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1.
Acta Ortop Mex ; 38(3): 197-201, 2024.
Article En | MEDLINE | ID: mdl-38862151

INTRODUCTION: severe, rigid hip abduction deformity in individuals with cerebral palsy (CP) is an exceptionally uncommon condition. This posture hinders the positioning in the wheelchair and the completion of basic activities of daily living (ADL). Addressing such severe deformities can be quite challenging. MATERIAL AND METHODS: a 14-year-old male, with spastic-dystonic quadriplegic CP, developed rigid and severe flexion-abduction contractures in both hips, characterized by 90 degrees of flexion and 100 degrees of abduction. These contractures severely impeded his ability to comfortably use a wheelchair and even pass through doorways. Performing basic ADLs became a significant challenge for both the patient and his caregivers. RESULTS: the treatment approach involved a two-stage surgical procedure, one for each hip, with a two-month interval between them. An extensive release of the fascia latae, gluteus maximus, external rotators, and hip flexors; in combination with a proximal femur osteotomy were performed. To maintain the corrections achieved, long-leg casts connected with two bars were employed, followed by orthotic support and physiotherapy. Following the procedure, lower limb adduction was achieved, and the patient and caregivers were highly satisfied, as ADLs and basic caregiving had been greatly facilitated. CONCLUSIONS: while the available literature on the management of severe rigid abduction hip contractures in non-ambulatory CP patients is limited, and treatment options are often complex, the present case underscores the effectiveness of a comprehensive approach involving soft tissue release and bone surgery. Achieving a more favorable wheelchair positioning and facilitating basic ADLs and care represents a significant success for patients and families.


INTRODUCCIÓN: la deformidad severa y rígida en abducción de cadera en individuos con parálisis cerebral (PC) es una condición infrecuente. Esta postura dificulta el posicionamiento en la silla de ruedas y la realización de actividades básicas de la vida diaria (AVD). El tratamiento de estas deformidades tan severas puede ser todo un reto. MATERIAL Y MÉTODOS: varón de 14 años, con PC tetrapléjica espástica-distónica, que desarrolló contracturas rígidas y severas de flexión-abducción en ambas caderas, caracterizadas por 90 grados de flexión y 100 grados de abducción. Estas contracturas impedían gravemente su capacidad para utilizar cómodamente una silla de ruedas e incluso pasar por las puertas. La realización de actividades básicas de la vida diaria se convirtió en un reto importante tanto para el paciente como para sus cuidadores. RESULTADOS: el tratamiento consistió en una intervención quirúrgica en dos fases, una para cada cadera, con un intervalo de dos meses entre ellas. Se realizó una amplia liberación de la fascia lata, el glúteo mayor, los rotadores externos y los flexores de la cadera; en combinación con una osteotomía proximal del fémur. Para mantener las correcciones conseguidas, se emplearon escayolas de pierna larga conectadas con dos barras, seguidas de soporte ortésico y fisioterapia. Tras la intervención, se consiguió la aducción de los miembros inferiores y el paciente y los cuidadores se mostraron muy satisfechos, ya que se habían facilitado en gran medida las AVD y los cuidados básicos. CONCLUSIONES: aunque la bibliografía disponible sobre el tratamiento de las contracturas rígidas graves de la cadera en abducción en pacientes no deambulantes con PC es limitada, y las opciones de tratamiento suelen ser complejas, el presente caso subraya la eficacia de un enfoque integral que incluye la liberación de los tejidos blandos y la cirugía ósea. Conseguir una posición más favorable en la silla de ruedas y facilitar las AVD básicas y los cuidados representa un éxito significativo para los pacientes y sus familias.


Cerebral Palsy , Hip Contracture , Humans , Cerebral Palsy/complications , Male , Adolescent , Hip Contracture/etiology , Hip Contracture/surgery , Severity of Illness Index , Activities of Daily Living , Contracture/surgery , Contracture/etiology
2.
Neurosurg Focus ; 56(6): E5, 2024 Jun.
Article En | MEDLINE | ID: mdl-38823046

OBJECTIVE: Spasticity is a challenging feature of cerebral palsy (CP) that may be managed with selective dorsal rhizotomy (SDR). Although standard work tools (SWTs) have recently been utilized to inform a standard of care for neurosurgical procedures, no SWTs for SDR have been previously described. The authors present the multidisciplinary approach SWTs for SDR used at their institutions to promote consistency in the field and minimize complication rates. METHODS: A multidisciplinary approach was used to define all steps in the SDR pathway. Preoperative, intraoperative, and postoperative workflows were synthesized, with specific efforts to improve mobility through inpatient rehabilitation and minimize infection. RESULTS: The SWTs have been implemented at two institutions for 7 years. An illustrative case of a patient aged 3 years 10 months with a history of premature birth at 29 weeks, spastic-diplegic CP, right-sided periventricular leukomalacia, and developmental delay who underwent L2-S1 SDR is presented. CONCLUSIONS: The authors detail SWTs for SDR developed by a multidisciplinary team with specific steps at all points in the patient pathway. The illustrative case emphasizes that SWTs may help ensure the safety of SDR while maximizing its long-term efficacy for individuals with CP.


