28 results on '"Vargus-Adams J"'
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2. Domains of importance for parents, medical professionals and youth with cerebral palsy considering treatment outcomes
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Vargus-Adams, J. N. and Martin, L. K.
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- 2011
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3. Assessing change in cerebral palsy: variability between individual perceptions and standardized measures: H5
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VARGUS-ADAMS, J, MARTIN, L, MAIGNAN, S, KLEIN, A, and SALISBURY, S
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- 2010
4. Practice Parameter: Pharmacologic treatment of spasticity in children and adolescents with cerebral palsy (an evidence-based review)
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Delgado, M. R., primary, Hirtz, D., additional, Aisen, M., additional, Ashwal, S., additional, Fehlings, D. L., additional, McLaughlin, J., additional, Morrison, L. A., additional, Shrader, M. W., additional, Tilton, A., additional, and Vargus-Adams, J., additional
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- 2010
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5. Longitudinal use of the Child Health Questionnaire in childhood cerebral palsy.
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Vargus-Adams J
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This study sought to describe change and stability in health-related quality of life (HRQL) over time in a cross-section of children with cerebral palsy (CP) using the Child Health Questionnaire (CHQ) through repeated surveys of the children's parents/caregivers. A total of 177 children with CP (98 males, 79 females); age range 3 to 18 years (mean age 8y 6mo, [SD 4y 2mo]; Gross Motor Function Classification System: Level I 40%, Level II 14%, Level III 14%, Level IV 16%, and Level V 17%) were enrolled as a convenience sample from the outpatient clinics at a tertiary-care children's hospital. The main outcome measure was HRQL as determined by the CHQ - Parent Form 50. Parents reported reduced HRQL for their children with CP relative to norms for the CHQ. Although Role - Physical functioning (reflecting how physical impairment interferes with daily activity) declined with time, all other subscales of the CHQ were stable over the course of 1 year. CHQ scores were not measurably affected by common medical interventions. Children with CP have lower CHQ scores than other children that are, on average, stable over the course of 1 year. [ABSTRACT FROM AUTHOR]
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- 2006
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6. Mental Health Diagnoses Risk Among Children and Young Adults With Cerebral Palsy, Chronic Conditions, or Typical Development.
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Bhatnagar S, Mitelpunkt A, Rizzo JJ, Zhang N, Guzman T, Schuetter R, Vargus-Adams J, Bailes AF, Greve K, Gerstle M, Pedapati E, Aronow B, and Kurowski BG
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- Humans, Male, Female, Case-Control Studies, Adolescent, Child, Chronic Disease epidemiology, Young Adult, Child, Preschool, Midwestern United States epidemiology, Cerebral Palsy epidemiology, Mental Disorders epidemiology
- Abstract
Importance: Mental health (MH) issues in children with cerebral palsy (CP) are poorly understood compared with other pediatric populations., Objective: To examine MH diagnosis code assignment among children and young adults with CP and compare with typically developing (TD) and chronic condition (CC) pediatric populations., Design, Setting, and Participants: This case-control study used International Statistical Classification of Diseases, Tenth Revision, Clinical Modification (ICD-10-CM) codes to create a CP case set and CC and TD control sets using electronic health record data of children and young adults from a large tertiary care children's hospital in the midwestern United States between 2010 and 2022. Case-control matching was performed to control for demographic factors. Data were analyzed from June to December 2023., Exposures: All MH diagnosis codes were mapped to ICD-10-CM and categorized using Clinical Classifications Software Refined (CCSR)., Main Outcomes and Measures: The incidence rates of MH CCSR categories were calculated. Descriptive and comparative statistics were used to evaluate the significance and odds associated with factors., Results: Data from 216 794 individuals (mean [SD] baseline age, 4.3 [5.1] years; 118 562 [55%] male) were analyzed, including 3544 individuals with CP, 142 160 individuals with CC, and 71 080 TD individuals. The CP cohort spread across Gross Motor Function Classification System (GMFCS) levels I (981 individuals [28%]), II (645 individuals [18%]), III (346 individuals [10%]), IV (502 individuals [14%]), and V (618 individuals [17%]). Rates varied significantly for anxiety (824 individuals with CP [23%]; 25 877 individuals with CC [9%]; 6274 individuals with TD [18%]), attention-deficit/hyperactivity disorder (534 individuals with CP [15%]; 22 426 individuals with CC [9%]; 6311 individuals with TD [16%]); conduct or impulse disorder (504 individuals with CP [14%]; 13 209 individuals with CC [5%]; 3715 individuals with TD [9%]), trauma or stress disorders (343 individuals with CP [10%]; 18 229 individuals with CC [8%]; 5329 individuals with