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Koebke P, Schafmeyer L, Lentzen B, Keller T, Schoenau E, Duran I. Development of bone mineral density and content in children with cerebral palsy: a retrospective, longitudinal study. J Pediatr Endocrinol Metab 2025; 38:374-382. [PMID: 39882784 DOI: 10.1515/jpem-2024-0519] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/27/2024] [Accepted: 01/16/2025] [Indexed: 01/31/2025]
Abstract
OBJECTIVES The aim of this study was to investigate long-term bone development in children with cerebral palsy (CP) using longitudinal measurements of total body less head bone mineral content (TBLH-BMC) and bone mineral density (TBLH-BMD). METHODS A retrospective longitudinal analysis was performed on 109 children with CP who participated in a rehabilitation programme from 2006 to 2018. Dual-energy X-ray absorptiometry (DXA) scans were performed at the beginning of the programme and repeated as clinically indicated. The study used SITAR growth curve analysis and the LMS method within the GAMLSS model to generate developmental trajectories for TBLH-BMC and TBLH-BMD. RESULTS The findings suggest that the expected developmental trajectories of TBLH-BMC and TBLH-BMD in children with CP approximately follow the third percentile of healthy children. The median annual increase in bone mineral density was similar between GMFCS I-II and III-V groups at 5.12 and 5.79 %, respectively. Girls with CP reached age at peak velocity (APV) earlier than boys. The intensive exercise programme may have contributed to greater annual bone growth in children with more severe CP. CONCLUSIONS Children with CP have bone growth trajectories close to the third percentile of their healthy peers, suggesting that age-adjusted z-scores for TBLH-BMC and BMD remain relatively stable throughout childhood and adolescence. Early and targeted interventions, including improving muscle strength and mobility and optimising nutritional and hormonal status, are essential to promote better bone health and quality of life in children with CP.
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Affiliation(s)
- Philipp Koebke
- Center of Prevention and Rehabilitation, UniReha, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
| | - Leonie Schafmeyer
- Department of Neuropediatrics, University Children's Hospital Klinikum Oldenburg, Oldenburg, Germany
| | - Bruno Lentzen
- Center of Prevention and Rehabilitation, UniReha, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
| | - Titus Keller
- Department of Pediatrics, Cologne, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
| | - Eckhard Schoenau
- Center of Prevention and Rehabilitation, UniReha, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
- Department of Pediatrics, Cologne, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
| | - Ibrahim Duran
- Center of Prevention and Rehabilitation, UniReha, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
- Department of Pediatrics, Cologne, University of Cologne, Medical Faculty and University Hospital, Cologne, Germany
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Herron MS, Wang L, von Bartheld CS. Prevalence and Types of Strabismus in Cerebral Palsy: A Global and Historical Perspective Based on a Systematic Review and Meta-Analysis. Ophthalmic Epidemiol 2025; 32:125-142. [PMID: 38635869 PMCID: PMC11486841 DOI: 10.1080/09286586.2024.2331537] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2024] [Revised: 02/25/2024] [Accepted: 03/08/2024] [Indexed: 04/20/2024]
Abstract
PURPOSE Strabismus is more frequent in cerebral palsy (CP) than in the normal population, but reports differ how much it is increased. We here examined the global prevalence and types of strabismus in CP, whether esotropia or exotropia is more frequent, and whether the prevalence differs between ethnicities and/or country income levels, and between generations. METHODS We compiled in a systematic review and meta-analysis the results of 147 CP studies that report the prevalence of strabismus or the ratio of esotropia to exotropia, and we conducted subgroup analyses for region (income level) and ethnicity. We performed a pooled analysis for the CP strabismus prevalence, and estimated the global number of CP cases with strabismus. RESULTS The pooled prevalence of strabismus in CP is 49.8% in high-income countries and 39.8% in lower-income countries. We estimate the global number of strabismus cases in CP as 12.2 million, with 7.6 million males and 4.6 million females, based on current estimates of 29.6 million global CP cases. Esotropia is more frequent than exotropia in Caucasians, while exotropia is more frequent than esotropia in Hispanic and in some Asian and African populations. The strabismus prevalence in CP increases with increasing country income levels. CONCLUSION Generational changes in strabismus prevalence appear to reflect a transition of CP types and an increase in prevalence as countries attain higher income and more effective maternal health care. The distribution of esotropia and exotropia in CP patients largely reflects the horizontal strabismus type that is predominant in the subject's ethnicity.
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Affiliation(s)
- Michael S. Herron
- Center of Biomedical Research Excellence in Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
| | - Lingchen Wang
- School of Public Health, University of Nevada, Reno, Nevada, USA
| | - Christopher S. von Bartheld
- Center of Biomedical Research Excellence in Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
- Department of Physiology and Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
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Chang E, Chen YR. Communication challenges and use of communication apps among individuals with cerebral palsy. Disabil Rehabil Assist Technol 2025:1-7. [PMID: 40111936 DOI: 10.1080/17483107.2025.2481425] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/23/2024] [Revised: 02/20/2025] [Accepted: 03/14/2025] [Indexed: 03/22/2025]
Abstract
PURPOSE Verbal communication is the most immediate way of expressing oneself and the fastest way to make a first impression on others. However, for individuals with cerebral palsy (CP), which is generally accompanied by speech impediments, verbal communication can present a major hurdle to social interactions. MATERIALS AND METHODS In this study, 10 individuals with CP (four men and six women) and speech impediments were surveyed using questionnaires and semistructured interviews to explore the following four points: (1) communication difficulties in life, (2) communication methods and related problems, (3) application of existing AAC methods, and (4) predicaments using existing communication technologies. RESULTS The study found that individuals with CP and speech impediments speak slowly and generally choose to avoid conversations or engage passively, relying on written words, simplified phrases, and physical gestures to compensate for their language impediments. Family education and educational attainment influence their communication strategies. CONCLUSION Following advancements in technology, individuals with CP have become more reliant on text communications via phone apps such as Line and Facebook Messenger, and use text and stickers to express themselves. Despite the many problems with the usability of assistive devices and tech products for individuals with CP, reducing the number of operational steps and time constraints can improve the convenience of these products for individuals with CP.
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Affiliation(s)
- Eva Chang
- Program in Interdisciplinary Studies, National Sun Yat-sen University, Kaohsiung, Taiwan
| | - Yi-Ran Chen
- The Department of Social Welfare, National Chung Cheng University, Chiayi, Taiwan
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Smigelski GD. Cerebral palsy: Current concepts and a glimpse into the future. JAAPA 2025; 38:30-35. [PMID: 39903097 DOI: 10.1097/01.jaa.0000000000000178] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2025]
Abstract
ABSTRACT Cerebral palsy (CP) is an encompassing term for a group of permanent neurologic disorders of movement and posture commonly seen in early childhood and caused by abnormal brain development early in life. The cause of CP is unclear in the majority of patients affected and can be multifactorial, occurring in various time frames such as prenatal, intrapartum, and postpartum. Because CP is a clinical diagnosis, clinicians must have a strong understanding of the clinical presentation to ensure early diagnosis and improved outcomes for patients. This article reviews CP and describes advances in diagnosis and management; it also provides an overview of new treatments that are under investigation.
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Affiliation(s)
- Garrett D Smigelski
- At the time this article was written, Garrett D. Smigelski practiced in trauma medicine at the University of Michigan Health-Sparrow in Lansing, Mich. He now is a medical sciences liaison in the pharmaceutical industry. The author has disclosed no potential conflicts of interest, financial or otherwise
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Walters R, Seary C, Beare B, Stevenson VL. Functional electrical stimulation for walking in adults with cerebral palsy: a service evaluation. J Neuroeng Rehabil 2025; 22:41. [PMID: 40022119 PMCID: PMC11869474 DOI: 10.1186/s12984-024-01538-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/28/2024] [Accepted: 12/20/2024] [Indexed: 03/03/2025] Open
Abstract
Cerebral palsy (CP) is a neuromotor disorder which can lead to walking difficulties. Functional electrical stimulation (FES) is approved by The National Institute for Health and Care Excellence (NICE) for managing foot drop in upper motor neuron disorders, however there is limited evidence for its use in CP. We discuss a cohort of 26 patients with CP using FES for a 3 month period and longitudinal data for a subset of 11 patients that have used FES for at least 4 years. Patients were referred for the following common barriers to walking: reported falls (54%), foot drop (46%) and tripping (15%). After application of FES at baseline, there was a small clinically insignificant orthotic effect on walking speed (0.01 m/s on/off difference). However, orthotic effects became statistically and clinically significant at three months of continuous use (0.12 m/s on/off difference, p = 0.01) and in the subset of 11 patients this remained significant at four years (0.24 m/s on/off difference, p = 0.01). Patient reported walking satisfaction (numerical rating scale) improved when comparing no-FES versus FES at three months and at four years. FES is a safe, cost-effective treatment option and should be considered, for adults with CP who have walking difficulties.