Cerebral Palsy , Rhizotomy , Humans , Cerebral Palsy/surgery , Cerebral Palsy/complications , Rhizotomy/methods , Child, Preschool , Muscle Spasticity/surgery , Male , Female
3.
Neurosurg Focus ; 56(6): E6, 2024 Jun.
Article En | MEDLINE | ID: mdl-38823044

OBJECTIVE: Single-level selective dorsal rhizotomy (SDR), typically indicated for ambulatory patients, is a controversial topic for severe spastic cerebral palsy (CP) with Gross Motor Function Classification System (GMFCS) level IV or V. The objective of this case series and systematic literature review was to outline the indication and outcome of palliative SDR for nonambulatory patients with CP and GMFCS level IV and V, focusing on improvement of spasticity and of patient and caregiver reported quality of life assessment. METHODS: A retrospective case series of patients with CP and GMFCS level IV or V who underwent single-level SDR at the authors' institution is presented. Furthermore, two databases (PubMed and Embase) were searched and a systematic review with a search string based on the terms "selective dorsal rhizotomy," "cerebral palsy," and "outcome" was conducted. The primary outcome was the reduction of spasticity based on the modified Ashworth scale (MAS). Secondary outcomes were change on the Gross Motor Function Measure-66 (GMFM-66), evaluation of patient-reported outcome measures (PROMs), surgical morbidity, and mortality. RESULTS: Eleven consecutive children under the age of 25 years undergoing palliative single-level SDR were included. All patients showed a reduction in MAS score (mean 1.09 ± 0.66 points) and no surgical morbidity and mortality occurred. For the systematic review results from our case series, in addition to 4 reports, 274 total patients were included. Reduction of spasticity based on MAS score was noted in all studies (mean range 1.09-3.2 points). Furthermore, in 2 studies spasticity of the upper extremities showed a MAS score reduction as well (range 1.7-2.8 points). The GMFM-66 score improved in 72% of the patients, while bladder function improved in 78% of the patients. Based on the PROMs, 92% of the patients/caregivers were satisfied with the outcome and their quality of life after the procedure. Two wound infections (2.7%) and one CSF leak (1.3%) occurred, while no surgery-related deaths were described. CONCLUSIONS: This analysis showed an improvement in spasticity, daily care, and comfort for patients with CP and GMFCS levels IV and V. Larger cohorts analyzing the outcome of palliative single-level SDR, based on the MAS, GMFM-66, and PROMs, are still needed and should be the focus of future studies. Systematic review registration no.: CRD42024495762 (https://www.crd.york.ac.uk/prospero/).


Cerebral Palsy , Muscle Spasticity , Palliative Care , Rhizotomy , Humans , Cerebral Palsy/surgery , Cerebral Palsy/complications , Rhizotomy/methods , Child , Male , Female , Child, Preschool , Retrospective Studies , Adolescent , Muscle Spasticity/surgery , Muscle Spasticity/etiology , Palliative Care/methods , Quality of Life , Treatment Outcome
4.
Neurosurg Focus ; 56(6): E11, 2024 Jun.
Article En | MEDLINE | ID: mdl-38823047

OBJECTIVE: Intrathecal baclofen (ITB) pumps are commonly used in pediatric patients with cerebral palsy (CP) and medically refractory spasticity. However, catheter malfunction and associated risk factors are not well understood. The aim of this study was to examine potential risk factors for spinal catheter malfunction and characterize postoperative follow-up to understand the clinical consequences. METHODS: Patients who received ITB pump replacement or revision at Boston Children's Hospital between 2010 and 2023 were retrospectively reviewed. The spinal catheter revision cohort (SCRC) included patients whose spinal catheter was occluded requiring lumbar catheter revision. The second cohort included abdominal pump replacements only (APRC). Between-group comparisons and multivariable regression identified factors associated with catheter revision and postoperative outcomes. RESULTS: Forty-one (33.6%) patients underwent spinal catheter revision and were compared with 81 patients (66.4%) who underwent abdominal pump replacement only. Younger age at surgery and an elevated preoperative lower-extremity modified Ashworth scale grade were associated with spinal catheter revision (p < 0.05). Catheter model type, tip location, and history of spinal fusion were not associated with obstruction. Postoperatively, SCRC patients experienced a higher rate of infection (17.1%) relative to APRC patients (0%) within 30 days from their ITB pump replacement procedure (p < 0.05) and greater likelihood of subsequent ITB system removal compared with the APRC (24.4% vs 7.4%, p < 0.05). Although not differing preoperatively, SCRC patients had lower postoperative ITB doses when compared with the APRC group (median dose 143 vs 350 µg/day, p < 0.05) at hospital discharge and remained statistically different at the 6-month and 1-year follow-ups (p < 0.05). There were no postoperative differences in baclofen overdose, withdrawal, or median number of hospital readmissions within 30 days. Overall, 31.7% of spinal catheter revisions were unanticipated by the clinical team at time of surgery. CONCLUSIONS: Younger age at surgery and increased preoperative lower-extremity tone may be risk factors for catheter obstruction, resulting in a higher rate of postoperative infection and subsequent ITB pump removal compared with pump replacement alone. Spinal catheter occlusion can complicate revision or replacement procedures, especially when unanticipated. Routine clinical assessment may be inadequate for diagnosing insidious catheter malfunction. Catheter occlusion deserves further study, and routine assessment of catheter patency may be warranted to prevent suboptimal tone therapy.


