Pam Thomason

Single-event multilevel surgery in children with spastic diplegia: a pilot randomized controlled trial.

BACKGROUND Single-event multilevel surgery is considered the standard of care to improve gait and functioning of children with spastic diplegic cerebral palsy. However, the evidence base is limited. This pilot study is the first randomized controlled trial of single-event multilevel surgery, to our knowledge. METHODS Nineteen children (twelve boys and seven girls with a mean age of nine years and eight months) with spastic diplegia were enrolled. Eleven children were randomized to the surgical group and eight, to the control group. The control group underwent a program of progressive resistance strength training. The randomized phase of the trial concluded at twelve months. The control group then exited the study and progressed to surgery, whereas the surgical group continued to be followed in a prospective cohort study. The primary outcome measures were the Gait Profile Score (GPS) and the Gillette Gait Index (GGI). Secondary outcome measures were gross motor function (Gross Motor Function Measure-66 [GMFM-66]), functional mobility (Functional Mobility Scale [FMS]), time spent in the upright position, and health-related quality of life (Child Health Questionnaire [CHQ]). RESULTS A total of eighty-five surgical procedures were performed, with a mean of eight procedures per child (standard deviation, four). The surgical group had a 34% improvement in the GPS and a 57% improvement in the GGI at twelve months. The control group had a small nonsignificant deterioration in both indices. The between-group differences for the change in the GPS (-5.5; 95% confidence interval, -7.6 to -3.4) and the GGI (-218; 95% confidence interval, -299 to -136) were highly significant. The differences between the groups with regard to the secondary outcome measures were not significant at twelve months. At twenty-four months after surgery, there was a 4.9% increase in the GMFM-66 score and improvements in the FMS score, time spent in the upright position, and the physical functioning domain of the CHQ in the surgical group. CONCLUSIONS This study provides Level-II evidence that single-event multilevel surgery improves the gait of children with spastic diplegic cerebral palsy twelve months after surgery. Improvements in other domains, including gross motor function and quality of life, were not observed until twenty-four months after surgery.

Does botulinum toxin a combined with bracing prevent hip displacement in children with cerebral palsy and "hips at risk"? A randomized, controlled trial.

BACKGROUND Cerebral palsy is the most common cause of childhood physical disability in developed countries, affecting two children per 1000 live births. Hip displacement affects about one-third of children with cerebral palsy and may result in pain, deformity, and impaired function. The prevention of hip displacement has not been studied in a randomized trial as far as we know. METHODS A randomized, controlled trial was conducted to examine the effect of intramuscular injections of botulinum toxin A combined with use of a variable hip abduction brace on the progression of hip displacement in children with cerebral palsy. The patients in the treatment group received injections of botulinum toxin A to the adductor and hamstring muscles every six months for three years and were prescribed a hip abduction brace to be worn for six hours per day. In the control group, no hip bracing was used nor were injections performed. The primary outcome measure was hip displacement from the acetabulum as determined by serial measurements of the migration percentage. RESULTS Ninety children with bilateral cerebral palsy and so-called hips at risk (a migration percentage of >10% but <40%) were entered into the study. Fifty-nine patients were boys, and the mean age was three years. Progressive hip displacement, as determined by serial measurements of the migration percentage, was found in both the treatment and control groups. The rate of hip displacement was reduced in the treatment group by 1.4% per year (95% confidence interval, -0.6% to 3.4%; p = 0.16) when weighted for the uncertainty in rates due to the differing numbers of migration percentage measurements per subject. CONCLUSIONS There may be a small treatment benefit for the combined intervention of intramuscular injection of botulinum toxin A and abduction hip bracing in the management of spastic hip displacement in children with cerebral palsy. However, progressive hip displacement continued to occur in the treatment group, and our data do not support recommending this treatment.