TD [13%]), obsessive-compulsive disorder (251 individuals with CP [7%]; 3795 individuals with CC [1%]; 659 individuals with TD [3%]), depression (108 individuals with CP [3%]; 12 224 individuals with CC [5%]; 4007 individuals with TD [9%]), mood disorders (74 individuals with CP [2%]; 4355 individuals with CC [2%]; 1181 individuals with TD [3%]), and suicidal ideation (72 individuals with CP [2%]; 7422 individuals with CC [5%]; 3513 individuals with TD [5%]). There was significant variation in odds of MH diagnoses by GMFCS level (I-II vs III-V: odds ratio [OR], 1.23; 95% CI, 1.09-1.40; P = .001). Among individuals with CP, males were more likely than females to have diagnosis codes for conduct or impulse disorders (OR, 1.41; 95% CI, 1.16-1.73) and attention-deficit/hyperactivity disorder (OR, 1.41 [95% CI, 1.15-1.73]). Black individuals, compared with White individuals, were more likely to have diagnoses for obsessive-compulsive disorder (OR, 1.57 [95% CI, 1.14-2.16]), other mood disorders (OR, 1.85 [95% CI, 1.01-3.38]), and trauma or stress disorders (OR, 1.94 [95% CI, 1.44-2.63]). Odds for trauma or stress disorders were elevated for individuals who identified as other races compared with White individuals (OR, 2.80 [95% CI, 2.03-3.87])., Conclusions and Relevance: In this case-control study of children and young adults with CP and matched comparisons, anxiety and conduct or impulse diagnoses were higher in individuals with CP. The lower diagnosis rates of depression and suicidal ideation may indicate underdiagnosis among individuals with CP. There is likely a need for assessment tools that are more suitable for children with CP.
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- 2024
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7. A big data approach to evaluate receipt of optimal care in childhood cerebral palsy.
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Mitelpunkt A, Stodola MA, Vargus-Adams J, Kurowski BG, Greve K, Bhatnagar S, Aronow B, Zahner J, and Bailes AF
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- Male, Female, Child, Humans, Adolescent, Quality of Life, Big Data, Muscle Spasticity therapy, Cerebral Palsy rehabilitation, Occupational Therapy
- Abstract
Purpose: Through automated electronic health record (EHR) data extraction and analysis, this project systematically quantified actual care delivery for children with cerebral palsy (CP) and evaluated alignment with current evidence-based recommendations., Methods: Utilizing EHR data for over 8000 children with CP, we developed an approach to define and quantify receipt of optimal care, and pursued proof-of-concept with two children with unilateral CP, Gross Motor Function Classification System (GMFCS) Level II. Optimal care was codified as a cluster of four components including physical medicine and rehabilitation (PMR) care, spasticity management, physical therapy (PT), and occupational therapy (OT). A Receipt of Care Score (ROCS) quantified the degree of adherence to recommendations and was compared with the Pediatric Outcomes Data Collection Instrument (PODCI) and Pediatric Quality of Life Inventory (PEDS QL)., Results: The two children (12 year old female, 13 year old male) had nearly identical PMR and spasticity component scores while PT and OT scores were more divergent. Functional outcomes were higher for the child who had higher adjusted ROCS., Conclusions: ROCSs demonstrate variation in real-world care delivered over time and differentiate between components of care. ROCSs reflect overall function and quality of life. The ROCS methods developed are novel, robust, and scalable and will be tested in a larger sample.IMPLICATIONS FOR REHABILITATIONOptimal practice, with an emphasis on integrated multidisciplinary care, can be defined and quantified utilizing evidence-based recommendations.Receipt of optimal care for childhood cerebral palsy can be scored using existing electronic health record data.Big Data approaches can contribute to the understanding of current care and inform approaches for improved care.
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- 2024
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8. Early Cerebral Palsy Detection and Intervention.
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Mendoza-Sengco P, Lee Chicoine C, and Vargus-Adams J
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- Child, Infant, Humans, Motor Skills, Risk Factors, Early Diagnosis, Risk Assessment, Cerebral Palsy diagnosis, Cerebral Palsy therapy
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Early identification of cerebral palsy (CP) facilitates optimal care, support, and outcomes for children and their families. Ideally, infants with risk factors or developmental deviations should be evaluated early using standardized assessments of neurodevelopment and brain imaging. If a diagnosis of CP or high risk for CP (HRCP) is established, specialized, evidence-informed therapy and family support should be initiated. With task-specific motor skill training and an enriched environment, infants with CP show greater gross motor and cognitive gains. These enhanced outcomes are only achievable with early diagnosis and subsequent intervention., (Copyright © 2023 Elsevier Inc. All rights reserved.)
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- 2023
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9. Gross motor function prediction using natural language processing in cerebral palsy.