Baclofen , Cerebral Palsy , Infusion Pumps, Implantable , Muscle Relaxants, Central , Humans , Baclofen/administration & dosage , Baclofen/adverse effects , Male , Female , Child , Infusion Pumps, Implantable/adverse effects , Risk Factors , Muscle Relaxants, Central/administration & dosage , Retrospective Studies , Adolescent , Cerebral Palsy/surgery , Cerebral Palsy/complications , Child, Preschool , Muscle Spasticity/drug therapy , Muscle Spasticity/etiology , Muscle Spasticity/surgery , Reoperation/methods , Injections, Spinal/methods , Treatment Outcome , Postoperative Complications/etiology , Equipment Failure , Cohort Studies
6.
Neurosurg Focus ; 56(6): E9, 2024 Jun.
Article En | MEDLINE | ID: mdl-38823052

OBJECTIVE: Children with cerebral palsy (CP) often experience medically refractory hypertonia, for which there are surgical therapies including neuromodulation and rhizotomy. Traditional surgical treatment for medically refractory mixed hypertonia or dystonia includes intrathecal baclofen pumps and selective dorsal rhizotomy. A nonselective lumbosacral ventral-dorsal rhizotomy (VDR; ventral and dorsal roots lesioned by 80%-90%) has the potential to address the limitations of traditional surgical options. The authors highlighted the institutional safety and efficacy of nonselective lumbosacral VDR for palliative tone management in nonambulatory patients with more severe CP. METHODS: The authors performed a retrospective analysis of patients who had undergone lumbosacral VDR between 2022 and 2023. Demographic factors, clinical variables, and operative characteristics were collected. The primary outcomes of interest included tone control and quality of life improvement. Secondary outcome measures included, as a measure of safety, perioperative events such as paresthesias. Postoperative complications were also noted. RESULTS: Fourteen patients (7 female) were included in the study. All patients had undergone a T12-L2 osteoplastic laminoplasty and bilateral L1-S1 VDR. Nine patients had quadriplegic mixed hypertonia, 4 had quadriplegic spasticity, and 1 had generalized secondary dystonia. Following VDR, there was a significant decrease in both lower-extremity modified Ashworth Scale (mAS) scores (mean difference [MD] -2.77 ± 1.0, p < 0.001) and upper-extremity mAS scores (MD -0.71 ± 0.76, p = 0.02), with an average follow-up of 3 months. In the patient with generalized dystonia, the lower-extremity Barry-Albright Dystonia Scale score decreased from 8 to 0, and the overall score decreased from 32 to 13. All parents noted increased ease in caregiving, particularly in terms of positioning, transfers, and changing. The mean daily enteral baclofen dose decreased from 47 mg preoperatively to 24.5 mg postoperatively (p < 0.001). Three patients developed wound dehiscence, 2 of whom had concurrent infections. CONCLUSIONS: Lumbosacral VDR is safe, is effective for tone control, and can provide quality of life improvements in patients with medically refractory lower-limb mixed hypertonia. Lumbosacral VDR can be considered for palliative tone control in nonambulatory patients with more severe CP. Larger studies with longer follow-ups are necessary to further determine safety and long-term benefits in these patients.


Cerebral Palsy , Muscle Hypertonia , Rhizotomy , Humans , Cerebral Palsy/surgery , Cerebral Palsy/complications , Female , Rhizotomy/methods , Male , Child , Retrospective Studies , Muscle Hypertonia/surgery , Muscle Hypertonia/drug therapy , Adolescent , Treatment Outcome , Child, Preschool , Lower Extremity/surgery , Lumbosacral Region/surgery , Quality of Life
7.
Neurorehabil Neural Repair ; 38(7): 479-492, 2024 Jul.
Article En | MEDLINE | ID: mdl-38842031