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Affiliation(s)
- Rebecca Walters
- National Hospital for Neurology and Neurosurgery, University College London Hospitals NHS Foundation Trust, London, WC1N 3BG, UK.
| | - Coralie Seary
- National Hospital for Neurology and Neurosurgery, University College London Hospitals NHS Foundation Trust, London, WC1N 3BG, UK
| | - Ben Beare
- College of Health and Life Sciences, Brunel University London, London, UK
| | - Valerie L Stevenson
- National Hospital for Neurology and Neurosurgery, University College London Hospitals NHS Foundation Trust, London, WC1N 3BG, UK
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Huang YY, Cheng YY, Chen HY, Fu RH, Chang YJ, Yang TH. Chinese herbal medicine for the treatment of children with cerebral palsy: a meta-analysis of randomized controlled trials with core herbs exploration. Front Pharmacol 2025; 16:1500095. [PMID: 40078275 PMCID: PMC11897310 DOI: 10.3389/fphar.2025.1500095] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/22/2024] [Accepted: 01/02/2025] [Indexed: 03/14/2025] Open
Abstract
Introduction Chinese herbal medicine (CHM) taken orally is frequently utilized to enhance functional ability and independence in cerebral palsy (CP); nonetheless, there is a lack of current evidence regarding the efficacy of oral CHM in treating CP. Additionally, the general complexities of CHM prescriptions often obscure the underlying mechanisms. Our study aims to assess the efficacy of oral CHM in treating CP, a meta-analysis will be conducted on randomized clinical trials (RCTs). Materials and methods We searched Cochrane Library, PubMed, Embase, Scopus, PubMed Central, ClinicalTrials.gov, and China National Knowledge Infrastructure (CNKI), from 1990 to 2022. The primary outcome was the improvement in Effectiveness rate (ER). The secondary outcome was the improvement of motor function (GMFM). Subgroup analysis and trial sequential analysis (TSA) were conducted to confirm results consistency. Core CHMs were investigated through system pharmacology analysis. Results Seventeen RCTs were analyzed, in which CHMs with Standard treatment (ST) were compared to ST alone. All participants were aged <11 years. More participants in the CHM group achieved prominent improvement in ER (RR: 1.21, 95% CI: 1.13-1.30, p-value < 0.001, I2 = 32%) and higher GMFM improvement (SMD: 1.49; 95% CI: 1.33-1.65, p-value < 0.001, I2 = 92%). TSA also showed similar results with proper statistical power. Core CHMs, such as Glycyrrhiza uralensis Fisch. Ex DC., Poria cocos (Schw.) Wolf, Paeonia lactiflora Pall., processed Rehmannia glutinosa (Gaertn.) DC., Astragalus mongholicus Bunge, and Angelica sinensis (Oliv.) Diels, exerted effects on immune modulation and metabolism systems. The subgroup analysis showed participants using core CHMs or longer CHM treatment duration, and studies enrolling CP with spastic or mixed type, or mild-to-moderate severity had better outcomes in CHM groups with less heterogeneity. Conclusion CHMs may have a positive impact on managing pediatric CP; however, the potential bias in study design should be improved. Systematic Review Registration Identifier CRD42023424754.
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Affiliation(s)
- Ying-Yu Huang
- Division of Chinese Internal and Pediatric Medicine, Center for Traditional Chinese Medicine, Chang Gung Memorial Hospital, Taipei, Taiwan
| | - Ya-Yun Cheng
- Division of Chinese Acupuncture and Traumatology, Center of Traditional Chinese Medicine, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Hsing-Yu Chen
- Division of Chinese Internal and Pediatric Medicine, Center for Traditional Chinese Medicine, Chang Gung Memorial Hospital, Taipei, Taiwan
- School of Traditional Chinese Medicine, College of Medicine, Chang Gung University, Taoyuan, Taiwan
| | - Ren-Huei Fu
- Department of Pediatrics and Neonatology, Chang Gung Memorial Hospital, Linkou, Taiwan
| | - Yi-Jung Chang
- Department of Pediatrics, Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Taoyuan, Taiwan
| | - Tsung-Hsien Yang
- Division of Chinese Internal and Pediatric Medicine, Center for Traditional Chinese Medicine, Chang Gung Memorial Hospital, Taipei, Taiwan
- School of Traditional Chinese Medicine, College of Medicine, Chang Gung University, Taoyuan, Taiwan
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Long Y, Jiang X, Li J, Liu B. Effects of mind-body exercise in children with cerebral palsy-A systematic review and meta-analysis. Complement Ther Clin Pract 2025; 58:101930. [PMID: 39536700 DOI: 10.1016/j.ctcp.2024.101930] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2024] [Revised: 10/24/2024] [Accepted: 11/06/2024] [Indexed: 11/16/2024]
Abstract
BACKGROUND AND PURPOSE An increasing number of studies have investigated the efficacy of mind-body exercise in the treatment of children with cerebral palsy (CP). However, the findings of these researches have produced inconsistent results. The objective of this study was to evaluate the effects of mind-body exercise on children with CP in order to provide more conclusive results. MATERIALS AND METHODS A comprehensive search was conducted in PubMed, Cochrane, Embase, Web of Science, and Ovid databases from inception to May 2024. One researcher extracted the data, including first author, study type, country, year of publication, population, age, sample size, intervention details and outcomes, accuracy was verified by a second researcher. The included studies were analyzed using Review Manager 5.4 (RevMan 5.4) and StataNow/MP 18.5 software, with publication bias examined using funnel plots, and result stability assessed through sensitivity analysis. RESULTS A total of six eligible studies, comprising 173 patients, were included in the meta-analysis. The results of the meta-analysis indicated that mind-body exercise can improve balance function in children with CP (SMD = 0.48, P = 0.006). There was no significant difference in the effect of mind-body exercise on walking function in children with CP (SMD = -0.23, P = 0.58). CONCLUSION Compared with routine care and rehabilitation, mind-body exercise may provide a safe and convenient approach to improving balance functions in children with CP, but showed no statistically significant beneficial effects on walking in children with CP. PROSPERO REGISTRATION NUMBER CRD42024534257.
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Affiliation(s)
- Ye Long
- Children Health Care and Rehabilitation Center, Loudi Central Hospital, Loudi, China.
| | - Xinping Jiang
- Children Health Care and Rehabilitation Center, Loudi Central Hospital, Loudi, China.
| | - Juan Li
- Loudi Vocational and Technical College, Loudi, China.
| | - Bingxue Liu
- Children Health Care and Rehabilitation Center, Loudi Central Hospital, Loudi, China.
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Jiang Y, Liu G, Deng B, Li X, Ren J, Zhao Y, Hu C, Xu L, Gao F, Mu X. White matter lesions and DTI metrics related to various types of dysfunction in cerebral palsy: A meta-analysis and systematic review. PLoS One 2025; 20:e0312378. [PMID: 39854387 PMCID: PMC11760009 DOI: 10.1371/journal.pone.0312378] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/07/2024] [Accepted: 10/04/2024] [Indexed: 01/26/2025] Open
Abstract
BACKGROUND Assessing various types of dysfunction in cerebral palsy is a key factor in the treatment and rehabilitation of patients. The objective of this study was to use meta-analysis and systematic review to identify the specific white matter lesions and DTI metrics strongly associated with various types of dysfunction in cerebral palsy. METHODS We conducted a literature search of PubMed, Embase, Cochrane Library and Web of Science databases to identify trials published that had evaluated the correlation between DTI metrics in sensorimotor pathways and function scores in cerebral palsy. Correlation coefficient (r) values were extracted for each study, and the extent of r was quantitatively explored. The remaining part of the study was analyzed qualitatively. RESULTS 46 studies involving 1458 children with cerebral palsy, were included. 19 articles for Meta-analysis and 27 articles were descriptively analyzed. DTI metrics such as FA、MD in both sensory and motor pathways significantly correlated with various function ratings. In overall motor dysfunction, compared with the CST and PTR, FA of the PLIC correlated more strongly with GMFCS, and showed no significant heterogeneity (r = -1.28, confidence interval [CI]-1.70 to -0.87,I2 = 38.2%,P = 0.233). In upper limb dysfunction, compared with the AHA and MA2, FA of the CST correlated more strongly with BBT, and showed no significant heterogeneity (r = -0.56, confidence interval [CI]-0.78 to -0.34,I2 = 0.0%,P = 0.511). Lower limb dysfunction and other dysfunctions we used qualitative analysis. The qualitative analysis offered a concise overview of each investigation. CONCLUSIONS This study basically identifies the specific white matter lesions corresponding to overall motor dysfunction, upper limb and lower limb motor deficits and other dysfunctions in patients with cerebral palsy, as well as the associated DTI metrics.
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Affiliation(s)
- Yu Jiang
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Gang Liu
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Bowen Deng
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Xiaoye Li
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Jingpei Ren
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Yi Zhao
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Chuanyu Hu
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Lin Xu
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Feng Gao
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
| | - Xiaohong Mu
- Dongzhimen Hospital, Beijing University of Chinese Medicine, Beijing, China
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Xue Z, Zhang W, Zhou N, Ma P, Yuan K, Zheng P, Li J, Chang J. Effects of virtual reality motor games on motor skills in children with cerebral palsy: a systematic review and meta-analysis. Front Psychol 2025; 15:1483370. [PMID: 39881687 PMCID: PMC11776641 DOI: 10.3389/fpsyg.2024.1483370] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/22/2024] [Accepted: 12/10/2024] [Indexed: 01/31/2025] Open
Abstract
Background Enhancing motor skills is crucial for the functional development of children with cerebral palsy. Virtual reality has emerged as a promising technology for rehabilitating these children. Objective The objective of this study was to systematically evaluate the effects of virtual reality motor games on the gross and fine motor skills of children with cerebral palsy. Methods A comprehensive search was conducted in databases including PubMed, ProQuest, EBSCOhost, Web of Science, and Wanfang Data, covering publications from their inception to June 1, 2024, to identify randomized controlled trials (RCTs) investigating the effects of virtual reality motor games on the gross and fine motor skills of children with cerebral palsy. The quality of the included studies was assessed using the PEDro scale, and data were analyzed with RevMan software (version 5.4). Results Nineteen studies involving 850 children with cerebral palsy were included. The results indicated that virtual reality motor games significantly improved gross motor skills [mean difference (MD) 1.67, 95% confidence interval (CI): 0.75-2.56; P < 0.001] and fine motor skills [standardized mean difference (SMD) 0.73, 95% CI: 0.30-1.16; P = 0.00008] in children with cerebral palsy. Conclusions Virtual reality motor games have the potential to significantly enhance both gross and fine motor skills in children with cerebral palsy. Systematic review registration https://www.crd.york.ac.uk/prospero/, PROSPERO [CRD42024558713].
