Association Between Two Methods of Spinal and Pelvic Analysis Among Children With Cerebral Palsy.

OBJECTIVES: Children with cerebral palsy have weak muscles, which may impair postural adjustments. These postural adjustments are required for gait and dynamic balance during the daily living activities. The aim of this study was to investigate the association between Cobb's angle and Formetric 4D surface topography system in evaluating spinal and pelvic deformity in children with cerebral palsy. METHODS: One hundred children with spastic diplegia (6 to 8 years old) diagnosed as cerebral palsy participated in this study and selected from the Outpatient Clinic of Faculty of Physical Therapy. Digital x- ray and formetric analysis were used to measure spinal deformities and pelvic deviation in children with cerebral palsy. RESULTS: There were positive correlations between Cobb's angle and formetric parameters, including trunk imbalance, lateral deviation, and pelvic tilt. Also, Formetric parameters were significant predictors of Cobb's angle, including trunk imbalance (for a one-degree increase, Cobb's angle increases by 0.227, lateral deviation (for a one-degree increase, Cobb's angle increases by 0.665), and pelvic tilt (for a one-degree increase, Cobb's angle increases by 0.252). CONCLUSION: Formetric 4D surface topography system was effective in evaluating spinal and pelvic deformity in children with cerebral palsy when compared with Digital x-ray.

Risk Factors and Outcomes for Cerebral Palsy With Hypoxic-Ischemic Brain Injury Patterns Without Documented Neonatal Encephalopathy.

BACKGROUND AND OBJECTIVES: Perinatal hypoxic-ischemic brain injury is a leading cause of term-born cerebral palsy, the most common lifelong physical disability. Diagnosis is commonly made in the neonatal period by the combination of neonatal encephalopathy (NE) and typical neuroimaging findings. However, children without a history of neonatal encephalopathy may present later in childhood with motor disability and neuroimaging findings consistent with perinatal hypoxic-ischemic injury. We sought to determine the prevalence of such presentations using the retrospective viewpoint of a large multiregional cerebral palsy registry. METHODS: Patient cases were extracted from the Canadian Cerebral Palsy Registry with gestational age >36 weeks, an MRI pattern consistent with hypoxic-ischemic injury (HII, acute total, partial prolonged, or combined), and an absence of postnatal cause for HII. Documentation of NE was noted. Maternal-fetal risk factors, labor and delivery, neonatal course, and clinical outcome were extracted. Comparisons were performed using χ2 tests and multivariable logistic regression with multiple imputation. Propensity scores were used to assess for bias. RESULTS: Of the 170 children with MRI findings typical for HII, 140 (82.4%, 95% confidence interval [CI] 75.7%-87.7%) had documented NE and 29 (17.0%, 95% CI 11.7%-23.6%) did not. The group without NE had more abnormalities of amniotic fluid volume (odds ratio [OR] 15.8, 95% CI 1.2-835), had fetal growth restriction (OR 4.7, 95% CI 1.0-19.9), had less resuscitation (OR 0.03, 95% CI 0.007-0.08), had higher 5-minute Apgar scores (OR 2.2, 95% CI 1.6-3.0), were less likely to have neonatal seizures (OR 0.004, 95% CI 0.00009-0.03), and did not receive therapeutic hypothermia. MRI was performed at a median 1.1 months (interquartile range [IQR] 0.67-12.8 months) for those with NE and 12.2 months (IQR 6.6-25.9) for those without (p = 0.011). Patterns of injury on MRI were seen in similar proportions. Hemiplegia was more common in those without documented NE (OR 5.1, 95% CI 1.5-16.1); rates of preserved ambulatory function were similar. DISCUSSION: Approximately one-sixth of term-born children with an eventual diagnosis of cerebral palsy and MRI findings consistent with perinatal hypoxic-ischemic brain injury do not have documented neonatal encephalopathy, which was associated with abnormalities of fetal growth and amniotic fluid volume, and a less complex neonatal course. Long-term outcomes seem comparable with their peers with encephalopathy. The absence of documented neonatal encephalopathy does not exclude perinatal hypoxic-ischemic injury, which may have occurred antenatally and must be carefully evaluated with MRI.