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Greve K, Ni Y, Bailes AF, Vargus-Adams J, Miley AE, Aronow B, McMahon MM, Kurowski BG, and Mitelpunkt A
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- Child, Humans, Male, Adolescent, Young Adult, Adult, Female, Natural Language Processing, Retrospective Studies, Cross-Sectional Studies, Electronic Health Records, Cerebral Palsy complications, Cerebral Palsy diagnosis
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Aim: To predict ambulatory status and Gross Motor Function Classification System (GMFCS) levels in patients with cerebral palsy (CP) by applying natural language processing (NLP) to electronic health record (EHR) clinical notes., Method: Individuals aged 8 to 26 years with a diagnosis of CP in the EHR between January 2009 and November 2020 (~12 years of data) were included in a cross-sectional retrospective cohort of 2483 patients. The cohort was divided into train-test and validation groups. Positive predictive value, sensitivity, specificity, and area under the receiver operating curve (AUC) were calculated for prediction of ambulatory status and GMFCS levels., Results: The median age was 15 years (interquartile range 10-20 years) for the total cohort, with 56% being male and 75% White. The validation group resulted in 70% sensitivity, 88% specificity, 81% positive predictive value, and 0.89 AUC for predicting ambulatory status. NLP applied to the EHR differentiated between GMFCS levels I-II and III (15% sensitivity, 96% specificity, 46% positive predictive value, and 0.71 AUC); and IV and V (81% sensitivity, 51% specificity, 70% positive predictive value, and 0.75 AUC)., Interpretation: NLP applied to the EHR demonstrated excellent differentiation between ambulatory and non-ambulatory status, and good differentiation between GMFCS levels I-II and III, and IV and V. Clinical use of NLP may help to individualize functional characterization and management., What This Paper Adds: Natural language processing (NLP) applied to the electronic health record (EHR) can predict ambulatory status in children with cerebral palsy (CP). NLP provides good prediction of Gross Motor Function Classification System level in children with CP using the EHR. NLP methods described could be integrated in an EHR system to provide real-time information., (© 2022 Mac Keith Press.)
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- 2023
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10. Electronic health record and patterns of care for children with cerebral palsy.
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Kurowski BG, Greve K, Bailes AF, Zahner J, Vargus-Adams J, Mcmahon MA, Aronow BJ, and Mitelpunkt A
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- Adolescent, Child, Child, Preschool, Electronic Health Records, Female, Humans, Infant, Male, Young Adult, Cerebral Palsy therapy, Patient Care Team
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Aim: To characterize the patterns of care of children with cerebral palsy (CP) in a tertiary healthcare system., Method: Electronic health record data from 2009 to 2019 were extracted for children with CP. Machine learning hierarchical clustering was used to identify clusters of care. The ratio of in-person to care coordination visits was calculated for each specialty., Results: The sample included 6369 children with CP (55.7% males, 44.3% females, 76.2% white, 94.7% non-Hispanic; with a mean age of 8y 2mo [SD 5y 10mo; range 0-21y; median 7y 1mo]) at the time of diagnosis. A total of 3.7 million in-person visits and care coordination notes were identified across 34 specialties. The duration of care averaged 5 years 5 months with five specialty interactions and 21.8 in-person visits per year per child. Seven clusters of care were identified, including: musculoskeletal and function; neurological; high-frequency/urgent care services; procedures; comorbid diagnoses; development and behavioral; and primary care. Network analysis showed shared membership among several clusters., Interpretation: Coordination of care is a central element for children with CP. Medical informatics, machine learning, and big data approaches provide unique insights into care delivery to inform approaches to improve outcomes for children with CP. What this paper adds Seven primary clusters of care were identified: musculoskeletal and function; neurological; high-frequency/urgent care services; procedures; comorbid diagnoses; development and behavioral; and primary care. The in-person to care coordination visit ratio was 1:5 overall for healthcare encounters. Most interactions with care teams occur outside of in-person visits. The ratio of in-person to care coordination activities differ by specialty., (© 2021 Mac Keith Press.)
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- 2021
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11. A Systematic Review of Assessments and Interventions for Chronic Pain in Young Children With or at High Risk for Cerebral Palsy.
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Letzkus L, Fehlings D, Ayala L, Byrne R, Gehred A, Maitre NL, Noritz G, Rosenberg NS, Tanner K, Vargus-Adams J, Winter S, Lewandowski DJ, and Novak I
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- Cerebral Palsy psychology, Child, Preschool, Female, Humans, Infant, Male, Pain Measurement trends, Physical Examination trends, Surveys and Questionnaires, Cerebral Palsy complications, Chronic Pain therapy, Pain Measurement methods, Physical Examination methods
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Background: Pain is common in children with cerebral palsy. The purpose of this systematic review was to evaluate the evidence regarding assessments and interventions for chronic pain in children aged ≤2 years with or at high risk for cerebral palsy., Methods: A comprehensive literature search was performed. Included articles were screened using PRISMA guidelines and quality of evidence was reviewed using best-evidence tools by independent reviewers. Using social media channels, an online survey was conducted to elicit parent preferences., Results: Six articles met criteria. Parent perception was an assessment option. Three pharmacologic interventions (gabapentin, medical cannabis, botulinum toxin type A) and 1 nonpharmacologic intervention were identified. Parent survey report parent-comfort and other nonpharmacologic interventions ranked as most preferable., Conclusion: A conditional GRADE recommendation was in favor of parent report for pain assessment. Clinical trials are sorely needed because of the lack of evidence for safety and efficacy of pharmacologic interventions.