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Affiliation(s)
- Zhuolin Xue
- School of Physical Education, Southwest University, Chongqing, China
- Institute of Motor Quotient, School of Physical Education, Southwest University, Chongqing, China
| | - Weiqiang Zhang
- Department of Physical Education, Central South University, Changsha, China
| | - Ni Zhou
- School of Physical Education, Southwest University, Chongqing, China
| | - Pengwei Ma
- School of Physical Education, Southwest University, Chongqing, China
- Institute of Motor Quotient, School of Physical Education, Southwest University, Chongqing, China
| | - Kun Yuan
- School of Physical Education, Southwest University, Chongqing, China
- Institute of Motor Quotient, School of Physical Education, Southwest University, Chongqing, China
| | - Peiyun Zheng
- School of Physical Education, Southwest University, Chongqing, China
- Institute of Motor Quotient, School of Physical Education, Southwest University, Chongqing, China
| | - Junfeng Li
- Ministry of Sports, Shandong Technology and Business University, Yantai, China
| | - Jindong Chang
- School of Physical Education, Southwest University, Chongqing, China
- Institute of Motor Quotient, School of Physical Education, Southwest University, Chongqing, China
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Katori S, Himuro N, Kitai Y, Tanabe R, Ohnishi H. Cross-cultural adaptation, validity and reliability of the Japanese version of ABILHAND-kids for children with cerebral palsy using Rasch measurement model. Disabil Rehabil 2025; 47:213-221. [PMID: 38591266 DOI: 10.1080/09638288.2024.2338201] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/02/2023] [Revised: 03/25/2024] [Accepted: 03/28/2024] [Indexed: 04/10/2024]
Abstract
PURPOSE The aim of this study was to develop the Japanese version of the ABILHAND-Kids and to examine its psychometric properties for Japanese children with cerebral palsy (CP). METHODS The experimental version of 75 items was developed using forward-backward translation method. Parents of 137 children with CP answered it. Their responses were analyzed to successive items, and psychometric properties of the final version were investigated through the Rasch measurement model. RESULTS The Japanese version of the ABILHAND-Kids contained 22 items. It showed valid item-patient targeting, no significant floor and ceiling effects, and no differential item functioning for demographic and clinical subgroups. All items contributed to the definition of one-dimensional measure. For internal consistency, the person separation index was 0.94. For test-retest reliability, the intraclass correlation coefficients were 0.96 (95% CI: 0.92-0.98). The minimal detectable difference was calculated with a logit score of 0.79 and a total raw score of 4.50. The logit score showed a strong correlation with the Manual Ability Classification System level (ρ= -0.70) and the Gross Motor Function Classification System level (ρ= -0.62). CONCLUSIONS The Japanese version of the ABILHAND-Kids was found to be valid and reliable. It appears to be a good tool for assessing manual abilities in daily activities in children with CP.
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Affiliation(s)
- Sayaka Katori
- Department of Pediatric Rehabilitation, Hokkaido Medical Center for Child Health and Rehabilitation, Sapporo, Japan
| | - Nobuaki Himuro
- Department of Public Health, School of Medicine, Sapporo Medical University, Sapporo, Japan
| | - Yukihiro Kitai
- Department of Pediatric Neurology, Bobath Memorial Hospital, Osaka, Japan
| | - Ryo Tanabe
- Department of Pediatric Neurology, Chiba Rehabilitation Center, Chiba, Japan
| | - Hirofumi Ohnishi
- Department of Public Health, School of Medicine, Sapporo Medical University, Sapporo, Japan
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Zhuo H, Ritz B, Warren JL, Pollitt KG, Liew Z. Ambient Toxic Air Contaminants in the Maternal Residential Area during Pregnancy and Cerebral Palsy in the Offspring. ENVIRONMENTAL HEALTH PERSPECTIVES 2025; 133:17008. [PMID: 39853265 PMCID: PMC11758984 DOI: 10.1289/ehp14742] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/31/2024] [Revised: 11/02/2024] [Accepted: 12/19/2024] [Indexed: 01/26/2025]
Abstract
BACKGROUND Cerebral palsy (CP) is the most common permanent neuromotor disorder diagnosed in childhood. Although most cases have unknown etiology, emerging evidence suggests environmental risk factors of CP. OBJECTIVES We investigated whether ambient toxic air contaminants (TACs) in the maternal residential area during pregnancy, specifically volatile organic compounds (VOCs) and metals, were associated with offspring CP risk in California. METHODS We conducted a case-cohort study that included CP cases (N = 906 ) and a 20% random sample of all live singleton births (N = 184,954 ) who lived within a 5 -mile (8-km) radius of air toxics monitoring stations in California during 2005-2015 as the control comparison group. CP cases were ascertained from diagnostic records of the California Department of Developmental Services. We a priori selected TACs with suspected neurotoxicity and developmental toxicity, including 14 VOCs and 6 metals. We estimated the adjusted risk ratio (RR) and 95% confidence interval (CI) for CP and the average maternal residential exposures to each TAC over the entire pregnancy using modified Poisson regression. For air contaminant mixtures, we used quantile-based g-computation to estimate the effects of mixtures of VOCs or metals. Finally, we performed a negative control exposure analysis on exposure estimates of 36-48 months after delivery to evaluate uncontrolled confounding bias. RESULTS Maternal residential exposures to six VOCs (benzene, toluene, 1,3-butadiene, acetone, acetonitrile, and methylene chloride) and four metals (antimony, lead, nickel, and vanadium) were associated with 3%-25% higher risk of CP per interquartile range increase, and the estimated mixture effects of VOCs (RR = 1.24 ; 95% CI: 1.08, 1.43) or metals (RR = 1.38 ; 95% CI: 1.20, 1.58) were stronger. The observed associations were close to null for negative control exposures (36-48 months after delivery) to mixtures of VOCs or metals and CP. DISCUSSION In California, maternal prenatal residential exposure to VOCs and metals in the outdoor air, largely attributed to mobile traffic emission sources, was associated with an increased risk of CP in offspring. https://doi.org/10.1289/EHP14742.
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Affiliation(s)
- Haoran Zhuo
- Department of Environmental Health Sciences, Yale School of Public Health, New Haven, Connecticut, USA
- Yale Center for Perinatal, Pediatric, and Environmental Epidemiology, Yale School of Public Health, New Haven, Connecticut, USA
| | - Beate Ritz
- Department of Epidemiology, Fielding School of Public Health, University of California, Los Angeles (UCLA), Los Angeles, California, USA
- Department of Neurology, School of Medicine, UCLA, Los Angeles, California, USA
| | - Joshua L. Warren
- Department of Biostatistics, Yale School of Public Health, New Haven, Connecticut, USA
| | - Krystal Godri Pollitt
- Department of Environmental Health Sciences, Yale School of Public Health, New Haven, Connecticut, USA
| | - Zeyan Liew
- Department of Environmental Health Sciences, Yale School of Public Health, New Haven, Connecticut, USA
- Yale Center for Perinatal, Pediatric, and Environmental Epidemiology, Yale School of Public Health, New Haven, Connecticut, USA
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Sanderlin R, Schluger C, Wu J, Eusebio FA, Roberts AL, Prosser L. Modeling the distribution and progression of motor ability among children with cerebral palsy: An analysis of three reference centile sets. Heliyon 2024; 10:e40615. [PMID: 39660208 PMCID: PMC11629233 DOI: 10.1016/j.heliyon.2024.e40615] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/04/2024] [Revised: 10/25/2024] [Accepted: 11/20/2024] [Indexed: 12/12/2024] Open
Abstract
Background Reference centiles describing gross motor function in children with cerebral palsy (CP) are used in clinical and research settings to guide treatments and evaluate interventions. However, it is unknown how existing references generalize to populations in novel settings. Aims The aim of this study is to evaluate the cross-sectional and longitudinal performance of three reference centiles to describe the motor function of children with CP aged 2-12 years at a large urban US pediatric hospital through a retrospective observational study. Methods and Procedures: We assessed cross-sectional performance by ranking our clinical population by quartile distributions described by the references. We assessed longitudinal performance by analyzing the distribution of prediction errors and correlations between predictions and observed scores. Outcomes and Results For cross-sectional distribution, the reference centiles by Hanna more closely described our population than those by Duran. For longitudinal progression, prediction error was less than 6 GMFM points for most records at 24-, 12-, and 6-month time scales for all three sets of reference centiles, but higher at a 48-month time scale. Prediction errors increased at younger ages and higher motor ability. Conclusions and implications Despite differences in cross-sectional performance, all three reference centiles achieved similar longitudinal performance and are sufficient for most clinical and research uses. Caution should be used when applying these curves to locations with different standards of care.