Predictive Value of Fidgety Movement Assessment and Magnetic Resonance Imaging for Cerebral Palsy in Infants.

BACKGROUND: The early prediction of cerebral palsy (CP) could enable the follow-up of high-risk infants during the neuroplasticity period. This study aimed to explore the predictive value of fidgety movement assessment (FMA) and brain magnetic resonance imaging (MRI) for the development of CP in clinic rehabilitation setting. METHODS: This retrospective observational study included infants who underwent FMA and brain MRI at age nine to 20 weeks at Children's Hospital, Zhejiang University School of Medicine, between March 2018 and September 2019. The area under the receiver operating characteristic curve (AUC), sensitivity, specificity, and accuracy of FMA and MRI for predicting the development of CP were assessed. RESULTS: A total of 258 infants (169 males, gestational age 37.4 ± 3.0 weeks, birth weight 2987.9 ± 757.1 g) were included. Fifteen children had CP after age two years. The diagnostic value of FMA and brain MRI combination showed 86.7% sensitivity (95% confidence interval [CI]: 58.4% to 97.7%), 98.4% specificity (95% CI: 95.6% to 99.5%), and 97.7% accuracy (95% CI: 95.0% to 99.1%); the combination diagnostic value also showed a significantly higher AUC for predicting CP after age two years than FMA alone (AUC: 0.981 vs 0.893, P = 0.013). CONCLUSIONS: The diagnostic value of FMA and brain MRI combination during infancy showed a high predictive value for CP development in clinical rehabilitation setting.

Therapeutic effects of extracorporeal shock wave therapy on patients with spastic cerebral palsy and Rett syndrome: clinical and ultrasonographic findings.

BACKGROUND: Extracorporeal shock wave therapy (ESWT) is reportedly effective for improving spasticity and motor function in children with cerebral palsy (CP). Because late-stage Rett syndrome has a similar presentation, this study aimed to investigate the effects of ESWT on these two diseases. MATERIAL AND METHODS: Patients diagnosed with spastic CP and Rett syndrome received 1500 impulses of ESWT at 4 Hz and 0.1 mJ/mm2, on their spastic legsonce weekly for a total of 12 weeks. Outcomes were assessed before and 4 and 12 weeks after ESWT. Clinical assessments included the Modified Ashworth Scale (MAS), passive range of motion (PROM), and Gross Motor Function Measure 88 (GMFM-88). Ultrasonographic assessments included muscle thickness, acoustic radiation force impulse (ARFI), and strain elastography. RESULTS: Fifteen patients with CP and six with Rett syndrome were enrolled in this study. After ESWT, patients with CP showed significant clinical improvement in the MAS (P = 0.011), ankle PROM (P = 0.002), walking/running/jumping function (P = 0.003), and total function (P < 0.001) of the GMFM-88. The patients with Rett syndrome showed improved MAS scores (P = 0.061) and significantly improved total gross motor function (P = 0.030). Under ARFI, patients with CP demonstrated decreased shear wave speed in the gastrocnemius medial head (P = 0.038). Conversely, patients with Rett syndrome show increased shear-wave speeds after ESWT. CONCLUSION: Our study provides evidence that a weekly course of low-dose ESWT for 12 weeks is beneficial for children with both CP and Rett syndrome, with the clinical effects of reducing spasticity and improving the gross motor function of the lower limbs. The ARFI sonoelastography reveals improvement of muscle stiffness in patients with CP after ESWT, but deteriorated in patients with Rett syndrome. The diverse therapeutic response to ESWT may be caused by the MECP2 mutation in Rett syndrome, having a continuous impact and driving the pathophysiology differently as compared to CP, which is secondary to a static insult. Trial registration IRB 201700462A3. Registered 22March 2017, https://cghhrpms.cgmh.org.tw/HRPMS/Default.aspx .