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- 2021
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12. Assessments and Interventions for Spasticity in Infants With or at High Risk for Cerebral Palsy: A Systematic Review.
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Ayala L, Winter S, Byrne R, Fehlings D, Gehred A, Letzkus L, Noritz G, Paton MCB, Pietruszewski L, Rosenberg N, Tanner K, Vargus-Adams J, Novak I, and Maitre NL
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- Humans, Infant, Muscle Spasticity etiology, Cerebral Palsy complications, Muscle Spasticity diagnosis, Muscle Spasticity therapy
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Background: The majority of children with cerebral palsy develop spasticity, which interferes with motor development, function, and participation. This systematic review appraised current evidence regarding assessments and interventions for spasticity in children aged less than two years with or at high risk for cerebral palsy and integrated findings with parent preferences., Methods: Five databases (CINAHL, EMBASE, OVID/Medline, SCOPUS, and PsycINFO) were searched. Included articles were screened using PRISMA guidelines. Quality of the evidence was reviewed by two independent reviewers using Quality Assessment of Diagnostic Accuracy Studies, second edition (QUADAS-2), the RTI Item Bank on Risk of Bias and Precision of Observational Studies (RTI), or The Cochrane Collaboration's tool for assessing risk of bias in randomized trials (RoB). An online survey was conducted regarding parent preferences through social media channels., Results: Twelve articles met inclusion criteria. No high-quality assessment tool emerged for this population. Six interventions (botulinum toxin-A, orthotic use, radial extracorporeal shock wave therapy, erythropoietic stimulating agents, medical cannabis, and homeopathy) were identified. There was low-quality evidence for the use of botulinum toxin-A and radial extracorporeal shock wave therapy to improve short-term outcomes. Survey respondents indicated that spasticity assessments and interventions are highly valued, with nonpharmacologic interventions ranked most preferably., Conclusions: Further research is needed to validate assessments for spasticity in children younger than two years. Conditional recommendations can be made for botulinum toxin-A and radial extracorporeal shock wave therapy based on low level of evidence to reduce spasticity in children aged less than two years., (Copyright © 2020 The Authors. Published by Elsevier Inc. All rights reserved.)
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- 2021
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13. Assessments and Interventions for Sleep Disorders in Infants With or at High Risk for Cerebral Palsy: A Systematic Review.
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Tanner K, Noritz G, Ayala L, Byrne R, Fehlings D, Gehred A, Letzkus L, Novak I, Rosenberg N, Vargus-Adams J, Winter S, and Maitre NL
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- Cerebral Palsy diagnosis, Cerebral Palsy therapy, Child, Preschool, Humans, Infant, Sleep Wake Disorders etiology, Cerebral Palsy complications, Sleep Wake Disorders diagnosis, Sleep Wake Disorders therapy
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Background: Children with cerebral palsy (CP) are five times more likely than typically developing children to have sleep problems, resulting in adverse outcomes for both children and their families. The purpose of this systematic review was to gather current evidence regarding assessments and interventions for sleep in children under age 2 years with or at high risk for CP and integrate these findings with parent preferences., Methods: Five databases (CINAHL, EMBASE, OVID/Medline, SCOPUS, and PsycINFO) were searched. Included articles were screened using preferred reporting items for systematic reviews and meta-analyses guidelines, and quality of the evidence was reviewed using best evidence tools by two independent reviewers at minimum. An online survey was conducted regarding parent preferences through social media channels., Results: Eleven articles met inclusion criteria. Polysomnography emerged as the only high-quality assessment for the population. Three interventions (medical cannabis, surgical interventions, and auditory, tactile, visual, and vestibular stimulations) were identified; however, each only had one study of effectiveness. The quality of evidence for polysomnography was moderate, while the quality and quantity of the evidence regarding interventions was low. Survey respondents indicated that sleep assessments and interventions are highly valued, with caregiver-provided interventions ranked as the most preferable., Conclusions: Further research is needed to validate affordable and feasible sleep assessments compared to polysomnography as the reference standard. In the absence of diagnosis-specific evidence of safety and efficacy of sleep interventions specific to young children with CP, it is conditionally recommended that clinicians follow guidelines for safe sleep interventions for typically developing children., (Copyright © 2020 The Authors. Published by Elsevier Inc. All rights reserved.)
