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Assessing Early Severity of Hypoxic-Ischemic Encephalopathy: The Role of Electroencephalogram Background in Addition to Sarnat Exam

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JOURNAL OF PEDIATRICS 2025年 277卷 114411-114411页

作者： Cornet, Marie-Coralie Numis, Adam L. Monsell, Sarah E. Chan, Natalie H. Gonzalez, Fernando F. Comstock, Bryan A. Juul, Sandra E. Wusthoff, Courtney J. Wu, Yvonne W. Glass, Hannah C. Univ Calif San Francisco Dept Pediat San Francisco CA 94143 USA Univ Calif San Francisco Dept Neurol San Francisco CA 94143 USA Univ Calif San Francisco Weill Inst Neurosci San Francisco CA 94143 USA Univ Washington Dept Biostat Seattle WA USA Univ Washington Sch Med Dept Pediat Seattle WA USA Stanford Univ Dept Neurol Palo Alto CA USA Univ Calif San Francisco Dept Epidemiol & Biostat San Francisco CA 94143 USA

Objective To assess the relationship between the Sarnat exam, early electroencephalogram (EEG) background, and death or neurodevelopmental impairment (NDI) at age 2 years among neonates with moderate to severe hypoxic-ischemic encephalopathy treated with therapeutic hypothermia. (n = 463) or amplitude-integrated electroencephalogram (n = 15) reports available on the first day after birth were included in this cohort study. A Sarnat exam was performed between 1 and 6 hours after birth, and neonates were classified into 3 groups of increasing severity based on the number of severe features (none, 1-2, or 3+). EEG background continuity was extracted from reports and categorized as normal, excessively discontinuous, or severely abnormal. The primary outcome was severe NDI or death at age 2. Results Among 478 neonates with hypoxic-ischemic encephalopathy, EEG background continuity was normal in 186 (39%), excessively discontinuous in 171 (36%), and severely abnormal in 121 (25%). For each additional severe feature on the Sarnat exam, the risk of abnormal EEG background increased by 16% (relative risk 1.16 [95% CI 1.09-1.23]). Both the Sarnat exam and EEG background severity were associated with an increased risk of severe NDI or death. After adjusting for Sarnat exam severity, severe EEG background remained associated with severe NDI and death (relative risk 5.7 [95% CI 3.7-8.9]). Conclusions The early EEG background provides additional information beyond the Sarnat exam and could be an additional early marker when assessing the severity of HIE. (J Pediatr 2025;277:114411).

关键词： infant hypoxia-ischemia brain hypothermia induced electroencephalography brain injury neurodevelopment aEEG Amplitude-integrated electroencephalogram EEG Electroencephalogram GMFCS gross motor function classification system HEAL High-dose Erythropoietin for Asphyxia and Encephalopathy HIE Hypoxic-ischemic encephalopathy NDI Neurodevelopmental impairment NICHD National Institute of Child Health and Human Development RR Relative risk TH Therapeutic hypothermia

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Femoral neck-shaft angle changes based on the severity of neurologic impairment in children with cerebral palsy and spinal muscular atrophy

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JOURNAL OF CHILDRENS ORTHOPAEDICS 2024年第5期18卷 523-530页

作者： da Silva, Luiz Carlos Almeida Hori, Yusuke Kaymaz, Burak Rogers, Kenneth J. Trionfo, Arianna Bowen, James Richard Howard, Jason J. Shrader, Michael Wade Miller, Freeman Nemours Childrens Hlth Dept Orthopaed Wilmington DE USA