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Affiliation(s)
- Rachel Sanderlin
- Department of Biomedical and Health Informatics, Children's Hospital of Philadelphia, Philadelphia, USA
| | - Charlotte Schluger
- Department of Biomedical and Health Informatics, Children's Hospital of Philadelphia, Philadelphia, USA
| | - Joe Wu
- Department of Biomedical and Health Informatics, Children's Hospital of Philadelphia, Philadelphia, USA
| | - Francis Ang Eusebio
- Division of Rehabilitation Medicine, Children's Hospital of Philadelphia, Philadelphia, USA
| | - Amy L. Roberts
- Department of Biomedical and Health Informatics, Children's Hospital of Philadelphia, Philadelphia, USA
| | - Laura Prosser
- Division of Rehabilitation Medicine, Children's Hospital of Philadelphia, Philadelphia, USA
- Department of Pediatrics, University of Pennsylvania, Philadelphia, USA
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Butera CD, Yeh A, Biniwale M, Bloch E, Craddock D, Doyle M, Iyer SN, Kretch KS, Liu N, Mirzaian CB, Sargent B, Solano P, Swaine M, Dusing SC. Development and Initial Outcomes of the Interdisciplinary 'Early Identification and Intervention for Infants Network' (Ei3) in Los Angeles. J Clin Med 2024; 13:7442. [PMID: 39685900 DOI: 10.3390/jcm13237442] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/30/2024] [Revised: 11/26/2024] [Accepted: 11/28/2024] [Indexed: 12/18/2024] Open
Abstract
Background/Objectives: The Early Identification and Intervention for Infants (Ei3) Network is an interdisciplinary team dedicated to improving early detection and intervention of cerebral palsy (CP) in California. This paper describes the key (1) awareness-building and (2) capacity-building strategies utilized by the Ei3 Network in the first two years. Methods: Awareness-building methods included interactive conference discussions, resource deliverable creation, and the creation of a framework for dissemination. Capacity-building methods were hosting assessment training, gathering stakeholder feedback, and implementation training. All deliverables were created with a minimum of 3 review and revision cycles. We planned, hosted, and provided scholarships for training, including the Hammersmith Infant Neurological Examination (HINE), Prechtl's GMA, and an implementation conference. Preliminary descriptive statistics and paired samples t-tests were performed on HINE training surveys. Results: Seven resource deliverables were created and distributed. A professional website, @steps2home.org, was launched. Online channels gained followers (146, Instagram; 198, X; 298, Mailchimp). Providers participated in various trainings: the Sykes Symposium on early detection and intervention (70), HINE (211), GMA Basic or Advanced (46), and Implementation Conference (six facilities from April 2023 to June 2024). HINE training participants reported increased ability to identify CP (p = 0.001), knowledge of (p = 0.004) early detection, ability to implement early detection guidelines (p < 0.001), and confidence in performing the HINE on the post-test (79.87%). Conclusions: The first two years of Ei3 resulted in increased awareness of the early detection guidelines through resources to help providers and families navigate complex care systems and dissemination through online and in-person strategies. We built capacity by training an increased number of providers to identify infants at risk of CP and implement the early detection guidelines. Engagement of stakeholders in focus groups, reviewing documents, and including a parent panel throughout the process increased the value of this work and should support the expansion of the network in the next year.
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Affiliation(s)
- Christiana D Butera
- Division of Biokinesiology and Physical Therapy, Herman Ostrow School of Dentistry, University of Southern California, Los Angeles, CA 90033, USA
| | - Amy Yeh
- Los Angeles General Medical Center, Los Angeles, CA 90033, USA
- Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
| | - Manoj Biniwale
- Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
- Cedars-Sinai Medical Center, Los Angeles, CA 90048, USA
| | - Edward Bloch
- Los Angeles County Department of Public Health, El Monte, CA 91731, USA
| | - Debi Craddock
- Los Angeles County California Children's Services, El Monte, CA 91731, USA
| | - Mary Doyle
- Los Angeles County California Children's Services, El Monte, CA 91731, USA
| | - Sai N Iyer
- Department of Pediatrics, University of California Los Angeles, Los Angeles, CA 90095, USA
| | - Kari S Kretch
- Division of Biokinesiology and Physical Therapy, Herman Ostrow School of Dentistry, University of Southern California, Los Angeles, CA 90033, USA
| | - Nora Liu
- Los Angeles County Department of Public Health, El Monte, CA 91731, USA
| | - Christine B Mirzaian
- Keck School of Medicine, University of Southern California, Los Angeles, CA 90033, USA
- Children's Hospital Los Angeles, Los Angeles, CA 90027, USA
| | - Barbara Sargent
- Division of Biokinesiology and Physical Therapy, Herman Ostrow School of Dentistry, University of Southern California, Los Angeles, CA 90033, USA
| | | | - Margaret Swaine
- North Los Angeles County Regional Center, Chatsworth, CA 91311, USA
| | - Stacey C Dusing
- Division of Biokinesiology and Physical Therapy, Herman Ostrow School of Dentistry, University of Southern California, Los Angeles, CA 90033, USA
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14
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Jang DH, Kim J, Schwabe AL, Lotze TE. Genetics of Cerebral Palsy: Diagnosis, Differential Diagnosis, and Beyond. Ann Rehabil Med 2024; 48:369-376. [PMID: 39736497 DOI: 10.5535/arm.240081] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/11/2024] [Accepted: 11/15/2024] [Indexed: 01/01/2025] Open
Abstract
Cerebral palsy (CP) is the most common motor disability in children, characterized by diverse clinical manifestations and often uncertain etiology, which has spurred increasing interest in genetic diagnostics. This review synthesizes findings from various studies to enhance understanding of CP's genetic underpinnings. The discussion is structured around five key areas: monogenic causes and copy number variants directly linked to CP, differential genetic disorders including atypical CP and mimics, ambiguous genetic influences, co-occurrence with other neurodevelopmental disorders, and polygenic risk factors. Case studies illustrate the clinical application of these genetic insights, underscoring the complexity of diagnosing CP due to the phenotypic overlap with other conditions and the potential for misdiagnosis. The review highlights the significant role of advanced genetic testing in distinguishing CP from similar neurodevelopmental disorders and assessing cases with unclear clinical presentations. Furthermore, it addresses the ongoing challenges in establishing a consensus on genetic contributors to CP, the need for comprehensive patient phenotyping, and the integration of rigorous genetic and functional studies to validate findings. This comprehensive examination of CP genetics aims to pave the way for more precise diagnostics and personalized treatment plans, urging continued research to overcome the current limitations and refine diagnostic criteria within this field.
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Affiliation(s)
- Dae-Hyun Jang
- Department of Rehabilitation Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
- Medical Genetics/Rare Disease Center, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Jaewon Kim
- Department of Rehabilitation Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
- Medical Genetics/Rare Disease Center, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Aloysia Leisanne Schwabe
- Department of Physical Medicine and Rehabilitation, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States
| | - Timothy Edward Lotze
- Division of Neurology and Developmental Neuroscience, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States
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15
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Sandran NG, Badawi N, Gecz J, van Eyk CL. Cerebral palsy as a childhood-onset neurological disorder caused by both genetic and environmental factors. Semin Fetal Neonatal Med 2024; 29:101551. [PMID: 39523172 DOI: 10.1016/j.siny.2024.101551] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2024]
Abstract
Cerebral palsy (CP) is a clinical term used to describe a spectrum of movement and posture disorders resulting from non-progressive disturbances in the developing fetal brain. The clinical diagnosis of CP does not include pathological or aetiological defining features, therefore both genetic and environmental causal pathways are encompassed under the CP diagnostic umbrella. In this review, we explore several genetic causal pathways, including both monogenic and polygenic risks, and present evidence supporting the multifactorial contributions to CP. Historically, CP has been associated with various risk factors such as pre-term birth, multiple gestation, intrauterine growth restriction (IUGR), maternal infection, and perinatal asphyxia. Thus, we also examine genetic predispositions that may contribute to these risk factors. Understanding the specific aetiology of CP enables more tailored treatments, especially with the increasing potential for early genetic testing.
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Affiliation(s)
- Nandini G Sandran
- Neurogenetics Research Program, Adelaide Medical School, University of Adelaide, Adelaide, Australia; Australian Collaborative Cerebral Palsy Research Group, Robinson Research Institute, University of Adelaide, Adelaide, Australia
| | - Nadia Badawi
- Children's Hospital Westmead Clinical School, University of Sydney, Sydney, Australia; Grace Centre for Newborn Intensive Care, The Children's Hospital Westmead, Sydney, Australia; Discipline of Child and Adolescent Health, Cerebral Palsy Alliance Research Institute, University of Sydney, Sydney, Australia
| | - Jozef Gecz
- Neurogenetics Research Program, Adelaide Medical School, University of Adelaide, Adelaide, Australia; Australian Collaborative Cerebral Palsy Research Group, Robinson Research Institute, University of Adelaide, Adelaide, Australia; South Australian Health and Medical Research Institute, Adelaide, Australia.
| | - Clare L van Eyk
- Neurogenetics Research Program, Adelaide Medical School, University of Adelaide, Adelaide, Australia; Australian Collaborative Cerebral Palsy Research Group, Robinson Research Institute, University of Adelaide, Adelaide, Australia
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Patel DR, Bovid KM, Rausch R, Ergun-Longmire B, Goetting M, Merrick J. Cerebral palsy in children: A clinical practice review. Curr Probl Pediatr Adolesc Health Care 2024; 54:101673. [PMID: 39168782 DOI: 10.1016/j.cppeds.2024.101673] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 08/23/2024]
Abstract
Cerebral palsy is a disorder characterized by abnormal tone, posture, and movement. In clinical practice, it is often useful to approach cerebral palsy based on the predominant motor system findings - spastic hemiplegia, spastic diplegia, spastic quadriplegia, extrapyramidal or dyskinetic, and ataxic. The prevalence of cerebral palsy is between 1.5 and 3 per 1,000 live births with higher percentage of cases in low to middle income countries and geographic regions. Pre-term birth and low birthweight are recognized as the most frequent risk factors for cerebral palsy; other risk factors include hypoxic-ischemic encephalopathy, maternal infections, and multiple gestation. In most cases of cerebral palsy, the initial injury to the brain occurs during early fetal brain development. Intracerebral hemorrhage and periventricular leukomalacia are the main pathologic findings found in preterm infants who develop spastic cerebral palsy. The diagnosis of cerebral palsy is primarily based on clinical findings. Early recognition of infants at risk for cerebral palsy as well as those with cerebral palsy is possible based on a combination of clinical history, use of standardized neuromotor assessment and findings on magnetic resonance imaging; however, in clinical practice, cerebral palsy is more reliably diagnosed by 2 years of age. Magnetic resonance imaging scan is indicated to delineate the extent of brain lesions and to identify congenital brain malformations. Genetic testing and tests for inborn errors of metabolism are indicated to identify specific disorders, especially treatable disorders. Because cerebral palsy is associated with multiple associated and secondary medical conditions, its management requires a sustained and consistent collaboration among multiple disciplines and specialties. With appropriate support, most children with cerebral palsy grow up to be adults with good functional abilities.