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- 2021
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14. Describing the Delivery of Evidence-Based Physical Therapy Intervention to Individuals With Cerebral Palsy.
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Bailes AF, Greve K, Long J, Kurowski BG, Vargus-Adams J, Aronow B, and Mitelpunkt A
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- Child, Humans, Orthotic Devices, Physical Therapy Modalities, Retrospective Studies, Cerebral Palsy
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Purpose: To characterize by evidence grades and examine variation in type of physical therapy intervention delivered in routine clinical care in individuals with cerebral palsy (CP)., Methods: Retrospective data collection from the electronic record over 1 year at a tertiary care pediatric outpatient therapy division., Results: Four hundred sixty-five individuals with CP received 28 344 interventions during 4335 treatment visits. Sixty-six percent of interventions were evidence-based interventions (EBIs). Significant variation was demonstrated across Gross Motor Function Classification System levels, with children classified as level V receiving the least and level III the most. The most frequent EBIs delivered were caregiver education, motor control, functional strengthening, ankle-foot orthoses, treadmill training, and fit of adaptive equipment., Conclusions: Further work is needed to determine whether amount of EBI is related to better outcomes. Combining this information with other aspects of dose (intensity, time, and frequency) may elucidate the contribution of each with outcomes., Competing Interests: The authors declare no conflicts of interest., (Copyright © 2021 Academy of Pediatric Physical Therapy of the American Physical Therapy Association.)
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- 2021
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15. Transformative journeys in childhood-onset disability with the AACPDM.
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Vargus-Adams J
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- Cerebral Palsy diagnosis, Child, Congresses as Topic, Humans, Societies, Medical, Cerebral Palsy therapy, Disabled Children
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- 2018
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16. Overview of Four Functional Classification Systems Commonly Used in Cerebral Palsy.
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Paulson A and Vargus-Adams J
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Cerebral palsy (CP) is the most common physical disability in childhood. CP comprises a heterogeneous group of disorders that can result in spasticity, dystonia, muscle contractures, weakness and coordination difficulty that ultimately affects the ability to control movements. Traditionally, CP has been classified using a combination of the motor type and the topographical distribution, as well as subjective severity level. Imprecise terms such as these tell very little about what a person is able to do functionally and can impair clear communication between providers. More recently, classification systems have been created employing a simple ordinal grading system of functional performance. These systems allow a more precise discussion between providers, as well as better subject stratification for research. The goal of this review is to describe four common functional classification systems for cerebral palsy: the Gross Motor Function Classification System (GMFCS), the Manual Ability Classification System (MACS), the Communication Function Classification System (CFCS), and the Eating and Drinking Ability Classification System (EDACS). These measures are all standardized, reliable, and complementary to one another.
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- 2017
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17. Transition of Care in Adolescents With Cerebral Palsy: A Survey of Current Practices.
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Bolger A, Vargus-Adams J, and McMahon M
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- Adolescent, Attitude of Health Personnel, Female, Humans, Male, Surveys and Questionnaires, United States, Cerebral Palsy therapy, Patient Transfer, Practice Patterns, Physicians', Transition to Adult Care
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Background: Transition of care from pediatric to adult health care providers for youth with special needs (including cerebral palsy [CP]) is of current interest because these individuals are now living well into adulthood. Studies have attempted to identify barriers to transition, ideal timing for transition of care, and key elements for successful transition programs. These studies often encompass a wide range of diagnoses, and results cannot be fully applied to those with CP., Objective: To identify and describe current transition-of-care (TOC) practices and beliefs among physician providers of adolescents with CP in multidisciplinary CP clinics., Design: Descriptive survey., Setting: Multidisciplinary CP clinics in the United States., Participants: Physician leaders in the aforementioned CP clinics., Methods: Respondents completed an electronic survey. Responses were deidentified and reported in aggregate by the use of descriptive statistics., Main Outcome Measure: Electronic survey addressing 3 domains: demographics of clinics, current opinions/practices related to TOC processes, and perceived barriers to successful TOC., Results: Fifteen surveys were sent with 11 returned (response rate = 73%). TOC practices varied among clinics surveyed. Fifty-five percent of clinics had a structured transition program, but only one transitioned 100% of their patients to adult providers by 22 years of age. Only one clinic had an absolute upper age limit for seeing patients, and 36% of clinics accepted new patients older than 21 years. No respondent was "completely satisfied" with their transition process, and only one respondent was "moderately satisfied." The majority of respondents felt the ideal care setting for adults with CP was a comprehensive, multidisciplinary adult-focused clinic in an adult hospital/clinic with primarily adult providers. They noted the top 3 perceived barriers to successful TOC were limited adult providers willing to accept CP patients, concern about the level of care in the adult health care system, and lack of financial resources., Conclusion: Current TOC practices vary considerably among multidisciplinary pediatric CP clinics and are not satisfactory to individual physician providers within these clinics. Respondents desired a multidisciplinary clinic in an adult care setting with adult providers; however, the top 3 perceived barriers involved the adult health care system, making it difficult for pediatric providers to develop effective TOC programs., Level of Evidence: Not applicable., (Copyright © 2017 American Academy of Physical Medicine and Rehabilitation. Published by Elsevier Inc. All rights reserved.)