BACKGROUND: Movement disorders in children and adolescents with dyskinetic cerebral palsy (CP) are commonly assessed from video recordings, however scoring is time-consuming and expert knowledge is required for an appropriate assessment. OBJECTIVE: To explore a machine learning approach for automated classification of amplitude and duration of distal leg dystonia and choreoathetosis within short video sequences. METHODS: Available videos of a heel-toe tapping task were preprocessed to optimize key point extraction using markerless motion analysis. Postprocessed key point data were passed to a time series classification ensemble algorithm to classify dystonia and choreoathetosis duration and amplitude classes (scores 0, 1, 2, 3, and 4), respectively. As ground truth clinical scoring of dystonia and choreoathetosis by the Dyskinesia Impairment Scale was used. Multiclass performance metrics as well as metrics for summarized scores: absence (score 0) and presence (score 1-4) were determined. RESULTS: Thirty-three participants were included: 29 with dyskinetic CP and 4 typically developing, age 14 years:6 months ± 5 years:15 months. The multiclass accuracy results for dystonia were 77% for duration and 68% for amplitude; for choreoathetosis 30% for duration and 38% for amplitude. The metrics for score 0 versus score 1 to 4 revealed an accuracy of 81% for dystonia duration, 77% for dystonia amplitude, 53% for choreoathetosis duration and amplitude. CONCLUSIONS: This methodology study yielded encouraging results in distinguishing between presence and absence of dystonia, but not for choreoathetosis. A larger dataset is required for models to accurately represent distinct classes/scores. This study presents a novel methodology of automated assessment of movement disorders solely from video data.


Athetosis , Cerebral Palsy , Dystonia , Video Recording , Humans , Adolescent , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Cerebral Palsy/classification , Cerebral Palsy/diagnosis , Male , Female , Child , Dystonia/physiopathology , Dystonia/diagnosis , Dystonia/classification , Dystonia/etiology , Athetosis/physiopathology , Athetosis/diagnosis , Athetosis/etiology , Lower Extremity/physiopathology , Machine Learning
8.
Acta Orthop ; 95: 200-205, 2024 May 06.
Article En | MEDLINE | ID: mdl-38708569

BACKGROUND AND PURPOSE: Reduced range of motion (ROM) and spasticity are common secondary findings in cerebral palsy (CP) affecting gait, positioning, and everyday functioning. These impairments can change over time and lead to various needs for intervention. The aim of this study was to analyze the development path of the changes in hamstring length, knee extension, ankle dorsiflexion, and spasticity in hamstrings and gastrosoleus from childhood into adulthood in individuals with CP at the Gross Motor Function Classification System (GMFCS) levels I-V. METHODS: A longitudinal cohort study was undertaken of 61,800 measurements in 3,223 individuals with CP, born 1990-2017 and followed for an average of 8.7 years (range 0-26). The age at examination varied between 0 and 30 years. The GMFCS levels I-V, goniometric measurements, and the modified Ashworth scale (MAS) were used for repeated assessments of motor function, ROM, and spasticity. RESULTS: Throughout the follow-up period, knee extension and hamstring length exhibited a consistent decline across all individuals, with more pronounced decreases evident in those classified at GMFCS levels III-V. Ankle dorsiflexion demonstrated a gradual reduction from 15° to 5° (GMFCS I-IV) or 10° (GMFCS V). Spasticity levels in the hamstrings and gastrosoleus peaked between ages 5 and 7, showing a propensity to increase with higher GMFCS levels. CONCLUSION: Passive ROM continues to decrease to 30 years of age, most pronouncedly for knee extension. Conversely, spasticity reached its peak at a younger age, with a more notable occurrence observed in the gastrosoleus compared with the hamstrings. Less than 50% of individuals had spasticity corresponding to MAS 2-4 at any age.


Ankle Joint , Cerebral Palsy , Knee Joint , Muscle Spasticity , Range of Motion, Articular , Humans , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Muscle Spasticity/physiopathology , Muscle Spasticity/etiology , Longitudinal Studies , Range of Motion, Articular/physiology , Child , Adolescent , Male , Female , Adult , Young Adult , Knee Joint/physiopathology , Child, Preschool , Ankle Joint/physiopathology , Infant , Hamstring Muscles/physiopathology , Cohort Studies
9.
JBJS Case Connect ; 14(2)2024 Apr 01.
Article En | MEDLINE | ID: mdl-38758928

CASE: A 17-year-old adolescent boy with Gross Motor Function Classification System 5 cerebral palsy and neuromuscular scoliosis underwent posterior spinal fusion and segmental spinal instrumentation from T3 to the pelvis. He developed a right ischial pressure injury a few months postoperatively, which persisted despite nonoperative measures. He subsequently underwent an ipsilateral transiliac-shortening osteotomy 16 months after spinal surgery to treat his residual pelvic obliquity and the ischial pressure injury, which healed completely. At the 1-year follow-up visit, there were no further signs of pressure injury. CONCLUSION: This case report describes transiliac-shortening osteotomy as a viable treatment option for non-healing ischial pressure injuries secondary to fixed pelvic obliquity.