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Affiliation(s)
- Dilip R Patel
- Neurodevelopmental Disabilities. Department of Pediatric and Adolescent Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States.
| | - Karen M Bovid
- Neurodevelopmental Disabilities. Department of Pediatric and Adolescent Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States; Department of Orthopedic Surgery and Department of Pediatric and Adolescent Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States
| | - Rebecca Rausch
- Neurodevelopmental Disabilities. Department of Pediatric and Adolescent Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States
| | - Berrin Ergun-Longmire
- Neurodevelopmental Disabilities. Department of Pediatric and Adolescent Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States
| | - Mark Goetting
- Department of Pediatric and Adolescent Medicine, Department of Medicine, Western Michigan University Homer Stryker MD School of Medicine, United States
| | - Joav Merrick
- National Institute of Child Health and Human Development, Israel; Professor of Pediatrics, Division of Pediatrics, Hadassah Hebrew University Medical Center, Kentucky; Children's Hospital, University of Kentucky, Lexington, United States; Professor of Public Health, Center for Healthy Development, School of Public Health, Georgia State University, Atlanta, United States
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Khuc THH, Karim T, Nguyen VAT, Giang NTH, Dũng TQ, Dossetor R, Cao Minh C, Van Bang N, Badawi N, Khandaker G, Elliott E. Associated impairments among children with cerebral palsy: findings from a cross-sectional hospital-based study in Vietnam. BMJ Open 2024; 14:e075820. [PMID: 39461866 PMCID: PMC11535665 DOI: 10.1136/bmjopen-2023-075820] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/19/2023] [Accepted: 08/06/2024] [Indexed: 10/29/2024] Open
Abstract
OBJECTIVE This study aims to explore the associated impairments of cerebral palsy (CP) and their correlates among children with CP in Vietnam. DESIGN Descriptive cross-sectional study using hospital-based surveillance. SETTING National Children's Hospital, Hanoi, Vietnam between June and November 2017. PARTICIPANTS 765 children with CP were recruited. OUTCOME MEASURES We assessed clinical characteristics of CP, associated impairments (epilepsy, intellectual, visual, hearing, speech impairments) and their correlates. We performed descriptive analyses (median, IQR and proportion). χ2 test and Fisher's exact test were used for categorical variables. Univariate logistic regression and multivariate logistic regression models were established and associated impairments were included as independent variables. RESULTS The median age of children was 1.7 years (IQR=2.7). Quadriplegia was the predominant subtype (69.5%) and 46.5% were at Gross Motor Function Classification System level IV-V. Of children, 76.3% had ≥one associated impairment, most commonly speech or intellectual impairments (59.1% and 57.8%, respectively). Severity of motor impairment, type of CP, maternal and perinatal factors (eg, gestational age, perinatal asphyxia, timing of injury causing CP) were associated with greater risk of associated impairments. CONCLUSION Children with CP have a high burden of associated impairments. Findings from our study will inform the development and implementation of appropriate screening and interventions and reduce the long-term adverse effects of these impairments on individuals with CP and their socioeconomic impact.
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Affiliation(s)
- Thi Hong Hanh Khuc
- Faculty of Medical Technology, Phenikaa University, Hanoi, Vietnam
- School of Health, Medical and Applied Sciences, Central Queensland University, Rockhampton, Queensland, Australia
| | - Tasneem Karim
- Cerebral Palsy Alliance, Sydney, New South Wales, Australia
- CSF Global, Dhaka, Bangladesh
- Asian Institute of Disability and Development (AIDD), University of South Asia, Dhaka, Bangladesh
- Specialty of Child and Adolescent Health, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
| | - Van Anh Thi Nguyen
- Medical Education and Skills Laboratory, Hanoi Medical University, Hanoi, Vietnam
| | | | | | - Rachael Dossetor
- Specialty of Child and Adolescent Health, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
| | - Chau Cao Minh
- Faculty of Medical Technology, Phenikaa University, Hanoi, Vietnam
| | - Nguyen Van Bang
- Pediatric Department, Medical Faculty, Phenikaa University, Hanoi, Vietnam
| | - Nadia Badawi
- Cerebral Palsy Alliance, Sydney, New South Wales, Australia
- Specialty of Child and Adolescent Health, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
- Grace Centre for Newborn Intensive Care, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia
| | - Gulam Khandaker
- Specialty of Child and Adolescent Health, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
- Central Queensland Public Health Unit, Central Queensland Hospital and Health Service, Rockhampton, Queensland, Australia
| | - Elizabeth Elliott
- Specialty of Child and Adolescent Health, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
- Kid’s Research, The Sydney Children’s Hospitals Network (Westmead), Sydney, New South Wales, Australia
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18
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Chen T, Wu Y, Zhong M, Xu K. Short- medium- and long-term effects of botulinum toxin on upper limb spasticity in children with cerebral palsy: A meta-analysis of randomized controlled trials. Ann Phys Rehabil Med 2024; 67:101869. [PMID: 39181066 DOI: 10.1016/j.rehab.2024.101869] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/17/2022] [Revised: 05/08/2024] [Accepted: 05/17/2024] [Indexed: 08/27/2024]
Abstract
BACKGROUND Botulinum toxin (BTX) is an effective management method for spasticity in children with cerebral palsy (CP), but the short- medium- and long-term effects remain unclear. OBJECTIVE The primary objective was to quantify the effects of BTX injections on upper limb spasticity over time in children with CP. The secondary objective was to evaluate efficacy according to the International Classification of Functioning, Disability, and Health-Children & Youth version framework. METHODS We conducted a systematic review and meta-analysis of randomized controlled trials that included control/comparison groups treated with a placebo or other treatments. We searched CINAHL, Embase, PubMed, Scopus, Web of Science, and PsycINFO from their inception to April 2024. The pooled mean difference (MD) or standard mean difference (SMD) with 95 % CI was calculated using a random effects model at the short-term (up to 3 months), medium-term (3 to 6 months), and long-term (over 6 months). RESULTS A total of 658 children with CP aged 1.8 to 19 years old in 12 eligible trials were involved. The primary outcome of the Melbourne Assessment percentile showed a significant increase in the medium- (MD = 2.63, 95 % CI 0.22 to 5.04, I² = 0 %) and long-term (MD = 4.72, 95 % CI 0.93 to 8.51, I² = 0 %) in favor of BTX. Pooled effects also showed that BTX significantly improved Modified Ashworth Scale scores in the short- (MD = -0.44, 95 % CI -0.88 to -0.01, I² = 88 %) and medium-term (MD = -0.20, 95 % CI -0.28 to -0.13, I² = 0 %), and individual goals and bimanual performance up to 6-months. No significantly higher risk of adverse events was observed with BTX. CONCLUSIONS AND IMPLICATIONS BTX injections sustainably improved the quality of affected upper limb function and temporarily improved individual goals and bimanual performance in children with CP. Our findings cautiously support a time interval of 3 to 6 months between BTX injections in the upper limbs of children with CP. TRIAL REGISTRATION This study was registered in the International Prospective Register of Systematic Reviews (PROSPERO) (Registration ID: CRD42022323672).
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Affiliation(s)
- Tingting Chen
- Department of Rehabilitation, Guangzhou Women and Children's Medical Centre, Guangzhou Medical University, No. 318 Renmin Middle Road, Guangzhou, 510120, China; Guangdong Provincial Clinical Research Center for Child Health, No. 9 Jinsui Road, Guangzhou, 510623, China
| | - Yin Wu
- Department of Rehabilitation, Guangzhou Women and Children's Medical Centre, Guangzhou Medical University, No. 318 Renmin Middle Road, Guangzhou, 510120, China; Guangdong Provincial Clinical Research Center for Child Health, No. 9 Jinsui Road, Guangzhou, 510623, China
| | - Mengru Zhong
- Department of Rehabilitation, Guangzhou Women and Children's Medical Centre, Guangzhou Medical University, No. 318 Renmin Middle Road, Guangzhou, 510120, China; Guangdong Provincial Clinical Research Center for Child Health, No. 9 Jinsui Road, Guangzhou, 510623, China
| | - Kaishou Xu
- Department of Rehabilitation, Guangzhou Women and Children's Medical Centre, Guangzhou Medical University, No. 318 Renmin Middle Road, Guangzhou, 510120, China; Guangdong Provincial Clinical Research Center for Child Health, No. 9 Jinsui Road, Guangzhou, 510623, China.
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Rathke CL, Pimentel VCDA, Alsina PJ, do Espírito Santo CC, Dantas AFODA. IoT-Based Wireless System for Gait Kinetics Monitoring in Multi-Device Therapeutic Interventions. SENSORS (BASEL, SWITZERLAND) 2024; 24:5799. [PMID: 39275710 PMCID: PMC11398167 DOI: 10.3390/s24175799] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/25/2024] [Revised: 08/24/2024] [Accepted: 09/04/2024] [Indexed: 09/16/2024]
Abstract
This study presents an IoT-based gait analysis system employing insole pressure sensors to assess gait kinetics. The system integrates piezoresistive sensors within a left foot insole, with data acquisition managed using an ESP32 board that communicates via Wi-Fi through an MQTT IoT framework. In this initial protocol study, we conducted a comparative analysis using the Zeno system, supported by PKMAS as the gold standard, to explore the correlation and agreement of data obtained from the insole system. Four volunteers (two males and two females, aged 24-28, without gait disorders) participated by walking along a 10 m Zeno system path, equipped with pressure sensors, while wearing the insole system. Vertical ground reaction force (vGRF) data were collected over four gait cycles. The preliminary results indicated a strong positive correlation (r = 0.87) between the insole and the reference system measurements. A Bland-Altman analysis further demonstrated a mean difference of approximately (0.011) between the two systems, suggesting a minimal yet significant bias. These findings suggest that piezoresistive sensors may offer a promising and cost-effective solution for gait disorder assessment and monitoring. However, operational factors such as high temperatures and sensor placement within the footwear can introduce noise or unwanted signal activation. The communication framework proved functional and reliable during this protocol, with plans for future expansion to multi-device applications. It is important to note that additional validation studies with larger sample sizes are required to confirm the system's reliability and robustness for clinical and research applications.