The relation between neuroimaging and visual impairment in children and adolescents with cerebral palsy: A systematic review.

OBJECTIVE: The structure-function relation between magnetic resonance imaging (MRI) and visual impairment (VI) in children with cerebral palsy (CP) has not been fully unravelled. The present systematic review aims to summarize the relation between brain lesions on MRI and VI in children and adolescents with CP. METHODS: PubMed, Embase, Web of Science Core Collection, and Cochrane Database were systematically searched according to the PRISMA checklist. A total of 45 articles met the inclusion criteria. RESULTS: White matter lesions were most frequently associated with VI. Only 25 studies described lesions within specific structures, mainly in the optic radiations. Only four studies reported on the thalamus. 8.4% of children with CP showed no brain abnormalities on MRI. Diffusion-weighted MRI studies showed that decreased structural connectivity in the optic radiations, superior longitudinal fasciculus, posterior limb of the internal capsule, and occipital lobe is associated with more severe VI. CONCLUSIONS: All types of brain lesions lead to visual dysfunctions, arguing for a comprehensive visual assessment in all children with CP. Whereas white matter damage is a well-known contributor, the exact contribution of specific visual structures requires further investigation, to enable early prediction, detection, and intervention.

Functional connectivity of sensorimotor network is enhanced in spastic diplegic cerebral palsy: A multimodal study using fMRI and MEG.

OBJECTIVE: To assess the effects to functional connectivity (FC) caused by lesions related to spastic diplegic cerebral palsy (CP) in children and adolescents using multiple imaging modalities. METHODS: We used resting state magnetoencephalography (MEG) envelope signals in alpha, beta and gamma ranges and resting state functional magnetic resonance imaging (fMRI) signals to quantify FC between selected sensorimotor regions of interest (ROIs) in 11 adolescents with spastic diplegic cerebral palsy and 24 typically developing controls. Motor performance of the hands was quantified with gross motor, fine motor and kinesthesia tests. RESULTS: In fMRI, participants with CP showed enhanced FC within posterior parietal regions; in MEG, they showed enhanced interhemispheric FC between sensorimotor regions and posterior parietal regions both in alpha and lower beta bands. There was a correlation between the kinesthesia score and fronto-parietal connectivity in the control population. CONCLUSIONS: CP is associated with enhanced FC in sensorimotor network. This difference is not correlated with hand coordination performance. The effect of the lesion is likely not fully captured by temporal correlation of ROI signals. SIGNIFICANCE: Brain lesions can show as increased temporal correlation of activity between remote brain areas. We suggest this effect is likely separate from typical physiological correlates of functional connectivity.

Prefrontal cortex hemodynamic activity during a test of lower extremity functional muscle strength in children with cerebral palsy: A functional near-infrared spectroscopy study.

Children with cerebral palsy (CP) exhibit impaired motor control and significant muscle weakness due to a brain lesion. However, studies that assess the relationship between brain activity and performance on dynamic functional muscle strength assessments in CP are needed. The aim of this study was to determine the effect of a progressive lateral step-up test on prefrontal cortex (PFC) hemodynamic activity in children with CP. Fourteen ambulatory children with spastic CP (Gross Motor Function Classification System level I; 5-11 y) and 14 age- and sex-matched typically developing control children completed a progressive lateral step-up test at incremental step heights (0, 10, 15 and 20 cm) using their non-dominant lower limb. Hemodynamic activity in the PFC was assessed using non-invasive, portable functional neuroimaging (functional near-infrared spectroscopy). Children with CP completed fewer repetitions at each step height and exhibited lower PFC hemodynamic activity across step heights compared to controls. Lower PFC activation in CP was maintained after statistically controlling for the number of repetitions completed at each step height. PFC hemodynamic activity was not associated with LSUT task performance in children with CP, but a positive relationship was observed in controls at the most challenging 20 cm step height. The results suggest there is an altered PFC recruitment pattern in children with CP during a highly dynamic test of functional strength. Further studies are needed to explore the mechanisms underlying the suppressed PFC activation observed in children with CP compared to typically developing children.