Functional outcome of botulinum toxin A injections to the lower limbs in cerebral palsy

We evaluated gross motor function following botulinum toxin A (BTX‐A) injections in the lower limbs of children with spastic cerebral palsy in a randomized clinical trial, using a cross‐over design. Forty‐nine children (24 males, 25 females, age range 22 to 80 months) were randomly allocated to two groups: group 1 received BTX‐A and physiotherapy, and group 2 received physiotherapy alone for 6 months. At the end of this period, group 2 received BTX‐A and physiotherapy and group 1 continued with physiotherapy alone. Assessment measures were the Gross Motor Function Measure (GMFM), the Vulpe Assessment Battery (VAB), joint range of movement, the Modified Ashworth Scale, and a parental questionnaire. Sustained gains in gross motor function were found in both groups of children but the only additional benefit found in group 1 was a significant increase in fine motor rating on the VAB. By contrast, parents rated the benefit of treatment highly. It is likely that assessment at 3 and 6 months post injection was too late to demonstrate peak gross motor function response and that changes in GMFM are not sustained over 6 months with a single dose. Further studies should investigate changes over shorter time periods and consider covariables such as BTX‐A dosage, number of injection sites, and the role of repeated injections combined with other interventions such as casting.

A randomized controlled trial of the impact of therapeutic horse riding on the quality of life, health, and function of children with cerebral palsy

This randomized controlled trial examined whether therapeutic horse riding has a clinically significant impact on the physical function, health and quality of life (QoL) of children with cerebral palsy (CP). Ninety‐nine children aged 4 to 12 years with no prior horse riding experience and various levels of impairment (Gross Motor Function Classification System Levels I−III) were randomized to intervention (10wks therapeutic programme; 26 males, 24 females; mean age 7y 8mo [SD 2y 5mo] or control (usual activities, 27 males, 22 females; mean age 8y 2mo [SD 2y 6mo]). Pre‐ and post‐measures were completed by 72 families (35 intervention and 37 control). Children’s gross motor function (Gross Motor Function Measure [GMFM]), health status (Child Health Questionnaire [CHQ]), and QoL (CP QoL‐Child, KIDSCREEN) were assessed by parents and QoL was assessed by children before and after the 10‐week study period. On analysis of covariance, there was no statistically significant difference in GMFM, CP QoL‐Child (parent report and child self‐report), and CHQ scores (except family cohesion) between the intervention and control group after the 10‐week study period, but there was weak evidence of a difference for KIDSCREEN (parent report). This study suggests that therapeutic horse riding does not have a clinically significant impact on children with CP. However, a smaller effect cannot be ruled out and the absence of evidence might be explained by a lack of sensitivity of the instruments since the QoL and health measures have not yet been demonstrated to be sensitive to change for children with CP.

The minimal clinically important difference for the Gait Profile Score

The minimally clinically important difference (MCID) is an important concept for interpreting the results of clinical research. This paper proposes a rationale for defining an MCID for the Gait Profile Score (GPS) based on an analysis of the difference in median GPS for children classified at different levels of the Functional Assessment Questionnaire. A strong linear correlation between median score and FAQ level was found. An MCID of 1.6° is therefore suggested, reflecting the mean difference between adjacent FAQ levels. Comparison of this value with (i) the standard deviation of GPS from typically developing children (1.4°) and (ii) the percentage of the difference between the median GPS for each FAQ level and that for typically developing children offers further support to suggest that 1.6° is an appropriate figure.

Single Event Multilevel Surgery in children with bilateral spastic cerebral palsy: A 5 year prospective cohort study

BACKGROUND Single Event Multilevel Surgery (SEMLS) is considered the standard of care to improve gait and function in children with bilateral spastic cerebral palsy (BSCP). We have demonstrated in a randomized controlled trial (RCT) of SEMLS, that gait was improved at 12 months after surgery and gross motor function at 24 months after surgery. The question addressed in this study, was to determine if improvements in gait and function, would be maintained at 5 year follow-up. METHODS Nineteen children with BSCP, GMFCS levels II (14 children) and III (5 children), mean age 9.7 years (range 7.7-12.2 years) participated in a prospective cohort study following participation in a RCT, with follow-up to 5 years. Outcome measures were Gait Profile Score (GPS), Gillette Gait Index (GGI), Gait Deviation Index (GDI), Gross Motor Function Measure (GMFM66) and Functional Mobility Scale (FMS). RESULTS Eighteen children have completed follow-up, with interval analysis at 1, 2 and 5 years post SEMLS. One child was excluded because of neurological deterioration and his diagnosis was revised to Hereditary Spastic Paraparesis (HSP). GPS improved by 5.29° and GMFM66 by 3.3% at 5 years post SEMLS. Differences between outcome measures at 1 versus 5 years and 2 versus 5 years (except GMFM66) were not significant, indicating that improvements in gait and gross motor function were stable over time. CONCLUSIONS SEMLS results in clinically and statistically significant improvements in gait and function, in children with BSCP, which were maintained at 5 years after surgery.