Ischium , Osteotomy , Pressure Ulcer , Humans , Male , Adolescent , Osteotomy/methods , Ischium/injuries , Ischium/surgery , Pressure Ulcer/surgery , Pressure Ulcer/etiology , Spinal Fusion/methods , Cerebral Palsy/surgery , Cerebral Palsy/complications , Scoliosis/surgery , Ilium/surgery
10.
Gait Posture ; 112: 1-7, 2024 Jul.
Article En | MEDLINE | ID: mdl-38718437

BACKGROUND: Femoral derotation osteotomy is treatment of choice in intoeing gait secondary to cerebral palsy (CP). RESEARCH QUESTION: The aim of this study was to critically appraise the literature regarding the long-term outcomes of femoral derotation surgery in CP. METHODS: Electronic databases of PubMed and Scopus was used for the literature review by two researchers independently (SB, SC). The study population included patients of cerebral palsy undergoing femoral derotation surgery. The keywords used were "cerebral palsy", "intoeing gait", "femur anteversion", "hip anteversion", "femur derotation" and "femur osteotomy". RESULTS: Nine studies which included 657 limbs in 407 patients were selected for this study of which eight were retrospective in nature. The improvement in hip rotation at stance at last follow up (more than five years) maintained a statistical significance (SMD 1.67 95 %CI 1.12-2.22). Similar statistically significant outcomes were noted for foot progression angle (SMD 1.19 95 %CI 0.92-1.47), anteversion (SMD 2.75 95 %CI 1.49-4.01) and total passive internal rotation (SMD 1.71 95 %CI 1.19-2.22). SIGNIFICANCE: Femoral derotation surgery is the procedure of choice for correction of intoeing gait in CP. Even though, there is deterioration of results on long-term as compared to short term, majority of the patients shall maintain overall correction without recurrence of an intoeing gait. Future studies with uniform criteria for defining recurrence on the basis of functional limitations shall provide better idea about the natural course of this procedure.


Cerebral Palsy , Femur , Gait Disorders, Neurologic , Osteotomy , Cerebral Palsy/complications , Cerebral Palsy/surgery , Cerebral Palsy/physiopathology , Humans , Femur/surgery , Osteotomy/methods , Gait Disorders, Neurologic/surgery , Gait Disorders, Neurologic/etiology , Treatment Outcome
12.
Res Dev Disabil ; 150: 104760, 2024 Jul.
Article En | MEDLINE | ID: mdl-38795555

BACKGROUND: Pain perception mechanisms in cerebral palsy remain largely unclear. AIMS: This study investigates brain activity in adults with cerebral palsy during painful and non-painful stretching to elucidate their pain processing characteristics. METHODS AND PROCEDURES: Twenty adults with cerebral palsy and 20 controls underwent EEG in three conditions: rest, non-painful stretching, and painful stretching. Time-frequency power density of theta, alpha, and beta waves in somatosensory and frontal cortices was analyzed, alongside baseline pressure pain thresholds. OUTCOMES AND RESULTS: Cerebral palsy individuals exhibited higher theta, alpha, and beta power density in both cortices during painful stretching compared to rest, and lower during non-painful stretching. Controls showed higher power density during non-painful stretching but lower during painful stretching. Cerebral palsy individuals had higher pain sensitivity, with those more sensitive experiencing greater alpha power density. CONCLUSIONS AND IMPLICATIONS: These findings confirm alterations in the cerebral processing of pain in individuals with cerebral palsy. This knowledge could enhance future approaches to the diagnosis and treatment of pain in this vulnerable population.


Cerebral Palsy , Electroencephalography , Pain Threshold , Humans , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Male , Female , Adult , Pain Threshold/physiology , Case-Control Studies , Young Adult , Muscle Stretching Exercises , Pain Perception/physiology , Pain/physiopathology , Pain/etiology , Frontal Lobe/physiopathology , Somatosensory Cortex/physiopathology
13.
Pediatr Neurol ; 156: 170-177, 2024 Jul.
Article En | MEDLINE | ID: mdl-38788279

BACKGROUND: Children with cerebral palsy (CP) frequently have associated disorders and complications, including gastrointestinal problems. Helicobacter pylori is a common infection worldwide, frequently associated with gastrointestinal manifestations. METHODS: To estimate the prevalence of H. pylori infection in children with CP, a cross-sectional study over an eight-month period was performed in the pediatric neurology outpatient clinic of Tanta University Hospital. The study included 100 patients with CP aged two to 17 years. All patients were tested for H. pylori antigen in stool by enzyme-linked immunosorbent assay. RESULTS: The mean age of studied children with CP was 7.03 ± 4.1 years; there were 57 males and 43 females. Spastic quadriplegic CP was the most common type of CP (34%). Forty-five children with CP were positive for H. pylori antigen in stool. Intellectual disability (ID), low sociodemographic scoring system, semisolid diet, and Eating and Drinking Ability Classification System (EDACS) levels 4 and 5 were significant predictors of H. pylori infection (odds ratio of 1.86, 2.63, 12, and 1.77, respectively, P < 0.05). Vomiting, abdominal pain, and gastrointestinal tract bleeding were significantly more frequent in H. pylori-infected children with CP than noninfected children with CP (P value < 0.05) CONCLUSION: H. pylori is a relatively common infection among children with CP. The main risk factors for H. pylori infection were low socioeconomic level, ID, semisolid diet, and EDACS levels 4 and 5.