Distinct manifestations and potential mechanisms of seizures due to cortical versus white matter injury in children.

PURPOSE: To study seizure manifestations and outcomes in children with cortical versus white matter injury, differences potentially explaining variability of epilepsy in children with cerebral palsy. METHODS: In this population-based retrospective cohort study, MRIs of children with cerebral palsy due to ischemia or haemorrhage were classified according to presence or absence of cortical injury. MRI findings were then correlated with history of neonatal seizures, seizures during childhood, epilepsy syndromes, and seizure outcomes. RESULTS: Of 256 children studied, neonatal seizures occurred in 57 and seizures during childhood occurred in 93. Children with neonatal seizures were more likely to develop seizures during childhood, mostly those with cortical injury. Cortical injury was more strongly associated with (1) developing seizures during childhood, (2) more severe epilepsy syndromes (infantile spasms syndrome, focal epilepsy, Lennox-Gastaut syndrome), and (3) less likelihood of reaching > 2 years without seizures at last follow-up, compared to children without cortical injury. Children without cortical injury, mainly those with white matter injury, were less likely to develop neonatal seizures and seizures during childhood, and when they did, epilepsy syndromes were more commonly febrile seizures and self-limited focal epilepsies of childhood, with most achieving > 2 years without seizures at last follow-up. The presence of cortical injury also influenced seizure occurrence, severity, and outcome within the different predominant injury patterns of the MRI Classification System in cerebral palsy, most notably white matter injury. CONCLUSIONS: Epileptogenesis is understood with cortical injury but not well with white matter injury, the latter potentially related to altered postnatal white matter development or myelination leading to apoptosis, abnormal synaptogenesis or altered thalamic connectivity of cortical neurons. These findings, and the potential mechanisms discussed, likely explain the variability of epilepsy in children with cerebral palsy and epilepsy following early-life brain injury in general.

Impaired glymphatic system revealed by DTI-ALPS in cerebral palsy due to periventricular leukomalacia: relation with brain lesion burden and hand dysfunction.

PURPOSE: Preterm children with cerebral palsy (CP) often have varying hand dysfunction, while the specific brain injury with periventricular leukomalacia (PVL) cannot quite explain its mechanism. We aimed to investigate glymphatic activity using diffusion tensor image analysis along the perivascular space (DTI-ALPS) method and evaluate its association with brain lesion burden and hand dysfunction in children with CP secondary to PVL. METHODS: We retrospectively enrolled 18 children with bilateral spastic CP due to PVL and 29 age- and sex-matched typically developing controls. The Manual Ability Classification System (MACS) was used to assess severity of hand dysfunction in CP. A mediation model was performed to explore the relationship among the DTI-ALPS index, brain lesion burden, and the MACS level in children with CP. RESULTS: There were significant differences in the DTI-ALPS index between children with CP and their typically developing peers. The DTI-ALPS index of the children with CP was lower than that of the controls (1.448 vs. 1.625, P = 0.003). The mediation analysis showed that the DTI-ALPS index fully mediated the relationship between brain lesion burden and the MACS level (c' = 0.061, P = 0.665), explaining 80% of the effect. CONCLUSION: This study provides new insights into the neural basis of hand dysfunction in children with CP, demonstrating an important role of glymphatic impairment in such patients. These results suggest that PVL might affect hand function in children with CP by disrupting glymphatic drainage.

From Hip Screening to Hip Surveillance: Transforming Care for Patients With Cerebral Palsy: An Analysis of a Single Institution.