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Affiliation(s)
- Christian Lang Rathke
- Graduate Program in Neuroengineering, Edmond and Lily Safra International Institute of Neuroscience, Macaíba 59280-000, RN, Brazil
| | - Victor Costa de Andrade Pimentel
- Graduate Program in Electrical and Computer Engineering, Federal University of Rio Grande do Norte, Natal 59078-970, RN, Brazil
- Department of Mechatronics, Federal Institute of Science, Education, and Technology of Rio Grande do Norte, Parnamirim Campus, Parnamirim 59143-455, RN, Brazil
| | - Pablo Javier Alsina
- Graduate Program in Electrical and Computer Engineering, Federal University of Rio Grande do Norte, Natal 59078-970, RN, Brazil
| | - Caroline Cunha do Espírito Santo
- Graduate Program in Neuroengineering, Edmond and Lily Safra International Institute of Neuroscience, Macaíba 59280-000, RN, Brazil
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20
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Asher RC, Shrewsbury VA, Innes B, Fitzpatrick A, Simmonds S, Cross V, Rose A, Hinton E, Collins CE. Feasibility and acceptability of a culinary nutrition programme for adults with mild-to-moderate intellectual disability: FLIP Food and Lifestyle Information Programme. JOURNAL OF APPLIED RESEARCH IN INTELLECTUAL DISABILITIES 2024; 37:e13281. [PMID: 39104041 DOI: 10.1111/jar.13281] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/11/2023] [Revised: 07/05/2024] [Accepted: 07/09/2024] [Indexed: 08/07/2024]
Abstract
BACKGROUND Culinary nutrition education can support improved diet-related health and wellbeing. This pre-post pilot study aimed to assess feasibility and acceptability of an eight-session culinary nutrition programme, the Food and Lifestyle Information Programme (FLIP), for adults with mild-to-moderate intellectual disability. A secondary aim was to evaluate preliminary programme effectiveness. METHOD Participants were recruited through a disability service provider. Feasibility measures were: recruitment and retention; implementation; engagement and participation; adverse outcomes; and feasibility of outcome measures. Acceptability was assessed using an interactive process evaluation. Effectiveness measures included cooking frequency, cooking and food skill confidence and diet quality. RESULTS Six of eight participants completed the intervention with high attendance and programme engagement. FLIP was well received by participants and support workers. No adverse outcomes occurred. Diet quality was feasible to assess. CONCLUSIONS Findings can inform content, delivery and evaluation of future culinary nutrition programmes for adults with mild-to-moderate intellectual disability.
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Affiliation(s)
- R C Asher
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
- Food and Nutrition Research Program, Hunter Medical Research Institute, New Lambton Heights, Australia
| | - V A Shrewsbury
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
- Food and Nutrition Research Program, Hunter Medical Research Institute, New Lambton Heights, Australia
| | - B Innes
- Sports 4 All, Fern Bay, Australia
| | - A Fitzpatrick
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
| | - S Simmonds
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
| | - V Cross
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
| | - A Rose
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
| | - E Hinton
- Challenge Community Services, Hamilton, Australia
| | - C E Collins
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Callaghan, Australia
- Food and Nutrition Research Program, Hunter Medical Research Institute, New Lambton Heights, Australia
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Kumar Panda P, Moirangthem V, Tomar A, Neyaz O, Sharawat IK. Efficacy of Oral Trihexyphenidyl Plus Clonazepam Versus Trihexyphenidyl for the Treatment of Dystonia in Children With Dystonic Cerebral Palsy: An Open-Label Randomized Controlled Trial. Pediatr Neurol 2024; 158:35-40. [PMID: 38945037 DOI: 10.1016/j.pediatrneurol.2024.06.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/24/2023] [Revised: 05/13/2024] [Accepted: 06/09/2024] [Indexed: 07/02/2024]
Abstract
BACKGROUND Trihexyphenidyl and clonazepam are commonly used to treat dystonia in children with cerebral palsy (CP). However, there is a notable gap in the literature when it comes to studies that combine these first-line agents for the management of dystonia. METHODS This open-label, randomized controlled trial aimed to compare the efficacy of adding oral clonazepam to trihexyphenidyl (THP + CLZ) versus using trihexyphenidyl alone (THP) in reducing the severity of dystonia, as measured by the Barry-Albright Dystonia (BAD) score. The study was conducted over a 12-week therapy period in children with dystonic CP aged two to 14 years. RESULTS Each group enrolled 51 participants. The THP + CLZ group showed significantly better improvement in dystonia severity at 12 weeks compared with the THP group alone (-4.5 ± 2.9 vs -3.4 ± 1.7, P = 0.02). Furthermore, the THP + CLZ group exhibited superior improvement in the severity of choreoathetosis, upper limb function, pain perception by the child, and quality of life, with P values of 0.02, 0.009, 0.01, and 0.01, respectively. The number of participants experiencing treatment-emergent adverse events was comparable in both groups (P = 0.67). Importantly, none of the participants in any of the groups reported any serious adverse events. CONCLUSION A combination of oral THP + CLZ proves to be more efficacious than using THP alone for the treatment of dystonic CP in children aged two to 14 years in terms of reducing the severity of dystonia.
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Affiliation(s)
- Prateek Kumar Panda
- Pediatric Neurology Division, Department of Pediatrics, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Vetoni Moirangthem
- Pediatric Neurology Division, Department of Pediatrics, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Apurva Tomar
- Pediatric Neurology Division, Department of Pediatrics, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Osama Neyaz
- Department of Physical Medicine and Rehabilitation, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
| | - Indar Kumar Sharawat
- Pediatric Neurology Division, Department of Pediatrics, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India.
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22
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Rapuc S, Stres B, Verdenik I, Lučovnik M, Osredkar D. Uncovering early predictors of cerebral palsy through the application of machine learning: a case-control study. BMJ Paediatr Open 2024; 8:e002800. [PMID: 39214549 PMCID: PMC11367350 DOI: 10.1136/bmjpo-2024-002800] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/06/2024] [Accepted: 08/04/2024] [Indexed: 09/04/2024] Open
Abstract
OBJECTIVE Cerebral palsy (CP) is a group of neurological disorders with profound implications for children's development. The identification of perinatal risk factors for CP may lead to improved preventive and therapeutic strategies. This study aimed to identify the early predictors of CP using machine learning (ML). DESIGN This is a retrospective case-control study, using data from the two population-based databases, the Slovenian National Perinatal Information System and the Slovenian Registry of Cerebral Palsy. Multiple ML algorithms were evaluated to identify the best model for predicting CP. SETTING This is a population-based study of CP and control subjects born into one of Slovenia's 14 maternity wards. PARTICIPANTS A total of 382 CP cases, born between 2002 and 2017, were identified. Controls were selected at a control-to-case ratio of 3:1, with matched gestational age and birth multiplicity. CP cases with congenital anomalies (n=44) were excluded from the analysis. A total of 338 CP cases and 1014 controls were included in the study. EXPOSURE 135 variables relating to perinatal and maternal factors. MAIN OUTCOME MEASURES Receiver operating characteristic (ROC), sensitivity and specificity. RESULTS The stochastic gradient boosting ML model (271 cases and 812 controls) demonstrated the highest mean ROC value of 0.81 (mean sensitivity=0.46 and mean specificity=0.95). Using this model with the validation dataset (67 cases and 202 controls) resulted in an area under the ROC curve of 0.77 (mean sensitivity=0.27 and mean specificity=0.94). CONCLUSIONS Our final ML model using early perinatal factors could not reliably predict CP in our cohort. Future studies should evaluate models with additional factors, such as genetic and neuroimaging data.
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Affiliation(s)
- Sara Rapuc
- Department of Pediatric Neurology, University Children's Hospital, University Medical Centre Ljubljana, Ljubljana, Slovenia
| | - Blaž Stres
- Department of Catalysis and Chemical Reaction Engineering, National Institute of Chemistry, Ljubljana, Slovenia
- Institute of Sanitary Engineering, Faculty of Civil and Geodetic Engineering, University of Ljubljana, Ljubljana, Slovenia
| | - Ivan Verdenik
- Department of Perinatology, Division of Obstetrics and Gynecology, University Medical Centre Ljubljana, Ljubljana, Slovenia
| | - Miha Lučovnik
- Department of Perinatology, Division of Obstetrics and Gynecology, University Medical Centre Ljubljana, Ljubljana, Slovenia
- Medical Faculty, University of Ljubljana, Ljubljana, Slovenia
| | - Damjan Osredkar
- Department of Pediatric Neurology, University Children's Hospital, University Medical Centre Ljubljana, Ljubljana, Slovenia
- Medical Faculty, University of Ljubljana, Ljubljana, Slovenia
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Belisheva NK, Drogobuzhskaya SV. Rare Earth Element Content in Hair Samples of Children Living in the Vicinity of the Kola Peninsula Mining Site and Nervous System Diseases. BIOLOGY 2024; 13:626. [PMID: 39194565 DOI: 10.3390/biology13080626] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/11/2024] [Revised: 07/31/2024] [Accepted: 08/09/2024] [Indexed: 08/29/2024]
Abstract
The aim of this study is to assess the rare earth element (REE) content in hair samples of children living in Lovozero village, near an REE mining site, and the possible effects of REEs on the prevalence of nervous system diseases in Lovozersky District (Murmansk region, Kola Peninsula). Fifty-three school-age children were recruited for the analysis of REE content in hair samples. REE (Y, La, Ce, Pr, Nd, Sm, Eu, Gd, Tb, Dy, Ho, Er, Tm, Yb, and Lu) content was estimated by means of inductively coupled plasma mass spectrometry (ICP-MS). The analysis of REE content in the hair of children living in Russia, Kazakhstan, and China indicated REE intake from the environment. The possible contribution of REEs to nervous system disorders is supported by the link between the REE content in hair samples of children living near REE mining areas (China) and the manifestation of cognitive disorders in these children. It is also found that the prevalence of nervous system diseases in children aged 15-17 years is higher in Lovozersky District compared to the other districts of the Murmansk region. In this paper, the possible contribution of REEs to the prevalence of episodic paroxysmal disorders (G40-G47), cerebral palsy (G80-G83), and epilepsy and status epilepticus (G40-G41) is discussed.