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- 2017
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18. Support for the stability of the Alberta Infant Motor Scale after 'back-to-sleep'.
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Vargus-Adams J
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- Female, Humans, Male, Child Development, Infant Behavior, Motor Skills
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- 2014
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19. Population pharmacokinetics of oral baclofen in pediatric patients with cerebral palsy.
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He Y, Brunstrom-Hernandez JE, Thio LL, Lackey S, Gaebler-Spira D, Kuroda MM, Stashinko E, Hoon AH Jr, Vargus-Adams J, Stevenson RD, Lowenhaupt S, McLaughlin JF, Christensen A, Dosa NP, Butler M, Schwabe A, Lopez C, Roge D, Kennedy D, Tilton A, Krach LE, Lewandowski A, Dai H, Gaedigk A, Leeder JS, and Jusko WJ
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- Absorption, Administration, Oral, Adolescent, Baclofen blood, Baclofen therapeutic use, Body Weight, Cerebral Palsy blood, Child, Child, Preschool, Dose-Response Relationship, Drug, Drug Administration Schedule, Female, Half-Life, Humans, Male, Metabolic Clearance Rate, Models, Statistical, Multivariate Analysis, Muscle Relaxants, Central blood, Muscle Relaxants, Central therapeutic use, Baclofen pharmacokinetics, Cerebral Palsy drug therapy, Muscle Relaxants, Central pharmacokinetics
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Objective: To characterize the population pharmacokinetics (PK) of oral baclofen and assess impact of patient-specific covariates in children with cerebral palsy (CP) in order to support its clinical use., Subjects Design: Children (2-17 years of age) with CP received a dose of titrated oral baclofen from 2.5 mg 3 times a day to a maximum tolerated dose of up to 20 mg 4 times a day. PK sampling followed titration of 10-12 weeks. Serial R- and S-baclofen plasma concentrations were measured for up to 16 hours in 49 subjects. Population PK modeling was performed using NONMEM 7.1 (ICON PLC; Ellicott City, Maryland)., Results: R- and S-baclofen showed identical concentration-time profiles. Both baclofen enantiomers exhibited linear and dose/kg-proportional PK, and no sex differences were observed. Average baclofen terminal half-life was 4.5 hours. A 2-compartment PK model with linear elimination and transit absorption steps adequately described concentration-time profiles of both baclofen enantiomers. The mean population estimate of apparent clearance/F was 0.273 L/h/kg with 33.4% inter-individual variability (IIV), and the apparent volume of distribution (Vss/F) was 1.16 L/kg with 43.9% IIV. Delayed absorption was expressed by a mean transit time of 0.389 hours with 83.7% IIV. Body weight, a possible genetic factor, and age were determinants of apparent clearance in these children., Conclusion: The PK of oral baclofen exhibited dose-proportionality and were adequately described by a 2-compartment model. Our population PK findings suggest that baclofen dosage can be based on body weight (2 mg/kg per day) and the current baclofen dose escalation strategy is appropriate in the treatment of children with CP older than 2 years of age., (Copyright © 2014 Elsevier Inc. All rights reserved.)
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- 2014
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20. Aberrant high-gamma oscillations in the somatosensory cortex of children with cerebral palsy: a meg study.
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Guo X, Xiang J, Mun-Bryce S, Bryce M, Huang S, Huo X, Wang Y, Rose D, Degrauw T, Gartner K, Song T, Schmit J, and Vargus-Adams J
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- Adolescent, Brain Mapping, Child, Electric Stimulation methods, Female, Fingers physiology, Humans, Male, Cerebral Palsy physiopathology, Magnetoencephalography methods, Oscillometry, Somatosensory Cortex physiopathology
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Objective: Our study is to investigate somatosensory dysfunction in children with spastic cerebral palsy (CP) using magnetoencephalography (MEG) and synthetic aperture magnetometry (SAM)., Methods: Six children with spastic CP and six age- and gender-matched typically developing children were studied using a 275-channel MEG system while their left and right index fingers were stimulated in random order. The latency and amplitude of somatosensory evoked magnetic fields were analyzed at sensor level. The patterns of high-gamma oscillations were investigated with SAM at source level., Results: In comparison to the children with typical development, the latency of the first response of somatosensory evoked magnetic fields (SEFs) in the children with spastic CP was significantly delayed (p<0.05). High-gamma oscillations were identified in the somatosensory cortex in both children with CP and typical developing children. Interestingly, children with spastic CP had significantly higher incidence of ipsilateral activation in the somatosensory cortex following right and left finger stimulation, compared to typically developing children (p=0.05)., Conclusion: The results suggest that children with spastic CP have a measurable delay of SEFs and high-gamma oscillations. The high rates of ipsilateral cortical activation imply the impairments of functional lateralization in the developing brain. This is the first MEG study to demonstrate abnormal high-gamma oscillations of somatosensory cortices representing the finger in children with spastic CP., (Copyright © 2011 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.)