INTRODUCTION: Surveillance programs aimed at monitoring hip displacement in patients with cerebral palsy have been demonstrated to decrease the incidence of hip dislocations and properly time surgical intervention, ultimately improving patient outcomes. The objective of this study was to determine whether the implementation of a hip screening to surveillance program at a tertiary academic teaching hospital in 2017 increased the frequency of radiographic evaluations and changed the timing of surgical intervention. METHODS: A total of 592 patients with cerebral palsy were identified, and 468 of these patients had initial radiograph date data available. In this analysis, 246 patients with initial radiograph dates after 2012 were included. The study population was divided into two groups based on the initial radiograph date, 2012 to 2016 versus 2017 to 2022. One hundred sixty patients (65%) were in the 2012 to 2016 group, and 86 (35%) were in the 2017 to 2022 group. Statistical analysis was conducted using various techniques, such as two-sample Student t-test, Mann-Whitney U test, chi square/Fisher exact test, and multivariable linear regression analysis. RESULTS: The average number of radiographs per year in the 2017 to 2022 group was 0.11 (95% CI: 0.02, 0.20, P = 0.017) higher than the 2012 to 2016 group. After adjusting for confounders using multivariable linear regression analysis, this difference was even larger (difference 0.16, 95% CI: 0.06, 0.25, P = 0.001). The surgical intervention rate was significantly lower in the 2017 to 2022 group compared with the 2012 to 2016 group (12.9% versus 40.6%, P < 0.001). DISCUSSION: The results of this study suggest that the implementation of a hip screening to surveillance program results in more frequent radiographic evaluations and possibly a reduced need for surgical intervention from 2017 to 2022. In the 2012 to 2016 group, more surgical interventions were performed likely because of the lack of any hip surveillance or screening program in place.

Characterization of Atypical Corticospinal Tract Microstructure and Hand Impairments in Early-Onset Hemiplegic Cerebral Palsy: Preliminary Findings.

Unilateral brain injuries occurring before at or shortly after full-term can result in hemiplegic cerebral palsy (HCP). HCP affects one side of the body and can be characterized in the hand with measures of weakness and a loss of independent hand control resulting in mirror movements. Hand impairment severity is extremely heterogeneous across individuals with HCP and the neural basis for this variability is unclear. We used diffusion MRI and tractography to investigate the relationship between structural morphology of the supraspinal corticospinal tract (CST) and the severity of two typical hand impairments experienced by individuals with HCP, grasp weakness and mirror movements. Results from nine children with HCP and eight children with typical development show that there is a significant hemispheric association between CST microstructure and hand impairment severity that may be explained by atypical development and fiber distribution of motor pathways. Further analysis in the non-lesioned (dominant) hemisphere shows significant differences for CST termination in the cortex between participants with HCP and those with typical development. These findings suggest that structural disparities at the cellular level in the seemingly unaffected hemisphere after early unilateral brain injury may be the cause of heterogeneous hand impairments seen in this population.Clinical Relevance- Quantitative measurement of the variability in hand function in individuals with HCP is necessary to represent the distinct impairments experienced by each person. Further understanding of the structural neural morphology underlying distal upper extremity motor deficits after early unilateral brain injury will help lead to the development of more specific targeted interventions that increase functional outcomes.

Prevalence, Clinical Features, Neuroimaging, and Genetic Findings in Children With Ataxic Cerebral Palsy in Europe.