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Affiliation(s)
- Natalia K Belisheva
- Research Centre for Human Adaptation in the Arctic, Federal Research Centre "Kola Science Centre of the Russian Academy of Sciences" (RCHAA KSC RAS), Akademgorodok, 41a, 184209 Apatity, Russia
| | - Svetlana V Drogobuzhskaya
- Tananaev Institute of Chemistry-Subdivision of the Federal Research Centre "Kola Science Centre of the Russian Academy of Sciences", Akademgorodok, 26 a, 184209 Apatity, Russia
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24
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Nova Díaz DM, Errea Rodríguez M, Cabasés Hita JM, Sánchez Iriso E. Self-perceived quality of life by institutionalised adults with cerebral palsy in Spain. GACETA SANITARIA 2024; 38:102416. [PMID: 39098171 DOI: 10.1016/j.gaceta.2024.102416] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 10/30/2023] [Revised: 04/22/2024] [Accepted: 06/16/2024] [Indexed: 08/06/2024]
Abstract
OBJECTIVE To assess the self-perceived quality of life of institutionalized adults with cerebral palsy and to identify factors that influence their well-being, including sociodemographic, clinical, and diagnostic characteristics, as well as the degree of institutionalization. METHOD A descriptive cross-sectional study was conducted using the San Martin Quality of Life Scale among adults with cerebral palsy. Data were collected in 2021 and 2022 in a sample of adults with cerebral palsy in Navarra, Spain. Multivariate regression was used to explore the relationship between quality of life and various influential factors. RESULTS The self-determination dimension positively influenced quality of life scores, whereas the social inclusion dimension had the opposite effect. Descriptive and regression analyses revealed that factors such as residing outside the city and a high degree of dependency had a negative influence on quality of life, while the degree of institutionalization had a positive impact. CONCLUSIONS It is important to highlight the positive effects of the degree of institutionalization on rehabilitation and well-being, as it seeks to enhance autonomy and social integration when talking about patient-centered models of institutionalization.
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Tillberg E, Persson JKE. Hemiplegic (unilateral) cerebral palsy in northern Stockholm: Intellectual disability and epilepsy. Seizure 2024; 120:110-115. [PMID: 38941801 DOI: 10.1016/j.seizure.2024.06.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/14/2023] [Revised: 06/11/2024] [Accepted: 06/12/2024] [Indexed: 06/30/2024] Open
Abstract
PURPOSE The purpose of this study was to describe intellectual disability and its association with epilepsy and brain imaging, in a population-based group of children with hemiplegic (unilateral) cerebral palsy, previously investigated and published in 2020. MATERIALS AND METHODS Forty-seven children of school age in northern Stockholm, fulfilling the Surveillance of Cerebral Palsy in Europe-criteria of hemiplegic (unilateral spastic) cerebral palsy, were invited to participate in the study. Twenty-one children consented to participate. A WISC (Wechsler Intelligence Scale for Children)-test was performed by an experienced psychologist. RESULTS In the study population of twenty-one children, 57 % (n 12) displayed uneven cognitive profiles, 38 % (n 8) intellectual disability and 62 % (n 13) had a normal IQ. 43 % (n 9) developed epilepsy. Children with extensive brain lesions had more severe intellectual disability. CONCLUSIONS In this study intellectual disability and/or epilepsy were associated with the type and extent of the underlying brain lesion. Intellectual disability and uneven cognitive profiles were common. We therefore recommend individual cognitive assessment to ensure an optimal school start.
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Affiliation(s)
- Elsa Tillberg
- Department of Clinical Neuroscience, Karolinska Institutet, Sweden.
| | - Jonas K E Persson
- Department of Clinical Neurophysiology, Karolinska University Hospital, Eugeniavägen 11 SE-171 76 Stockholm, Sweden
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Chrościńska-Kawczyk M, Zdolińska-Malinowska I, Boruczkowski D. The Impact of Umbilical Cord Mesenchymal Stem Cells on Motor Function in Children with Cerebral Palsy: Results of a Real-world, Compassionate use Study. Stem Cell Rev Rep 2024; 20:1636-1649. [PMID: 38877284 DOI: 10.1007/s12015-024-10742-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/27/2024] [Indexed: 06/16/2024]
Abstract
The aim of this study was to analyze the impact of human umbilical cord-derived MSCs (hUC-MSCs) on motor function in children with cerebral palsy (CP). The study enrolled 152 children with CP who received up to two courses of five hUC-MSCs injections. Children's motor functions were assessed with the Gross Motor Function Measure (GMFM), 6-Minute Walk Test (6-MWT), Timed Up and Go test (Up&Go test), and Lovett's test, and mental abilities were assessed with the Clinical Global Impression (CGI) scale. Data collected at visit 1 (baseline) and visit 5 (after four injections) were analyzed retrospectively. After four hUC-MSCs administrations, all evaluated parameters improved. The change in GMFM score, by a median of 1.9 points (IQR: 0.0-8.0), correlated with age. This change was observed in all GFMCS groups and was noticed in all assessed GMFM areas. A median increase of 75 m (IQR: 20.0-115.0) was noted on the 6-MWT, and this correlated with GMFM score change. Time on the Up&Go test was reduced by a median of 2 s (IQR: -3 to - 1) and the change correlated with age, GMFM score at baseline, and the difference observed on the 6-MWT. Results of Lovett's test indicated slight changes in muscle strength. According to the CGI, 75.5% (96/151) of children were seriously (level VI) or significantly ill (level V) at the 1st visit, with any improvement observed in 63.6% (96/151) of patients at the 5th visit, 23.8% (36/151) with improvement (level II) or great improvement (level I). In conclusion, the application of hUC-MSCs generally enhanced functional performance, but individual responses varied. The therapy also benefited children with high level of disability but not to the same extent as the initially less disabled children. Although younger patients responded better to the treatment, older children can also benefit. Trial Registration 152/2018/KB/VII and 119/2021/KB/VIII. Retrospective registration in ClinicalTrials: ongoing.
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Pekeles H, Husein N, Kirton A, Oskoui M, Fehlings DL, Dunbar M, Shevell MI. Association of Gestational Age at Birth and Changes on MRI With Prevalence and Spectrum of Comorbidities in Children With Cerebral Palsy. Neurology 2024; 103:e209571. [PMID: 38889390 DOI: 10.1212/wnl.0000000000209571] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2024] [Accepted: 04/15/2024] [Indexed: 06/20/2024] Open
Abstract
BACKGROUND AND OBJECTIVES For individuals with cerebral palsy (CP) and caregivers, comorbidities may be a greater challenge than neuromotor impairment. Clinicians may make assumptions regarding risk of comorbidities based simply on term vs preterm birth, but this has not been well examined. To better understand factors affecting comorbidity pattern, we investigated the relationship between gestational age (GA) and imaging pattern on the presence of specific comorbidities. METHODS This is a cross-sectional study of data extracted from the Canadian Cerebral Palsy Registry of children with CP. Multivariable analysis was used to evaluate the relationship between brain injury, GA, and comorbidities. Comorbidities included in the analysis were communication, cognitive, visual, and auditory impairment, seizures in the past year, and gavage feeding. Each comorbidity was assessed as a separate nonexclusive outcome, with GA, MRI pattern, birth weight, postneonatal insult, 5-minute Apgar score, and male sex considered as potential modifiers. RESULTS The only comorbidity affected by GA on multivariable analysis was seizures within the past year that were more prevalent in term children (odds ratio [OR] 1.1 95% CI 1.0-1.2) and was also affected by Apgar score (OR 0.9 95% CI 0.85-0.94), but not MRI pattern. MRI pattern appeared important for communication impairment (deep gray OR 4.2 95% CI 1.8-10.0; total brain injury OR 8.5, 95% CI 3.2-22.6; malformation OR 2.7, 95% CI 1.3-5.7) and cognitive impairment (deep gray OR 5.6, 95% CI 2.4-13.2; total brain injury OR 10.1, 95% CI 4.0-25.3; malformation OR 3.3, 95% CI 1.6-6.8; watershed OR 3.6, 95% CI 1.4-8.9). Focal injury compared with normal MRI was associated with reduced odds of visual impairment (OR 0.24, 95% CI 0.12-0.48), auditory impairment (OR 0.2195% CI 0.10-0.46) and communication impairment (OR 0.46, 95% CI 0.26-0.82), and overall number of comorbidities (coefficient -0.73, 95% CI -1.2 to -0.31). The number of comorbidities was increased by total brain injury pattern (coefficient 0.65, 95% CI 0.15-1.13) and reduced by focal brain injury (coefficient -0.73, 95% CI -1.2 to -0.31) and increasing 5-minute Apgar score (coefficient -0.11, 95% CI -0.16 to -0.07). DISCUSSION In those with brain injuries sufficient to cause CP, development of additional comorbidities is less affected by GA at birth and more related to the underlying cause of CP as reflected by MRI patterns.
