when taking stimulants compared to placebo. Taken together these data are suggestive of a small benefit at group level. CONCLUSIONS: In this pilot study, there was sufficient evidence that stimulants may be useful in management of behavioral and cognitive sequelae following TBI, to warrant a larger trial. TRIAL REGISTRATION: he trial was registered with the Australian and New Zealand Clinical Trials Registry: registration number ACTRN12609000873224.

Central nervous system stimulants for secondary attention deficit-hyperactivity disorder after paediatric traumatic brain injury: a rationale and protocol for single patient (n-of-1) multiple cross-over trials.

BACKGROUND: It is estimated that 22,800 children were living with an Acquired Brain Injury (ABI) (0.6% of children aged under 15 years) in Australia during 2003. Many children after a traumatic brain injury will experience difficulties with attention and concentration; a condition termed secondary Attention Deficit-Hyperactivity Disorder. There is conflicting evidence on whether treatment with stimulant therapy with medications such as methylphenidate or dexamphetamine will improve the attention and behavior of children with this condition. METHODS/DESIGN: Single patient trials (n-of-1s or SPTs) evaluate the effect of titrated doses of psychostimulants methylphenidate or dexamphetamine compared to placebo on attention and behavior, in children with TBI and secondary ADHD. The aggregation of multiple SPTs will produce a population estimate of the benefit. Forty-two children will be registered into the trial through rehabilitation services at three large children's hospitals in Australia. Patients will complete up to 3 cycles of treatment. Each cycle is 2 weeks long comprising seven days each of treatment and placebo, with the first two days of each cycle considered a washout period and the data not analysed. The order of treatment and placebo is randomly allocated for each cycle. The Conners' Parent Rating Scales long forms will be employed to measure change in attention-deficit/hyperactivity and related problems of the child, and the primary outcome measure is the Conners' Global Index Parent Version. Secondary outcomes include the teacher and child (if aged > 12 years) Conners' Rating Scales, the Behaviour Rating Inventory of Executive Function among other measures. This study will provide high-level evidence using a novel methodological approach to inform clinicians about the most appropriate treatment for individual children. Through aggregation of individual trials, a population estimate of treatment effect will be provided to guide clinical practice in the treatment of children with secondary ADHD after a traumatic brain injury. DISCUSSION: This study employs an innovative methodological approach on the effectiveness of CNS stimulants for secondary ADHD from a brain injury. The findings will both guide clinicians on treatment recommendations, and inform the concept and acceptance of SPTs in paediatric research. TRIAL REGISTRATION: Australian New Zealand Clinical Trials Registry. ACTRN12609000873224.

Evaluation of bone mineral density and morphology using pQCT in children after spinal cord injury.

OBJECTIVE: To evaluate the effects of spinal cord injury (SCI) on bone density and morphology in children using peripheral quantitative computer tomography (pQCT). DESIGN: Retrospective cohort study of 19 paediatric patients with SCI (9 paraplegics and 10 tetraplegics). RESULTS: There was significant reduction in tibial metaphysial volumetric bone mineral density (vBMD), diaphysial cortical cross-sectional area (CSA), cortical thickness and polar strength-strain index. There was a significant loss of calf muscle CSA. Those who were able to stand had greater trabecular vBMD, tibial cortical thickness and tibial muscle CSA Z-scores. Lower limb fractures did not occur if tibial trabecular vBMD was greater than 100 mg/cm³. Tibial geometry following SCI was more circular compared to controls. CONCLUSIONS: pQCT provides a valuable insight into the regional changes in bone and muscle development in children following SCI. Residual muscle function with the ability to weight bear provides a significant benefit to bone development.

Use of the King's Outcome Scale for Childhood Head Injury in the evaluation of outcome in childhood traumatic brain injury.

OBJECTIVE: To examine the inter-rater reliability of The King's Outcome Scale for Childhood Head Injury (KOSCHI) with clinicians of varying experience in paediatric traumatic brain injury (TBI); and to examine change in outcome during long-term follow-up of children following traumatic brain injury (TBI) using KOSCHI. METHOD: Retrospective assessment of detailed clinic reports of 97 children followed-up by a tertiary specialist paediatric brain injury service. Investigators were blinded to each other's scores. RESULTS: Inter-rater reliability was substantial (weighted kappa 0.71) and similar for investigators of varying experience. KOSCHI outcome was strongly associated with markers of injury severity (p = 0.028). In longitudinal follow-up, KOSCHI score worsened in 7 (23%) children who were injured under 8 years but in no older children (p = 0.02). CONCLUSION: KOSCHI has high inter-rater reliability for investigators of different experience. Long-term KOSCHI outcome is associated with injury severity. Some young children may develop worse disability over time.

Use of intrathecal baclofen therapy in ambulant children and adolescents with spasticity and dystonia of cerebral origin: a systematic review.

AIM: Studies on the use of intrathecal baclofen (ITB) for ambulant adults with spasticity and/or dystonia of cerebral origin are scarce, and are even more limited for children and adolescents. This systematic review investigates the use of ITB to improve walking, transfer ability, and gross motor activities in ambulant children and adolescents with spasticity and/or dystonia of cerebral origin. METHOD: Electronic databases (MEDLINE, CINAHL, PsycINFO, EMBASE, full Cochrane Library, and PEDro) were searched from the earliest date available until March 2011 using combined subject headings and free text if supported by the databases. Studies were included if they had examined individuals who: (1) received ITB therapy by any method (bolus injection, an external delivery system, or an implanted pump); (2) had spasticity and/or dystonia of cerebral origin; (3) were able to ambulate with or without a walking device, i.e. individuals with cerebral palsy (CP) who were in levels I to III of the Gross Motor Function Classification System or individuals with similar functional mobility if they did not have CP; and (4) were aged 18 years or under. Publications in English in peer-reviewed journals reporting any type of research design, except reviews and expert opinions, were included. Studies were excluded if participants had spasticity and/or dystonia of spinal origin and if baclofen was administered only orally. Studies that compared ITB with other interventions such as botulinum toxin were also excluded. RESULTS: Two independent reviewers scored 16 studies against the guidelines for developing systematic reviews from the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM). INTERPRETATION: Fifteen studies were of levels IV or V evidence and only one of level II according to the evidence levels of the AACPDM guidelines, but all were of low quality. No study was found on the use of ITB in ambulant children or adolescents with dystonia of cerebral origin. Not all studies used objective outcome measures to assess the ambulation, transfer ability, and gross motor activities of the participants. A proportion of participants showed improvement in all these areas but adverse events were common. A proportion of participants compromised their ambulatory and transfer abilities after ITB. There was no evidence to support the clinical use of ITB in ambulant individuals with hypertonicity without further rigorous longitudinal studies.