Introduction: The neck-shaft angle and head-shaft angle in children with varying levels of neurological disability were evaluated to define change over different ***: Children aged 1-12 years with spastic cerebral palsy, spinal muscular atrophy types 1 and 2, or typical development were reviewed to evaluate the neck-shaft angle and head-shaft angle. Patients were divided into five groups: gross motor function classification system levels I and II, gross motor function classification system level III, gross motor function classification system levels IV and V, spinal muscular atrophy types 1 and 2, and typical development. A linear mixed model was utilized to evaluate neck-shaft angle and head-shaft ***: Data from 196 children (mean age 4.8 +/- 4.5 years) were included. gross motor function classification system levels I and II: 22 children, 130 hip radiographs measured, neck-shaft angle 143.7 +/- 7.4, and head-shaft angle 160.0 +/- 7.1. gross motor function classification system level III: 8 children, 33 hips evaluated, neck-shaft angle 153.1 +/- 4.3, and head-shaft angle 163.4 +/- 4.2. gross motor function classification system levels IV and V: 30 children, 137 hip radiographs measured, neck-shaft angle 156.4 +/- 5.6, and head-shaft angle 167.9 +/- 6.8. Spinal muscular atrophy types 1 and 2: 32 children, 83 hip radiographs measured, neck-shaft angle 161.9 +/- 9.7, and head-shaft angle 173.4 +/- 7.4. Typical development: 104 children, 222 hip radiographs measured, neck-shaft angle 138.6 +/- 7.0, and head-shaft angle 156.4 +/- 5.9. There were significant statistical differences when comparing neck-shaft angle and head-shaft ***: As children grow, neck-shaft angle and head-shaft angle tend to decrease in typical development and gross motor function classification system levels I and II groups. However, in low-tone (spinal muscular atrophy types 1 and 2) and high-tone groups (gross motor function classification system levels IV and V), n

关键词： Cerebral palsy spinal muscular atrophy gross motor function classification system neck-shaft angle head-shaft angle proximal femoral guided growth linear mixed model

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Scoliosis in children with severe cerebral palsy: a population-based study of 206 children at GMFCS levels III-V

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EUROPEAN SPINE JOURNAL 2023年第11期32卷 4030-4036页

作者： Vinje, Svend Terjesen, Terje Kibsgard, Thomas Oslo Univ Hosp Div Orthopaed Surg Rikshosp Postboks 4950 Nydalen N-0424 Oslo Norway Univ Oslo Inst Clin Med Fac Med Oslo Norway

PurposeTo evaluate the prevalence of scoliosis and the rate of scoliosis progression in children with severe cerebral palsy (CP) at GMFCS levels *** hundred and six children (86 girls, 120 boys), born 2002-2008, were recruited from The Norwegian Quality and Surveillance Registry for Cerebral Palsy (NorCP). Inclusion criteria were bilateral CP and GMFCS levels III-V. Scoliosis was evaluated annually by examination of the spine by a physiotherapist. Radiographic examination was performed in children with moderate or severe scoliosis at clinical evaluation. The Cobb angle was used as a measure of curve ***, defined as Cobb angle & GE;10 & DEG;, occurred in 121 children (59%). Severe scoliosis (Cobb angle & GE;40 & DEG;) developed in 80 of the 206 patients (39%) at a mean age of 10.9 years (range 5-16) and was more prevalent in children at GMFCS level V (62%) than at levels IV (19%) and III (6%). Initial Cobb angle, Cobb angle & GE;30 & DEG;at age 10 years, and GMFCS level V were independent risk factors for severe scoliosis. In children at GMFCS level V, the rate of scoliosis progression decreased with age from a mean of 9.7 & DEG;per year at age 3-5 years to 2-3 & DEG;per year in children & GE;11 *** prevalence of scoliosis among children with CP increased with decreasing motor functional level. The most important risk factors for progression of scoliosis were high initial Cobb angle, Cobb angle & GE;30 & DEG;at age 10 years, and GMFCS level V.

关键词： Scoliosis Cerebral palsy Natural history gross motor function classification system Cobb angle

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Physical and Psychosocial Consequences of Falls in Ambulatory Individuals With Cerebral Palsy by Age and gross motor function

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ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION 2025年第5期106卷 688-695页

作者： Esterley, Marissa Krach, Linda E. Pederson, Kari Wandersee, Nathan G. Tierney, Sandy Callen Boyer, Elizabeth R. Gillette Childrens Dept Res St Paul MN USA Gillette Childrens Dept Adult Phys Med & Rehabil Dept Rehabil Med St Paul MN USA Univ Minnesota Twin Cities Dept Rehabil Med Minneapolis MN USA Univ Minnesota Twin Cities Dept Orthoped Surg Minneapolis MN USA