Cerebral Palsy , Helicobacter Infections , Helicobacter pylori , Humans , Male , Female , Cross-Sectional Studies , Child , Helicobacter Infections/complications , Helicobacter Infections/epidemiology , Cerebral Palsy/complications , Cerebral Palsy/epidemiology , Child, Preschool , Helicobacter pylori/isolation & purification , Adolescent , Prevalence
14.
Dev Neurorehabil ; 27(1-2): 34-43, 2024.
Article En | MEDLINE | ID: mdl-38695209

PURPOSE: Many youth and adults with Cerebral Palsy (CP) experience high levels of fatigue. This study aimed to compare three fatigue self-report questionnaires to guide clinicians. METHOD: Thirty youth and adults (age range 17-64) with CP were assessed with Danish versions of the Fatigue Impact and Severity Self-Assessment questionnaire, the Modified Mental Fatigue Scale, and the Multidimensional Fatigue Inventory. Psychometric properties were investigated. Rank order and classification models were compared across questionnaires. RESULTS: The Reduced Motivation and Physical Fatigue subscales of the Multidimensional Fatigue Inventory showed inadequate internal consistency. Participants were frequently ranked differently with the questionnaires. There were issues related to the conceptualization of physical fatigue. CONCLUSION: The choice of assessment tool should be based on assessment purpose as the questionnaires assess different aspects of fatigue severity, impact, and management. Also, test selection can have important implications on the conclusions that are made about fatigue type and severity.


Cerebral Palsy , Fatigue , Psychometrics , Self Report , Humans , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Male , Adult , Female , Fatigue/diagnosis , Adolescent , Denmark , Young Adult , Middle Aged , Surveys and Questionnaires/standards , Severity of Illness Index
15.
BMC Musculoskelet Disord ; 25(1): 391, 2024 May 18.
Article En | MEDLINE | ID: mdl-38762469

BACKGROUND: Pain is common in individuals with cerebral palsy (CP) and the most reported pain site is the foot/lower leg. We analyzed the prevalence of pain in the foot/lower leg and the associations with age, sex, gross motor function, and clinical findings in individuals with CP. METHOD: This was a cross-sectional register-study, based on data reported to the Swedish Cerebral Palsy Follow-up Program (CPUP). All participants in CPUP, four years-of-age or older, were included. Pearson chi-square tests and logistic regression were used to analyze the prevalence and degree of pain in the foot/lower leg. RESULTS: In total, 5,122 individuals were included from the CPUP database: 58% were males and 66% were under 18 years-of-age. Overall, 1,077 (21%) reported pain in the foot/lower leg. The odds ratios (ORs) of pain were higher in females (OR 1.31, 95% confidence interval (CI) 1.13-1.53), individuals who could ambulate (Gross Motor Function Classification System Level I (OR 1.84, CI 1.32-2.57) and II (OR 2.01, CI 1.46-2.79) compared to level V), and in individuals with decreased range of motion of the ankle (dorsiflexion 1-10 degrees (OR 1.43, CI 1.13-1.83) and ≤ 0 degrees (OR 1.46, CI 1.10-1.93) compared to ≥ 20 degrees). With increasing age the OR of pain increased (OR 1.02, CI 1.01-1.03) as well as the reported pain intensity (p < 0.001). CONCLUSIONS: Pain in the foot and lower leg appears to be a significant problem in individuals with CP, particularly in those who walk. As with pain in general in this population, both pain intensity and frequency increase with age. The odds of pain in the foot and lower leg were increased in individuals with limited dorsiflexion of the ankle. Given the cross-sectional design causality cannot be inferred and it is unknown if pain causes decreased range of motion of the ankle or if decreased range of motion causes pain. Further research is needed on causal pathways and importantly on prevention.