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- 2012
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21. Parent stress and children with cerebral palsy.
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Vargus-Adams J
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- Child, Humans, Cerebral Palsy psychology, Parent-Child Relations, Parents psychology
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- 2011
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22. Homozygous mutation in SAMHD1 gene causes cerebral vasculopathy and early onset stroke.
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Xin B, Jones S, Puffenberger EG, Hinze C, Bright A, Tan H, Zhou A, Wu G, Vargus-Adams J, Agamanolis D, and Wang H
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- Adolescent, Adult, Age of Onset, Base Sequence, Cerebrovascular Disorders pathology, Child, Child, Preschool, DNA Mutational Analysis, Ethnicity genetics, Female, Humans, Infant, Infant, Newborn, Male, Pedigree, SAM Domain and HD Domain-Containing Protein 1, Stroke pathology, Young Adult, Cerebrovascular Circulation, Cerebrovascular Disorders genetics, Homozygote, Monomeric GTP-Binding Proteins genetics, Mutation, Stroke genetics
- Abstract
We describe an autosomal recessive condition characterized with cerebral vasculopathy and early onset of stroke in 14 individuals in Old Order Amish. The phenotype of the condition was highly heterogeneous, ranging from severe developmental disability to normal schooling. Cerebral vasculopathy was a major hallmark of the condition with a common theme of multifocal stenoses and aneurysms in large arteries, accompanied by chronic ischemic changes, moyamoya morphology, and evidence of prior acute infarction and hemorrhage. Early signs of the disease included mild intrauterine growth restriction, infantile hypotonia, and irritability, followed by failure to thrive and short stature. Acrocyanosis, Raynaud's phenomenon, chilblain lesions, low-pitch hoarse voice, glaucoma, migraine headache, and arthritis were frequently observed. The early onset or recurrence of strokes secondary to cerebral vasculopathy seems to always be associated with poor outcomes. The elevated erythrocyte sedimentation rate (ESR), IgG, neopterin, and TNF-α found in these patients suggested an immune disorder. Through genomewide homozygosity mapping, we localized the disease gene to chromosome (Chr) 20q11.22-q12. Candidate gene sequencing identified a homozygous mutation, c.1411-2A > G, in the SAMHD1 gene, being associated with this condition. The mutation appeared at the splice-acceptor site of intron 12, resulted in the skipping of exon 13, and gave rise to an aberrant protein with in-frame deletion of 31 amino acids. Immunoblotting analysis showed lack of mutant SAMHD1 protein expression in affected cell lines. The function of SAMHD1 remains unclear, but the inflammatory vasculopathies of the brain found in the patients with SAMHD1 mutation indicate its important roles in immunoregulation and cerebral vascular hemeostasis.
- Published
- 2011
- Full Text
- View/download PDF
23. Pharmacologic treatment of spasticity in children.
- Author
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Tilton A, Vargus-Adams J, and Delgado MR
- Subjects
- Age Factors, Baclofen administration & dosage, Baclofen therapeutic use, Botulinum Toxins therapeutic use, Child, Clonidine analogs & derivatives, Clonidine therapeutic use, Dantrolene therapeutic use, Diazepam therapeutic use, Humans, Injections, Spinal, Muscle Spasticity diagnosis, Neuromuscular Agents administration & dosage, Muscle Spasticity drug therapy, Neuromuscular Agents therapeutic use
- Abstract
Many clinicians frequently face the dilemma of whether and how to medically treat spasticity. When pharmacologic intervention is deemed appropriate, treatment decisions must first be based on accurate assessment using valid and reliable clinical instruments, and, importantly, specific, measurable, achievable, and realistic treatment goals should be delineated. For the treatment of localized or segmental spasticity, botulinum toxin (BoNT-A) is recommended as an effective and generally safe treatment. For more generalized spasticity, a number of useful oral agents and intrathecal baclofen are available, each with their positive and negative attributes. Fundamental knowledge of pharmacologic properties and toxicities of these medications is required for safe and appropriate use. To achieve optimum results, spasticity treatment should be part of an integrated therapeutic approach in which patients, caregivers, therapists, physicians, and surgeons have an open and clear communication about the overall rehabilitation process of the patient. This review summarizes the current pharmacologic approaches to spasticity treatment in children, critically evaluating published studies in the context of established evidence-based criteria., (Copyright © 2010 Elsevier Inc. All rights reserved.)
