Clinical Phenotype of Cerebral Palsy Depends on the Cause: Is It Really Cerebral Palsy? A Retrospective Study.
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ABSTRACT:
SUBMITTER: Metz C
PROVIDER: S-EPMC8804944 | biostudies-literature |
REPOSITORIES: biostudies-literature
Similar Datasets
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Project description:BackgroundCerebral palsy is the most common cause of childhood physical disability, with multiple associated comorbidities. Administrative claims data provide population-level prevalence estimates for cerebral palsy surveillance; however, their diagnostic accuracy has never been validated in Quebec. This study aimed to assess the accuracy of administrative claims data for the diagnosis of cerebral palsy.MethodsWe conducted a retrospective cohort study of children with cerebral palsy born between 1999 and 2002 within 6 health administrative regions of Quebec. Provincial cerebral palsy registry data (reference standard) and administrative physician claims were linked. We explored differences between true-positive and false-negative cases using subgroup sensitivity analysis.ResultsA total of 301 children were identified with confirmed cerebral palsy from the provincial registry, for an estimated prevalence of 1.8 (95% confidence interval [CI] 1.6-2.1) per 1000 children 5 years of age. The sensitivity and specificity of administrative claims data for cerebral palsy were 65.5% (95% CI 59.8%-70.8%) and 99.9% (95% CI 99.9%-99.9%), respectively, yielding a prevalence of 2.0 (95% CI 1.9-2.3) per 1000 children 5 years of age. The positive and negative predictive values were 58.8% (95% CI 53.3%-64.1%) and 99.9% (95% CI 99.9%-99.9%), respectively. The κ value was 0.62 (95% CI 0.57-0.67). Administrative claims data were more sensitive for children from rural regions, born preterm, with spastic quadriparesis and with higher levels of motor impairment.InterpretationAdministrative claims data do not capture the full spectrum of children with cerebral palsy. This suggests the need for a more sensitive case definition and caution when using such data without validation.
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Project description:Cerebral palsy (CP) is a disorder characterized by abnormal tone, posture and movement and clinically classified based on the predominant motor syndrome-spastic hemiplegia, spastic diplegia, spastic quadriplegia, and extrapyramidal or dyskinetic. The incidence of CP is 2-3 per 1,000 live births. Prematurity and low birthweight are important risk factors for CP; however, multiple other factors have been associated with an increased risk for CP, including maternal infections, and multiple gestation. In most cases of CP the initial injury to the brain occurs during early fetal brain development; intracerebral hemorrhage and periventricular leukomalacia are the main pathologic findings found in preterm infants who develop CP. The diagnosis of CP is primarily based on clinical findings. Early diagnosis is possible based on a combination of clinical history, use of standardized neuromotor assessment and findings on magnetic resonance imaging (MRI); however, in most clinical settings CP is more reliably recognized by 2 years of age. MRI scan is indicated to delineate the extent of brain lesions and to identify congenital brain malformations. Genetic tests and tests for inborn errors of metabolism are indicated based on clinical findings to identify specific disorders. Because CP is associated with multiple associated and secondary medical conditions, its management requires a multidisciplinary team approach. Most children with CP grow up to be productive adults.
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Project description:Psoas muscle spasticity is hypothesised as a rare cause of low back pain in patients with infantile cerebral palsy. The authors describe a new manoeuvre for the study of psoas tenderness and ultrasound (US)-guided transabdominal botulinum toxin injection technique. A possible causal relationship between psoas tension and low back pain was found incidentally in two examined cases. In subsequent patients, botulinum toxin was injected and, in cases of disappearance of symptoms, the psoas tendon was sectioned at the pelvic brim with definitive disappearance of pain. The relationship between psoas tension and low back pain in patients with infantile cerebral palsy seems likely, given the result in the four patients.
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Project description:AimThis study aimed to investigate whether children with cerebral palsy (CP) had equal access to timely physiotherapy. Additionally, to learn more about clinical characteristics of infants with CP, we explored differences in neonatal clinical history and CP profile between children referred by a neonatologist or enrolled in neonatal follow-up and those referred by other healthcare professionals as well as those referred before and after 5 months corrected age.MethodsWe conducted a retrospective chart review study including children born in Uppsala County, Sweden, from 2010 to 2016, who had received a CP diagnosis by July 2019. Entries by doctors and physiotherapists working at Uppsala University Children's Hospital were reviewed.ResultsThirty-eight children were included (21 girls, 55.3%) in the study. Twenty-two (57.9%) were born at term. Twenty-five children (66%) had their first visit to a physiotherapist before 5 months corrected age, and this included all children (n = 22, 57.9%) referred by a neonatologist or enrolled in neonatal follow-up. The latter group had significantly earlier access to physiotherapy compared to children referred by other healthcare professionals, with a median of 1.9 (min-max: -1-4) and 7.6 (min-max: 1-24) months, respectively (p < 0.0001). Referral source explained unique variance in predicting time of referral to physiotherapist (R2 0.550, B 4.213, p < 0.0001) when controlling for both number of risk factors and severity of motor impairment. However, number of risk factor was vital for early access to physiotherapy for children referred by other health care professionals. Children referred by a neonatologist or enrolled in neonatal follow-up or referred before 5 months corrected age differed on all measured variables concerning neonatal clinical history and CP profile, compared to children referred by other healthcare professionals or after 5 months corrected age. The latter groups had milder forms of CP. In total, twenty-eight children (73.7%) were ambulatory at 2 years of age. Bilateral spastic CP was most common among those referred by a neonatologist or enrolled in neonatal follow-up or referred before 5 months corrected age, while unilateral spastic CP was most common among those referred by other healthcare professionals or after 5 months corrected age.ConclusionInfants with CP have unequal access to timely physiotherapy, and children considered at low risk for CP receive therapy later. Neonatal follow-up of infants considered at high risk for CP that involves an assessment of motor performance using an evidence-based method during the first months of life corrected age seems to be effective in identifying CP early. Conversely, measuring milestone attainment seems to be a less reliable method for early identification. To provide safe and equal care, all professionals performing developmental surveillance should receive proper training and use evidence-based assessment methods. Physiotherapy should be available prior to formal medical diagnosis.
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Project description:ObjectiveTo apply practice-based evidence to clinical management of cerebral palsy (CP). The process of establishing purpose, structure, logistics, and elements of a multi-institutional registry and the baseline characteristics of initial enrollees are reported.DesignA consensus-building process among consumers, clinicians, and researchers used a participatory action process.SettingCommunity, hospitals, and universities.ParticipantsMore than 100 clinicians, researchers, and consumers and more than 1858 enrollees in the registry.Main outcome measuresNot applicable.ResultsConsensus was that the purpose of registry was to (1) quantify practice variation, (2) facilitate quality improvement (QI), and (3) perform comparative effectiveness research (CER). Collecting data during routine clinical care using the electronic medical record was determined to be a sustainable plan for data acquisition and management. Clinicians from multiple disciplines defined salient characteristics of individuals and interventions for the registry elements. The registry was central to the clinical research network, and a leadership structure was created. A leading electronic health record platform adopted the registry elements. Twenty-four sites have initiated the data collection process and agreed to export data to the registry. Currently 12 are collecting data. Number of enrollees and characteristics were similar to other population registers.ConclusionsThis is the first multi-institutional CP registry that contains the patient and treatment characteristics needed for QI and CER. The Cerebral Palsy Research Network registry elements are implemented in a versatile electronic platform and minimize burden to clinicians. The resultant registry is available for any institution to participate and is growing rapidly.
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