Objective: To quantify physical and psychosocial impacts of falls by age and gross motor classification system (GMFCS) level in ambulatory individuals with cerebral palsy (CP). Design: Cross-sectional survey. Setting: Tertiary specialty hospital and online CP communities. Participants: Ambulatory individuals with CP (N=201 adults;18-76 years old) or the caregivers of minors (N=180;5-17 years old). Main Outcome Measure(s): Participants completed online surveys to report fall frequency, fall characteristics, injuries, concern about falling (Short Falls Efficacy Scale-International), activity avoidance due to concern about falling (Short Falls Efficacy Scale-International Avoidance Behavior), and psychosocial constructs related to perceived consequences of falling (Consequences of Falling-Damage to Identity subscale). Results: Most participants fell in the past 12 months (86%). Fall frequency in the past 12 months differed by age (P<.001) and GMFCS level (P=.021). Individuals aged 5-12 years or GMFCS level II reported the most falls. The percentage of participants who experienced a past serious fall-related injury (eg, head/face stitches, concussion, fractures) increased with age (P<.001), affecting 80% of >= 50-year olds. Falls often occurred in the forward direction, when wearing shoes, and during ambulation. Uneven surfaces and fatigue were notable causes. Concern about falling and associated activity avoidance scores differed by age and GMFCS level (all P<.01), both of which were highest for >= 50-year olds and GMFCS level III. Although psychosocial consequences of falls (eg, embarrassment, lost confidence) were elevated across all groups, they did not differ by age or GMFCS level (P=.130, P=.083). Nearly everyone (88%) wished they fell less. Conclusions: Falls are common for ambulatory children and adults with CP. Physical and psychosocial consequences of falls were frequent and impacted behavior. Differences observed by age and GMFCS level should be considered in

关键词： Accidental falls Anxiety Falls efficacy Postural balance CP cerebral palsy FES-I Falls Efficacy Scale-International FES-IAB Falls Efficacy Scale-International Avoidance Behavior GMFCS gross motor function classification system

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Relationship Between Epileptic Activity and Developmental Outcome in KCNQ2-Related Epilepsy

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PEDIATRIC NEUROLOGY 2023年 144卷 11-15页

作者： Casas-Alba, Didac Aguilar, Anna Alonso, Itziar Garcia, Maria Teresa Cilio, Maria Roberta Fons, Carmen Barcelona Univ Hosp Sant Joan Deu Pediat Neurol Dept ERN EpiCare Barcelona Spain Inst Recerca Sant Joan Deu Barcelona Spain Barcelona Univ Hosp Sant Joan Deu Pediat Inst Rare Dis IPER Dept Genet Med Barcelona Spain Hosp Infantil Univ Miguel Servet Pediat Neurol Unit Zaragoza Spain Catholic Univ Louvain St Luc Univ Hosp Div Pediat Neurol Brussels Belgium Ctr Invest Biomed Red Enfermedades Raras CIBERER ISCIII Madrid Spain

Background: We aim to describe a cohort of patients with KCNQ2-related epilepsy and evaluate the relationship between epileptic activity and developmental outcome. This topic is relevant for the se-lection of clinical end points in future clinical trials, since cessation of seizures may or may not be the most important outcome. Methods: This retrospective cohort study of children with self-limited (familial) neonatal epilepsy and developmental and epileptic encephalopathy due to pathogenic variants in KCNQ2 was conducted be-tween 2019 and 2021. We collected clinical, therapeutic, and genetic information. Available electroen-cephalographic recordings were reviewed by a neurophysiologist. gross motor function was determined using the gross motor function classification system (GMFCS). The Vineland Adaptive Behavior Com-posite standard score (ABC SS) was used to measure adaptive functioning. Results: Among 44 children (mean age 8.1 +/- 4.0 years, 45.5% were male), 15 of 44 had S(F)NE, and 29 of 44 had DEE. Delayed seizure freedom was more frequent in DEE than in S(F)NE (P = 0.025), but no correlation was observed between age at seizure freedom and developmental outcome in patients with DEE. Multifocal interictal epileptiform abnormalities at epilepsy onset were more frequent in DEE than in S(F)NE (P = 0.014), and were associated with higher GMFCS (P = 0.027) and lower ABC SS (P = 0.048) in patients with DEE. Disorganized background activity at follow-up was more frequent in DEE than in S(F)NE (P = 0.001), and was associated with higher GMFCS levels (P = 0.009) and lower ABC SS (P = 0.005) in patients with DEE. Conclusions: This study shows a partial correlation between epileptic activity and developmental outcome in KCNQ2-related epilepsy. (c) 2023 Elsevier Inc. All rights reserved.