BACKGROUND AND OBJECTIVES: To report on prevalence, associated impairments, severity, and neuroimaging findings in children with ataxic cerebral palsy (CP). METHODS: In children coded as having ataxic CP in the Central database of Joint Research Center-Surveillance of Cerebral Palsy in Europe (JRC-SCPE) and born during 1980-2010, birth characteristics, severity profiles including associated impairments, neuroimaging patterns, and the presence of syndromes were analyzed. Definitions were according to validated SCPE guidelines. Prevalence over time was estimated using Poisson regression. RESULTS: In total, 679 children with ataxic CP were identified in 20 European CP registers. The proportion with ataxic CP was 3.8% and varied from 0% to 12.9%. Prevalence over time showed no significant trend. Approximately 70% of children with ataxic CP were able to walk, and 40% had severe intellectual impairment and a high impairment index. Children with ataxic CP were mostly born at term (79%) and with normal birth weight (77%). Neuroimaging patterns revealed normal findings in 29%, brain maldevelopments in 28.5%, miscellaneous findings in 23.5%, and brain injuries in 19%, according to the SCPE classification. Genetic syndromes were described in 9%. DISCUSSION: This register-based multicenter study on children with ataxic CP provides a large sample size for the analysis of prevalence, severity, and origin of this rare CP subtype. Even with strict inclusion and classification criteria, there is variation between registers on how to deal with this subtype, and diagnosis of ataxic CP remains a challenge. Ataxic cerebral palsy differs from other CP subtypes: children with ataxic CP have a disability profile that is more pronounced in terms of cognitive than gross motor dysfunction. They are mostly term born and the origin rarely suggests acquired injuries. In addition to neuroimaging, a comprehensive genetic workup is particularly recommended for children with this CP type.

Reduced brainstem volume is associated with mobility impairments in youth with cerebral palsy.

BACKGROUND: Persons with cerebral palsy (CP) have impaired mobility that has been attributed to changes in structure and function within the nervous system. The brainstem is a region that plays a critical role in mobility by connecting the cortex and cerebellum to the spinal cord, yet this region has been largely unstudied in persons with CP. RESEARCH QUESTION: We used high-resolution structural MRI and biomechanical analyses to examine whether the volume of the whole brainstem and its constituent elements are altered in CP and if these alterations relate to the mobility impairments within this population. METHODS: A cohort study was conducted to assess the volume of the whole brainstem, pons, midbrain, medulla, and superior cerebellar peduncle in a cohort of persons with CP (N = 26; Age = 16.3 ± 1.0 years; GMFCS levels I-IV, Females = 12) and a cohort of neurotypical (NT) controls (N = 38; Age = 14.3 ± 0.4 years, Females = 14) using structural MR imaging of the brainstem. Outside the scanner, a digital mat was used to quantify the spatiotemporal gait biomechanics of these individuals. RESULTS: We found a significant decrease in volume of the total brainstem, midbrain, and pons in persons with CP in comparison to the NT controls. Furthermore, we found that the altered volumes were related to reduced gait velocity and step length. SIGNIFICANCE: The structural changes in the brainstems of persons with CP may contribute to the mobility impairments that are ubiquitous within this population.

Pelvis radiographs in children with cerebral palsy: effects of patient positioning on calculating migration percentages.

BACKGROUND: Hip displacement in children with cerebral palsy (CP) is monitored by measuring migration percentage on anteroposterior pelvis radiographs. However, proper positioning for radiography in children with spasticity is difficult. The reliability and accuracy of migration percentage as a function of patient positioning is unknown. OBJECTIVE: To determine the effects of patient positioning on migration percentage measurements in children with CP. MATERIALS AND METHODS: We identified children with CP (≤18-year-old) with pelvis CT and anteroposterior pelvis radiograph obtained <6 months apart (10/2018-11/2021). Digitally reconstructed radiographs were generated from each pelvis CT, to simulate nine different patient positions: neutral; 10° and 20° lordosis and kyphosis; and 10° and 20° right rotation and left rotation. Two radiologists measured migration percentages from the simulated and real pelvis radiographs. We used Spearman's rho to assess inter-rater reliability, and Wilcoxon signed rank test to determine statistical significance. RESULTS: We studied sixty-three children (male=41; median age=8 years; range=4-18 years). The two radiologists' migration percentage measurements were highly correlated with each other across all simulated and real radiographs (Spearman's rho=0.86-0.99, P<0.01). For both readers and hips, migration percentages measured from real radiographs were significantly different from those measured from neutral simulated radiographs (P<0.01), with median absolute difference=5-6 percentage points (PP) and interquartile range (IQR)=9-12 PP. When comparing migration percentage measurements from neutral simulated radiographs to those in kyphosis/lordosis and right/left rotations, median absolute differences were 2-4 PP (IQR=3-8 PP) and 4-15 PP (IQR=6-17 PP), respectively. CONCLUSION: Inter-rater reliability of measured migration percentages is high, but accuracy decreases with patient positional changes.