A classification system for hip disease in cerebral palsy

In population‐based studies, hip displacement affects approximately one‐third of children with cerebral palsy (CP). Given the extreme range of clinical phenotypes in the CP spectrum, it is unsurprising that hip development varies from normality, to dislocation and degenerative arthritis. Numerous radiological indices are available to measure hip displacement in children with CP; however, there is no grading system for assessing hip status in broad categorical terms. This makes it difficult to audit the incidence of hip displacement, determine the relationship between hip displacement and CP subtypes, assess the outcome of intervention studies, and to communicate hip status between health care professionals. We developed a categorical, radiographic classification of hip morphology based on qualitative indices and measurement of the key continuous variable, the migration percentage of Reimers. One hundred and thirty‐four radiographs were reviewed of 52 female and 82 male adolescents with CP who were at, or close to, skeletal maturity (mean age 16y 1mo [SD 1y 4mo] range 14y to 19y 1mo). Twenty‐nine were classified at Gross Motor Function Classification System level I, 25 at level II, 27 at level III, 24 at level IV, and 29 at level V. A classification system was developed to encapsulate the full spectrum of hip morphology in CP, with and without intervention.

Progressive resistance training and mobility‐related function in young people with cerebral palsy: a randomized controlled trial

The aim of this study was to investigate whether individualized resistance training improves the physical mobility of young people with cerebral palsy (CP).

Stability of the Gross Motor Function Classification System after single‐event multilevel surgery in children with cerebral palsy

Aim  There are conflicting reports about the stability of the Gross Motor Function Classification System (GMFCS) in children with cerebral palsy (CP) after orthopaedic surgery. We studied the stability of the GMFCS in children with bilateral spastic CP after single‐event multilevel surgery, using the Gait Profile Score (GPS) as the primary outcome measure.

Multilevel Surgery for Equinus Gait in Children with Spastic Diplegic Cerebral Palsy: Medium-term Follow-up with Gait Analysis

BACKGROUND In children with spastic diplegia, surgery for ankle equinus contracture is associated with a high prevalence of both overcorrection, which may result in a calcaneal deformity and crouch gait, and recurrent equinus contracture, which may require revision surgery. We sought to determine if conservative surgery for equinus gait, in the context of multilevel surgery, could result in the avoidance of overcorrection and crouch gait as well as an acceptable rate of recurrent equinus contracture at the time of medium-term follow-up. METHODS This was a retrospective, consecutive cohort study of children with spastic diplegia who had had surgery for equinus gait between 1996 and 2006. All children had distal gastrocnemius recession or differential gastrocnemius-soleus complex lengthening, on one or both sides, as part of single-event multilevel surgery. The primary outcome measures were the Gait Variable Scores (GVS) and Gait Profile Score (GPS) at two time points after surgery. RESULTS Forty children with spastic diplegia, Gross Motor Function Classification System (GMFCS) level II or III, were included in this study. There were twenty-five boys and fifteen girls. The mean age was ten years at the time of surgery and seventeen years at the time of final follow-up. The mean postoperative follow-up period was 7.5 years. The mean ankle GVS improved from 18.5° before surgery to 8.7° at the time of short-term follow-up (p < 0.005) and 7.8° at the time of medium-term follow-up. The equinus gait was successfully corrected in the majority of children, with a low rate of overcorrection (2.5%) and a high rate of recurrent equinus (35%), as determined by sagittal ankle kinematics. Mild recurrent equinus was usually well tolerated and conferred some advantages, including contributing to strong coupling at the knee and independence from using an ankle-foot orthosis. CONCLUSIONS Surgical treatment for equinus gait in children with spastic diplegia was successful, at a mean of seven years, in the majority of cases when combined with multilevel surgery, orthoses, and rehabilitation. No patient developed crouch gait, and the rate of revision surgery for recurrent equinus was 12.5%.