- Published
- 2010
- Full Text
- View/download PDF
24. Outcome measures used in studies of botulinum toxin in childhood cerebral palsy: a systematic review.
- Author
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Baird MW and Vargus-Adams J
- Subjects
- Anti-Dyskinesia Agents therapeutic use, Child, Humans, Outcome Assessment, Health Care, Treatment Outcome, Botulinum Toxins therapeutic use, Cerebral Palsy drug therapy
- Abstract
This literature review uses the International Classification of Functioning, Disability and Health to describe the quality and variety of the studies of botulinum toxin in children with cerebral palsy since 2001. Articles were identified via electronic query and then reviewed for strength of evidence and classification of outcome measures. The distribution of levels of evidence for the 63 articles was I (n = 8), II (n = 12), III (n = 21), and IV (n = 22). One or more measurements were used in the International Classification of Functioning, Disability and Health domains of body structure and function (47 papers), activity (47 papers), participation (12 papers), and environmental/personal factors (10 papers). A total of 67% of all outcomes demonstrated a significant difference (I 49%, II 39%, III 74%, IV 88%). This review illustrates that few studies provide a high level of evidence and that outcomes focus on arenas such as spasticity or range of motion rather than activity or participation domains such as walking.
- Published
- 2010
- Full Text
- View/download PDF
25. Understanding function and other outcomes in cerebral palsy.
- Author
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Vargus-Adams J
- Subjects
- Disability Evaluation, Humans, Cerebral Palsy therapy, Clinical Trials as Topic methods, Outcome Assessment, Health Care methods
- Abstract
Describing the status of children with cerebral palsy (CP) and quantifying change in their status are 2 central challenges to research and clinical management of CP. The science of assessing and reporting status is outcome measurement, and it is rapidly developing in the arena of CP. Because of the large number of domains to measure, the variability of CP manifestations, and a limited number of "gold standard" evaluations, creating an accurate, comprehensive, responsive, and broadly applicable measurement strategy is a serious endeavor. A range of outcome measures are available to address CP issues across the spectrum of disability. The use of these measures, and others yet to be developed, provides researchers and clinicians the best means of understanding CP and the effects of treatments.
- Published
- 2009
- Full Text
- View/download PDF
26. Quality of life for adolescents with cerebral palsy: perspectives of adolescents and parents.
- Author
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Vargus-Adams J
- Subjects
- Adolescent, Humans, Cerebral Palsy psychology, Parent-Child Relations, Quality of Life
- Published
- 2009
- Full Text
- View/download PDF
27. Health-related quality of life in childhood cerebral palsy.
- Author
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Vargus-Adams J
- Subjects
- Adolescent, Chi-Square Distribution, Child, Child, Preschool, Female, Health Status, Humans, Male, Regression Analysis, Surveys and Questionnaires, Cerebral Palsy physiopathology, Cerebral Palsy psychology, Quality of Life
- Abstract
Objective: To describe the health-related quality of life (HRQOL) of a cross-section of children with cerebral palsy (CP) using the Child Health Questionnaire., Design: Survey of the parents or guardians of children with CP., Setting: Outpatient clinics at a tertiary care children's hospital., Participants: Children (N=177; 98 boys, 79 girls; mean age +/- standard deviation [SD], 8.6+/-4.2y) with CP were enrolled as a convenience sample. Study subjects were stratified by severity of CP using the Gross Motor Function Classification System., Interventions: Not applicable., Main Outcome Measure: HRQOL scores, as determined by the Child Health Questionnaire-Parent Form 50., Results: Parents reported reduced HRQOL for their children with CP. In the physical function domain, most children scored over 3 SDs below a normative sample, and scores in parental impact domains were at least 1 SD below the normative sample. Psychosocial domains were less impaired. Most of the effects on physical and parent impact domains were greater in children with more severe CP, although they were significant in most strata of severity., Conclusions: Children with CP have reduced HRQOL and the degree to which it is reduced is related to the severity of their CP.
- Published
- 2005
- Full Text
- View/download PDF
28. Oral motor dysfunction in nephropathic cystinosis.
- Author
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Vargus-Adams J
- Subjects
- Adult, Humans, Male, Cystinosis etiology, Cystinosis physiopathology, Kidney Diseases complications, Kidney Tubules, Mouth physiopathology, Movement
- Published
- 2002
- Full Text
- View/download PDF
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