Cerebellar growth, volume and diffusivity in children cooled for neonatal encephalopathy without cerebral palsy.

Children cooled for HIE and who did not develop cerebral palsy (CP) still underperform at early school age in motor and cognitive domains and have altered supra-tentorial brain volumes and white matter connectivity. We obtained T1-weighted and diffusion-weighted MRI, motor (MABC-2) and cognitive (WISC-IV) scores from children aged 6-8 years who were cooled for HIE secondary to perinatal asphyxia without CP (cases), and controls matched for age, sex, and socioeconomic status. In 35 case children, we measured cerebellar growth from infancy (age 4-15 days after birth) to childhood. In childhood, cerebellar volumes were measured in 26 cases and 23 controls. Diffusion properties (mean diffusivity, MD and fractional anisotropy, FA) were calculated in 24 cases and 19 controls, in 9 cerebellar regions. Cases with FSIQ ≤ 85 had reduced growth of cerebellar width compared to those with FSIQ > 85 (p = 0.0005). Regional cerebellar volumes were smaller in cases compared to controls (p < 0.05); these differences were not significant when normalised to total brain volume. There were no case-control differences in MD or FA. Interposed nucleus volume was more strongly associated with IQ in cases than in controls (p = 0.0196). Other associations with developmental outcome did not differ between cases and controls.

Comparative Analysis of Physical Examination, CT Scan, and Three-Dimensional Gait Analysis in Evaluating Lower Extremity Torsion Deformities in Children with Cerebral Palsy.

BACKGROUND The aim of this study was to analyze the correlation and the accuracy of lower-extremity torsion deformities measured by physical examination, CT scan, and three-dimensional gait analysis in children with CP. MATERIAL AND METHODS The study group included 72 children with CP with lower-extremity torsion deformities. All subjects were assessed by: 1. physical examination: maximum internal rotation (MIR), maximum external rotation (MER) for hip joint torsion, and transmalleolar axis (TMA) for tibial torsion; 2. CT scanning: femoral anteversion (FAV) and tibial torsion (TT); 3. three-dimensional gait analysis kinematic parameters: single-support phase of femoral rotation, double-support phase of femoral rotation, swing phase of femoral rotation and single-support phase of tibial rotation, double-support phase of tibial rotation, and swing phase of tibial rotation. Statistical analysis was performed using the Pearson correlation test. A significance level of P<0.05 was set. RESULTS In femurs, MIR and MER were correlated with FAV, and the correlation of MER was higher, while physical examination and FAV were not correlated with any kinematic data in gait analysis. In tibias, there was no correlation between TMA and TT, but both TMA and TT were correlated with the gait analysis kinematic data, and the correlation of TT was higher. TMA was more correlated with tibial rotation during swing phase, while TT was more correlated with tibial rotation in single-support phase. CONCLUSIONS Three-dimensional gait analysis can analyze the tibial rotation of children with cerebral palsy, which is highly correlated with CT and physical examination. However, femoral rotation was not associated with CT and physical examination.

Radiological and Genetic Evaluation in Hypotonic Infants.