Cerebral Palsy , Leg , Registries , Humans , Cerebral Palsy/epidemiology , Cerebral Palsy/complications , Cerebral Palsy/physiopathology , Male , Female , Cross-Sectional Studies , Adult , Adolescent , Child , Young Adult , Sweden/epidemiology , Child, Preschool , Prevalence , Foot/physiopathology , Middle Aged , Pain/epidemiology , Pain/diagnosis , Pain/etiology , Pain Measurement
16.
Neurosurg Rev ; 47(1): 142, 2024 Apr 08.
Article En | MEDLINE | ID: mdl-38587684

Cervical perivascular sympathectomy (CPVS) can improve communication disorders in children with cerebral palsy (CP); however, there are no research reports on the factors affecting surgical efficacy. This study aimed to establish a nomogram for poor prognosis after CPVS. We collected data from 313 CP patients who underwent CPVS at the Neurosurgery Cerebral Palsy Center of the Second Affiliated Hospital of Xinjiang Medical University from January 2019 to January 2023. Among them, 70% (n = 216) formed the training cohort and 30% (n = 97) the validation cohort. The general data and laboratory examination data of both groups were analyzed. In training cohort, 82 (37.96%) showed improved postoperative communication function. Logistic analysis identified motor function, serum alkaline phosphatase, serum albumin, and prothrombin activity as the prognostic factors. Using these four factors, a prediction model was constructed with an area under the curve (AUC) of 0.807 (95% confidence interval [CI], 0.743-0.870), indicating its ability to predict adverse outcomes after CPVS. The validation cohort results showed an AUC of 0.76 (95% CI, 0.650-0.869). The consistency curve and Hosmer-Lemeshow test (χ2 = 10.988 and p = 0.202, respectively) demonstrated good consistency between the model-predicted incidence and the actual incidence of poor prognosis. Motor function, serum alkaline phosphatase, serum albumin, and prothrombin activity are independent risk factors associated with the prognosis of communication disorders after CPVS. The combined prediction model has a good clinical prediction effect and has promising potential to be used for early prediction of prognosis of CPVS.


Cerebral Palsy , Communication Disorders , Child , Humans , Alkaline Phosphatase , Cerebral Palsy/complications , Cerebral Palsy/surgery , Prothrombin , Sympathectomy , Serum Albumin
17.
Gait Posture ; 111: 105-121, 2024 Jun.
Article En | MEDLINE | ID: mdl-38663321

BACKGROUND: Among neurological pathologies, cerebral palsy and stroke are the main contributors to walking disorders. Machine learning methods have been proposed in the recent literature to analyze gait data from these patients. However, machine learning methods still fail to translate effectively into clinical applications. This systematic review addressed the gaps hindering the use of machine learning data analysis in the clinical assessment of cerebral palsy and stroke patients. RESEARCH QUESTION: What are the main challenges in transferring proposed machine learning methods to clinical applications? METHODS: PubMed, Web of Science, Scopus, and IEEE databases were searched for relevant publications on machine learning methods applied to gait analysis data from stroke and cerebral palsy patients until February the 23rd, 2023. Information related to the suitability, feasibility, and reliability of the proposed methods for their effective translation to clinical use was extracted, and quality was assessed based on a set of predefined questions. RESULTS: From 4120 resulting references, 63 met the inclusion criteria. Thirty-one studies used supervised, and 32 used unsupervised machine learning methods. Artificial neural networks and k-means clustering were the most used methods in each category. The lack of rationale for features and algorithm selection, the use of unrepresentative datasets, and the lack of clinical interpretability of the clustering outputs were the main factors hindering the clinical reliability and applicability of these methods. SIGNIFICANCE: The literature offers numerous machine learning methods for clustering gait data from cerebral palsy and stroke patients. However, the clinical significance of the proposed methods is still lacking, limiting their translation to real-world applications. The design of future studies must take into account clinical question, dataset significance, feature and model selection, and interpretability of the results, given their criticality for clinical translation.


Cerebral Palsy , Gait Analysis , Machine Learning , Stroke , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Humans , Stroke/complications , Stroke/physiopathology , Gait Analysis/methods , Gait Disorders, Neurologic/etiology , Gait Disorders, Neurologic/physiopathology
18.
Pediatr Neurol ; 155: 84-90, 2024 Jun.
Article En | MEDLINE | ID: mdl-38608553

BACKGROUND: Primary symptoms of cerebral palsy (CP), such as spasm and weakness, can lead to secondary musculoskeletal problems. Exploring the interplay and impact of secondary symptoms is essential in CP management. METHODS: A total of 56 children (32 males and 24 females) aged eight to 12 years in level I to III of Gross Motor Function Classification System (GMFCS) completed The Pediatric Balance Scale and Wong-Baker Faces Scale and the Posture and Postural Ability Scale. Relationships between the three groups were examined using the Kruskal-Wallis test, Tukey test, gamma coefficient, De Somers D, phi coefficient, Cramér V, and one-way analysis of variance. RESULTS: There was a significant correlation between balance and postural asymmetry (P < 0.001), and no significant difference in balance was there between the severe and moderate asymmetry groups (P = 0.759) and between the mild asymmetry and no asymmetry groups (P = 0.374). Furthermore, there was a significant relationship between postural asymmetry and each of the variables of pain (P < 0.001) and gross motor function (P = 0.002). Although a meaningful correlation was identified between balance and gross motor function (P < 0.001), the relationship between postural asymmetry and balance in GMFCS levels was not found (P = 0.052, P = 0.052, P = 0.233). Conversely, no significant relationship was detected between pain and gross motor function (P = 0.072). SIGNIFICANCE: Postural asymmetry negatively impacts balance and correlates with pain intensity. Addressing postural problems can contribute to pain management and improved balance.