关键词： KCNQ2 Self-limited (familial) neonatal epilepsy Developmental and epileptic encephalopathy gross motor function classification system Vineland Adaptive Behavior Scales

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The Profile of Epilepsy and its characteristics in Children with Cerebral Palsy

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SEIZURE-EUROPEAN JOURNAL OF EPILEPSY 2022年 101卷 190-196页

作者： Archana, K. Saini, Lokesh Gunasekaran, Pradeep Kumar Singh, Paramjeet Sahu, Jitendra Kumar Sankhyan, Naveen Sharma, Rajni Bhati, Ankita Yadav, Jaivinder Sharawat, Indar Kumar Postgrad Inst Med Educ & Res Dept Pediat Chandigarh 160012 India All India Inst Med Sci Dept Pediat Jodhpur 342005 Rajasthan India Postgrad Inst Med Educ & Res Dept Radiodiag Chandigarh 160012 India All India Inst Med Sci Dept Pediat Rishikesh 249201 Uttaranchal India Inst All India Inst Med Sci Dept Pediat Jodhpur Rajasthan India

Purpose: To determine the characteristics of epilepsy in children with Cerebral Palsy (CP) visiting the Pediatric Outpatient Department (OPD) of a tertiary care hospital. Methods: This cross-sectional study was carried out at a tertiary care pediatric hospital. All children with CP aged between 1 and 12 years seen at this hospital during an 18 months period (January 2018 to July 2019) were included. Children with CP who had seizures were studied in detail. Seizure semiologies were classified ac-cording to the International League Against Epilepsy (ILAE) 1981 and 2017 classifications. The severity of sei-zures was assessed with the Early Childhood Epilepsy Severity Scale (E-Chess). functional impairment was characterised using the gross motor function classification system (GMFCS) score. Results: Of 300 children with CP, 207 (69%) were male and 93 (31%) female. The mean age was 45.17 & PLUSMN;31.12 months. Seizures were present in 79 (26%) children. 89.9% of children had drug-responsive epilepsy, and 10.1% had refractory epilepsy. Seizures were present in 30.4% of children with a spastic hemiplegia CP subtype, 28.7% with spastic quadriplegia, 26.3% with spastic diplegia, 24% with mixed type CP, and 6.3% with dyskinetic CP. On E-Chess assessment, the median score was 8 (4-14). The majority had poor gross motor function Classifi-cation system (GMFCS) scores (> III). Conclusion: The prevalence of epilepsy in the studied population of children with CP was 26%. The highest incidence of seizures was in the spastic hemiplegia subtype (30.4%). The severity of cortical damage is positively correlated with the risk of having epilepsy. The primary determinant of severity of the GMFCS score was the type of CP and not the presence or absence of epilepsy.

关键词： Cerebral palsy epilepsy ILAE classification gross motor function classification system

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Determinants of Hospital-Based Health Service Utilization in Cerebral Palsy: a systematic Review

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ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION 2022年第8期103卷 1628-1637页

作者： Paget, Simon Ostojic, Katarina Goldsmith, Shona Nassar, Natasha McIntyre, Sarah Univ Sydney Childrens Hosp Fac Med & Hlth Westmead Clin Sch Sydney NSW Australia Childrens Hosp Kids Rehab Westmead NSW Australia Univ Sydney Cerebral Palsy Alliance Discipline Child & Adolescent Hlth Sydney NSW Australia Univ Sydney Childrens Hosp Fac Med & Hlth Westmead Clin SchChild Populat & Translat Hlth Sydney NSW Australia