OBJECTIVE: To investigate the importance and diagnostic yield of genetic and radiological evaluations in children with hypotonia. STUDY DESIGN: Comparative observational study. Place and Duration of the Study: Department of Pediatrics Neurology, Namik Kemal University, Tekirdag, Turkey, between 2019 and 2022. METHODOLOGY: Patients' medical histories, laboratory results, radiological examinations, and genetic tests, if any, were obtained retrospectively from the patients' clinic files. Children with hypotonia detected since the infantile period and who were on regular follow-up were included in the study. Patients who lost the follow-up were excluded. RESULTS: Out of one hundred and seventy patients, 61.8% (n=105) were boys and 38.2% (n=65) were girls. The admission age of the patients ranged from 1 to 121 months; the mean age at presentation was 13.52±17.35 months. Hypotonia was central in 85.3% (n=145), peripheral in 12.4% (n=21), and mixed in 2.3% (n=4). Cerebral palsy was the predominant, non-genetic clinical cause of hypotonia (n=66, 39%). Brain magnetic resonance imaging (MRI) was normal in 48.2% (n=82). The most common MRI abnormality was periventricular leukomalacia in 15.9% (n=27). Sixty-five (38.2%) patients were diagnosed genetically. More than half of the patients with a genetic diagnosis were diagnosed by whole exome sequencing (WES). CONCLUSION: Brain MRI is the first choice for the patients with central hypotonia. Patients who cannot be diagnosed with clinical findings and brain MRI should undergo WES. This is helpful for the long-term prognosis and management. KEY WORDS: Hypotonia, Whole exome sequencing, Magnetic resonance, Spinal muscular atrophy, Cerebral palsy.

The gait pattern and not the femoral morphology is the main contributor to asymmetric hip joint loading.

Gait asymmetry and skeletal deformities are common in many children with cerebral palsy (CP). Changes of the hip joint loading, i.e. hip joint contact force (HJCF), can lead to pathological femoral growth. A child's gait pattern and femoral morphology affect HJCFs. The twofold aim of this study was to (1) evaluate if the asymmetry in HJCFs is higher in children with CP compared to typically developing (TD) children and (2) identify if the bony morphology or the subject-specific gait pattern is the main contributor to asymmetric HJCFs. Magnetic resonance images (MRI) and three-dimensional gait analysis data of twelve children with CP and fifteen TD children were used to create subject-specific musculoskeletal models and calculate HJCF using OpenSim. Root-mean-square-differences between left and right HJCF magnitude and orientation were computed and compared between participant groups (CP versus TD). Additionally, the influence on HJCF asymmetries solely due to the femoral morphology and solely due to the gait pattern was quantified. Our findings demonstrate that the gait pattern is the main contributor to asymmetric HJCFs in CP and TD children. Children with CP have higher HJCF asymmetries which is probably the result of larger asymmetries in their gait pattern compared to TD children. The gained insights from our study highlight that clinical interventions should focus on normalizing the gait pattern and therefore the hip joint loading to avoid the development of femoral deformities.

Impact of lower muscle stiffness on ankle dorsiflexion restriction in children with cerebral palsy evaluated using ultrasound elastography.

BACKGROUND: Plantar flexor muscles always contribute to limiting the range of motion of ankle dorsiflexion in children with spastic cerebral palsy, but the individual contributions of these muscles are not well defined. This study aimed to identify which muscles' stiffness impacts the dorsiflexion range of motion in children with cerebral palsy. METHODS: Twenty-five children with cerebral palsy were included. The maximum passive dorsiflexion range of motion was measured in two positions: hip and knee joints in flexion, and both joints in full extension. Strain ratios indicating muscle stiffness were measured using strain elastography of the lateral and medial gastrocnemius, soleus, flexor hallucis longus, peroneus longus, peroneus brevis, and tibialis posterior muscles. To analyze which muscles impact the limitation of the dorsiflexion range, multiple regression analyses were conducted. The values of muscle stiffness were included as independent valuables, and the values of the dorsiflexion range were included as dependent valuables. A p-value <0.05 was considered statistically significant. FINDINGS: In the analyses, the soleus and flexor hallucis longus muscle stiffness were significant independent factors for the dorsiflexion range of motion of hip and knee flexion (adjusted R2: 0.50). The lateral gastrocnemius muscle stiffness was a significant independent factor for the dorsiflexion range of motion with both joints in full extension (adjusted R2: 0.61). INTERPRETATION: Flexor hallucis longus muscle stiffness, in addition to triceps surae muscle stiffness, was shown to impact dorsiflexion range; attention should be paid to muscle stiffness in children with cerebral palsy.