Cerebral Palsy , Pain , Postural Balance , Humans , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Child , Female , Male , Postural Balance/physiology , Pain/physiopathology , Pain/etiology , Severity of Illness Index
19.
Pediatr Neurol ; 155: 133-140, 2024 Jun.
Article En | MEDLINE | ID: mdl-38640862

BACKGROUND: Children with congenital heart disease (CHD) have a higher prevalence of motor impairment secondary to brain injury, resulting in cerebral palsy (CP). The purpose of this study is to determine the prevalence of CP in CHD in a single-center cohort, stratify risk based on surgical mortality using Society of Thoracic Surgeons-European Association for Cardio-Thoracic Surgery (STAT) categories and identify risk factors. METHODS: Retrospective cohort study of pediatric patients registered in the University of Florida (UF) Society of Thoracic Surgeons Congenital Heart Surgery database from 2006 to 2017 with a diagnosis of CHD who continued follow-up for more than two years at UF. RESULTS: A total of 701 children with CHD met inclusion criteria. Children identified to have CP were 54 (7.7%). Most common presentation was spastic hemiplegic CP with a Gross Motor Function Classification System of level 2. Analysis of surgical and intensive care factors between the two groups showed that children with CHD and CP had longer time from admission to surgery (P = 0.003), higher STAT categories 4 and 5 (P = 0.038), and higher frequency of brain injury and seizures (P < 0.001). Developmental disabilities and rehabilitation needs were significantly greater for children with CHD and CP when compared with those with CHD alone (P < 0.001). CONCLUSIONS: In our cohort, 7.7% children with CHD develop CP; this is significantly higher than the 2010 US population estimate of 0.3%. Our study suggests higher STAT categories, brain injury, and seizures are associated with developing CP in children with CHD.


Cerebral Palsy , Heart Defects, Congenital , Humans , Cerebral Palsy/epidemiology , Cerebral Palsy/complications , Cerebral Palsy/etiology , Male , Female , Heart Defects, Congenital/surgery , Heart Defects, Congenital/complications , Heart Defects, Congenital/mortality , Retrospective Studies , Prevalence , Risk Factors , Infant , Child, Preschool , Child , Adolescent
20.
Eur J Paediatr Neurol ; 50: 41-50, 2024 May.
Article En | MEDLINE | ID: mdl-38614013

INTRODUCTION: Impaired upper limb movements are a key feature in dyskinetic cerebral palsy (CP). However, information on how specific movement patterns relate to manual ability, performance and underlying movement disorders is lacking. Insight in these associations may contribute to targeted upper limb management in dyskinetic CP. This study aimed to explore associations between deviant upper limb movement patterns and (1) manual ability, (2) severity of dystonia/choreoathetosis, and (3) movement time/trajectory deviation during reaching and grasping. PARTICIPANTS/METHODS: Participants underwent three-dimensional upper limb analysis during reaching forwards (RF), reaching sideways (RS) and reach-and-grasp vertical (RGV) as well as clinical assessment. Canonical correlation and regression analysis with statistical parametric mapping were used to explore associations between clinical/performance parameters and movement patterns (mean and variability). RESULTS: Thirty individuals with dyskinetic CP participated (mean age 16±5 y; 20 girls). Lower manual ability was related to higher variability in wrist flexion/extension during RF and RS early in the reaching cycle (p < 0.05). Higher dystonia severity was associated with higher mean wrist flexion (40-82 % of the reaching cycle; p = 0.004) and higher variability in wrist flexion/extension (31-75 %; p < 0.001) and deviation (2-14 %; p = 0.007/60-73 %; p = 0.006) during RF. Choreoathetosis severity was associated with higher elbow pro/supination variability (12-19 %; p = 0.009) during RGV. Trajectory deviation was associated with wrist and elbow movement variability (p < 0.05). CONCLUSION: Current novel analysis of upper limb movement patterns and respective timings allows to detect joint angles and periods in the movement cycle wherein associations with clinical parameters occur. These associations are not present at each joint level, nor during the full movement cycle. This knowledge should be considered for individualized treatment strategies.


Cerebral Palsy , Dystonia , Severity of Illness Index , Upper Extremity , Humans , Male , Female , Cerebral Palsy/physiopathology , Cerebral Palsy/complications , Adolescent , Upper Extremity/physiopathology , Child , Young Adult , Dystonia/physiopathology , Hand Strength/physiology , Athetosis/physiopathology , Movement/physiology
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