Objective: To systematically review and synthesize evidence of determinants associated with hospital-based health service utilization among individuals with cerebral palsy (CP). Data Sources: Electronic databases MEDLINE, Embase, APA Psycinfo were searched from January 2000 to April 2020. Study Selection: Observational studies were included that described people with CP, reported quantitative measures of hospital-based health service utilization (inpatient, outpatient, emergency department), and based in high-income countries. We excluded studies that included only subsets of people with CP, or those that only reported therapy service utilization. Data Extraction: After initial screen, 2 reviewers reviewed full texts for inclusion and performed data extraction and risk of bias assessment using the Newcastle-Ottawa scale. Determinants of health service utilization were identified and categorized using the Andersen behavioral model. Data Synthesis: Seventeen studies met inclusion criteria. Study quality was high. Twenty-six determinants were reported across 8 Andersen model characteristics. Individual predisposing factors such as sex showed no difference in health service utilization;inpatient admissions decreased with increasing age during childhood and was lower in adults. Increased health service utilization was associated with "individual need" including severe gross motor disability, epilepsy, developmental/ intellectual disability and gastrostomy-use across inpatient, outpatient and emergency department settings. There was little information reported on socio-demographic and health system contextual determinants. Conclusions: CP health service utilization is associated with age, severity and comorbidities. Improved understanding of determinants of health service utilization can support health service access for people with CP. Crown Copyright (c) 2021 Published by Elsevier Inc. on behalf of the American Congress of Rehabilitation Medicine.

关键词： Cerebral palsy Health services Health services accessibility Rehabilitation CP cerebral palsy GMFCS gross motor function classification system IRR incident rate ratio OR odds ratio

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Interobserver reliability of the Turkish version of the expanded and revised gross motor function classification system

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DISABILITY AND REHABILITATION 2012年第12期34卷 1030-1033页

作者： El, Ozlem Baydar, Meltem Berk, Haluk Peker, Ozlen Kosay, Can Demiral, Yucel Dokuz Eylul Univ Fac Med Dept Phys Med & Rehabil TR-35340 Izmir Turkey Dokuz Eylul Univ Fac Med Dept Orthopaed TR-35340 Izmir Turkey Dokuz Eylul Univ Fac Med Dept Publ Hlth TR-35340 Izmir Turkey

Purpose: Cerebral palsy (CP) is the most common disability in childhood. The gross motor function classification system (GMFCS) has become an important tool to assess motor function in CP patient. In 2007, the expanded and revised (E&R) version of GMFCS which includes age band for youth 12-18 years of age was developed. The aim of this study was to evaluate reliability of Turkish version of expanded and revised GMCS. Methods: We assessed interobserver reliability between two physical medicine and rehabilitation specialists in 136 children with CP and test-retest reliability within a subgroup of 48 patients. Percent agreement, intraclass correlation coefficient (ICC) and mu statistics were used to evaluate reliability. Result: The ICC between two physicians was 0.97 and the total agreement was 89%. This result indicates excellent agreement. The overall weighted mu was 0.86. High test-retest reliability was found (ICC: 0.94 95% confidence interval) and the total agreement was 75% for test-retest reliability. Conclusion: The Turkish version of the E&R GMFCS is shown to be reliable and valid for assessment of Turkish CP children.

关键词： Cerebral palsy gross motor function classification system reliability Turkey

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Long-term Follow-up of Patients With Cerebral Palsy Undergoing Catheterizable Channel Creation Without Concurrent Bladder Augmentation

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Urology 2022年 170卷 234-239页

作者： DeWitt-Foy, Molly E. Gotlieb, Rachael Elliott, Sean P. Cleveland Clinic Department of Urology Glickman Urological and Kidney Institute Cleveland OH Department of Urology Lahey Hospital and Medical Center Burlington MA Department of Urology University of Minnesota Minneapolis MN United States

Objective: To characterize long-term outcomes for adults with cerebral palsy who have undergone catheterizable channel creation without concurrent bladder augmentation. Methods: Retrospective review was conducted of patients who underwent catheterizable channel creation without augmentation by the senior author. Variables of interest included development of de novo neurogenic detrusor overactivity, change in continence, escalation in therapy, and upper tract changes. Descriptive statistics were conducted using t-tests and chi-squared tests as appropriate. Results: Nine patients were followed for an average of 70 months. Prior to surgery two patients were on regular clean intermittent catheterization (CIC), six were not on CIC, and one was on occasional CIC. Patients not on CIC preoperatively were more likely to develop de novo neurogenic detrusor overactivity (83% vs 0%, P = .02), and have statistically significant decreases in average compliance (P = .04 vs P = .31). They were also more likely to require escalation in bladder therapy (83% vs 50%) and have worsening of incontinence (67% vs 0%), though these did not reach statistical significance (P = .34, 0.1). Five patients underwent repeat urodynamics an average of 46 months after initial postoperative study because of persistent urgency - 4 of 5 had stable urodynamic findings and one demonstrated >50% reduction in compliance and capacity. Conclusion: Adults with cerebral palsy who are not on CIC prior to creation of a catheterizable channel are at high risk for development of de novo neurogenic detrusor overactivity and decrease in bladder compliance. Prophylactic augmentation should be considered in this group. © 2022 Elsevier Inc.

关键词： AC anticholinergics BoNT botulinum toxin CC catheterizable channel CIC clean intermittent catheterization CP cerebral palsy DO detrusor overactivity DSD detrusor sphincter dyssynergia GMFCS gross motor function classification system NDO neurogenicdetrusor overactivity UDS urodynamics UUI urge urinary incontinence

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NEUROREHABILITATION 2021年第4期48卷 481-491页

作者： Mohon, Ricky T. Sawyer, Kim Pickett, Kaci Bothwell, Samantha Brinton, John T. Sobremonte-King, Michelle DelRosso, Lourdes M. Univ Colorado Childrens Hosp Colorado Dept Pediat Sch Med Aurora CO 80045 USA Univ Colorado Div Pulm & Sleep Med Dept Pediat Sch Med Aurora CO 80045 USA Univ Colorado Childrens Hosp Colorado Dept Rehabil Sch Med Aurora CO 80045 USA Univ Colorado Colorado Sch Publ Hlth Dept Biostat & Informat Aurora CO 80045 USA Univ Colorado Dept Pediat Sch Med Anschutz Med Campus Aurora CO 80045 USA Univ Washington Seattle Childrens Hosp Seattle WA 98195 USA Univ Washington Div Pulm & Sleep Med Seattle WA 98195 USA

BACKGROUND: Patients with cerebral palsy and other static encephalopathies (CP) are known to be at increased risk of sleep-related breathing disorders (SRBD). Few studies have reviewed whether intrathecal baclofen (ITB) can contribute to SRBD. OBJECTIVE: To assess the prevalence of SRBD in patients with CP receiving ITB by using nocturnal polysomnography (NPSG). METHODS: We performed a retrospective chart review of patients receiving ITB who had NPSG at Children's Hospital Colorado (CHCO) and Seattle Children's Hospital (SCH) from 1995 to 2019. The gross motor function classification system (GMFCS) measured the severity of motor disability. Screening sleep questionnaires collected subjective data and NPSG provided objective data of SRBD. RESULTS: All patients except one were GMFCS 4 or 5 with median age at ITB pump placement of 9.7 years. The screening questionnaire for SRBD detected one or more nighttime symptoms in > 82% of all patient groups. Pre-ITB criteria for a SRBD was met in 83% of patients at CHCO and 91% at SCH. Post-ITB prevalence remained similarly high. CONCLUSIONS: NPSG identified a high prevalence of SRBD in these cohorts from CHCO and SCH. Our study showed neither improvement nor worsening of SRBD in patients receiving ITB.

关键词： Cerebral palsy static encephalopathies gross motor function classification system GMFCS nocturnal polysomnography NPSG Baclofen intrathecal Baclofen ITB obstructive sleep apnea OSA central sleep apnea CSA periodic breathing PB sleep-related hypoxemia and hypoventilation

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