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Affiliation(s)
- Heather Pekeles
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Nafisa Husein
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Adam Kirton
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Maryam Oskoui
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Darcy L Fehlings
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Mary Dunbar
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
| | - Michael I Shevell
- From the Department of Neurology & Neurosurgery (H.P., M.O., M.I.S.), McGill University; Canadian Cerebral Palsy Registry (N.H.), Research Institute of McGill University Health Centre, Montreal, Quebec; Departments of Pediatrics and Clinical Neurosciences (A.K., M.D.), University of Calgary, Alberta Children's Hospital Research Institute, University of Calgary; Department of Paediatrics (D.L.F.), Bloorview Research Institute, University of Toronto, Ontario; and Department of Pediatrics (M.I.S.), Montreal Children's Hospital-McGill University Health Center, Quebec, Canada
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Saranti A, Dragoumi P, Papavasiliou A, Zafeiriou D. Current approach to cerebral palsy. Eur J Paediatr Neurol 2024; 51:49-57. [PMID: 38824721 DOI: 10.1016/j.ejpn.2024.05.015] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/23/2024] [Revised: 05/26/2024] [Accepted: 05/28/2024] [Indexed: 06/04/2024]
Abstract
This teaching review aims to provide an overview of the current approach to children with cerebral palsy (CP), retrieving the best available evidence and summarizing existing knowledge in the field of CP in children. We also highlight areas where more research is needed and novel strategies for diagnosing and treating cerebral palsy. CP includes a group of permanent disorders of movement and posture that cause activity limitation. Multiple risk factors, occurring preconceptionally, prenatally, perinatally, or postneonatally, are involved in the pathogenesis of CP, with the prenatal ones accounting for 80-90 % of cases. Due to its heterogeneity, CP has various classifications, but usually is classified based on clinical findings and motor impairment. Standardized function classification systems have been developed to address inconsistencies in previous classifications. The combination of clinical assessment and validated predictive tools is recommended for an early diagnosis, which is important for early intervention and prevention of secondary impairments. The therapeutic regimen in CP involves prevention and management of the motor and associated problems. It includes the enhancement of motor performance, the enrichment of cognition and communication skills, the prevention of secondary impairments, and the support of parents and caregivers. The care of CP children demands a multidisciplinary approach focused on improving motor skills, reducing comorbidities, enhancing the quality of life, and prolonging survival.
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Affiliation(s)
- Anna Saranti
- 1th Department of Pediatrics, Aristotle University of Thessaloniki, G. Hippokration Hospital, Thessaloniki, Greece
| | - Pinelopi Dragoumi
- 1th Department of Pediatrics, Aristotle University of Thessaloniki, G. Hippokration Hospital, Thessaloniki, Greece
| | | | - Dimitrios Zafeiriou
- 1th Department of Pediatrics, Aristotle University of Thessaloniki, G. Hippokration Hospital, Thessaloniki, Greece.
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Hao J, Huang B, Remis A, He Z. The application of virtual reality to home-based rehabilitation for children and adolescents with cerebral palsy: A systematic review and meta-analysis. Physiother Theory Pract 2024; 40:1588-1608. [PMID: 36847396 DOI: 10.1080/09593985.2023.2184220] [Citation(s) in RCA: 3] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/25/2022] [Revised: 02/18/2023] [Accepted: 02/20/2023] [Indexed: 03/01/2023]
Abstract
BACKGROUND Home-based rehabilitation enables children and families to participate in therapeutic activities built into their daily routines without the barriers of arrangement and transportation to facilities. Virtual reality is an emerging technology which has shown promising outcomes in rehabilitation. PURPOSE This systematic review aims to examine the feasibility and effects of virtual reality-enhanced home rehabilitation on Body functions and structures, Activity, and Participation outcomes in children and adolescents with cerebral palsy. METHODS Interventional studies were searched across five biomedical databases on November 26, 2022. Two independent reviewers conducted study selection, data extraction, and quality assessment. The Physiotherapy Evidence Database scale and National Institutes of Health Study Quality Assessment Tools were used to evaluate the quality of included studies. Meta-analysis was performed to examine the effects of the intervention. RESULTS Eighteen studies were included in this review. Home-based virtual reality rehabilitation appears feasible with effects on upper extremity and gross motor function, strength, bone density, cognition, balance, walking, daily activity performance, and participation. Meta-analyses revealed significant improvements in hand function (SMD = 0.41, p= .003), gross motor function (SMD = 0.56, p= .0002), and walking capacity (SMD = 0.44, p= .01) following home-based virtual reality intervention. CONCLUSION Home-based virtual reality may serve as an adjunct to conventional facility-based therapy to promote participation in therapeutic exercises and maximize rehabilitation outcomes. Further properly designed randomized controlled trials using valid and reliable outcome measures with adequately powered sample sizes are warranted to enhance the current body of evidence using home-based virtual reality in cerebral palsy rehabilitation.
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Affiliation(s)
- Jie Hao
- Department of Health and Rehabilitation Sciences, College of Allied Health Professions, University of Nebraska Medical Center, Omaha, United States
| | - Biying Huang
- Department of Health and Rehabilitation Sciences, College of Allied Health Professions, University of Nebraska Medical Center, Omaha, United States
| | - Andréas Remis
- Department of Physical Medicine and Rehabilitation, Gate Parkway Primary Care Center, Jacksonville
| | - Zhengting He
- Department of Epidemiology, Bloomberg School of Public Health, Johns Hopkins University, Baltimore
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Micheletti S, Galli J, Vezzoli M, Scaglioni V, Agostini S, Calza S, Merabet LB, Fazzi E. Academic skills in children with cerebral palsy and specific learning disorders. Dev Med Child Neurol 2024; 66:778-792. [PMID: 37990438 DOI: 10.1111/dmcn.15808] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/29/2022] [Revised: 09/02/2023] [Accepted: 10/17/2023] [Indexed: 11/23/2023]
Abstract
AIM To investigate the prevalence and clinical manifestations of reading, writing, and mathematics disorders in children with cerebral palsy (CP). We explored how the clinical profile of these children differed from those with specific learning disorders (SLDs), taking into account several factors, particularly IQ scores, neuropsychological aspects, and the presence of a visual impairment. METHOD A prospective cross-sectional study was conducted in 42 children with CP (mean age 9 years 8 months; SD = 2 years 2 months) and 60 children with SLDs (mean age 10 years; SD = 1 year 7 months). Clinical characteristics, neuromotor and cognitive profiles, neuropsychological aspects (speech performance, academic skills, visual attention, phonological awareness, working memory), and signs of visual impairment (visual acuity, contrast sensitivity, visual field, oculomotor functions) were assessed. A machine learning approach consisting of a random forest algorithm, where the outcome was the diagnosis and the covariates were the clinical variables collected in the sample, was used for the analyses. RESULTS About 59% of the children with CP had reading, writing, or mathematics disorders. Children with CP with learning disorders had a low performance IQ, normal phonological awareness, and working memory difficulties, whereas children with SLDs had normal performance IQ, impaired phonological awareness, and mild working memory difficulties. There were no differences in verbal IQ between the two groups. INTERPRETATION Learning disorders are frequently associated with CP, with different clinical characteristics, compared with SLDs. Assessment of academic skills is mandatory in these children, even if the IQ is normal. At school age, specific interventions to promote academic skills in children with CP could be a major rehabilitative goal. WHAT THIS PAPER ADDS Reading, writing, and mathematics disorders in cerebral palsy have specific clinical characteristics. Their underlying mechanisms differ from those described in specific learning disorders. Working memory impairment can be considered a hallmark of learning disorders in children with cerebral palsy.
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Affiliation(s)
- Serena Micheletti
- Unit of Child Neurology and Psychiatry, ASST Spedali Civili of Brescia, Brescia, Italy
| | - Jessica Galli
- Unit of Child Neurology and Psychiatry, ASST Spedali Civili of Brescia, Brescia, Italy
- Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy
| | - Marika Vezzoli
- Unit of Biostatistics and Bioinformatics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy
| | - Vera Scaglioni
- Unit of Child Neurology and Psychiatry, ASST Spedali Civili of Brescia, Brescia, Italy
| | - Stefania Agostini
- Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy
| | - Stefano Calza
- Unit of Biostatistics and Bioinformatics, Department of Molecular and Translational Medicine, University of Brescia, Brescia, Italy
| | - Lotfi B Merabet
- Laboratory for Visual Neuroplasticity, Department of Ophthalmology, Massachusetts Eye and Ear, Harvard Medical School, Boston, MA, USA
| | - Elisa Fazzi
- Unit of Child Neurology and Psychiatry, ASST Spedali Civili of Brescia, Brescia, Italy
- Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy
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Zhang Y, Hui Z, Qi W, Zhang J, Wang M, Zhu D. Clinical study on the safety and feasibility of AiWalker-K for lower limbs exercise rehabilitation in children with cerebral palsy. PLoS One 2024; 19:e0303517. [PMID: 38776339 PMCID: PMC11111022 DOI: 10.1371/journal.pone.0303517] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/05/2023] [Accepted: 04/24/2024] [Indexed: 05/24/2024] Open
Abstract
BACKGROUND Robotic-assisted gait training (RAGT) devices are effective for children with cerebral palsy (CP). Many RAGT devices have been created and put into clinical rehabilitation treatment. Therefore, we aimed to investigate the safety and feasibility of a new RAGT for children with CP. METHODS This study is a cross-over design with 23 subjects randomly divided into two groups. The occurrence of adverse events and changes in heart rate and blood pressure were recorded during each AiWalker-K training. Additionally, Gross Motor Function Measure-88 (GMFM-88), Pediatric Balance Scale (PBS), 6 Minutes Walking Test (6MWT), Physiological Cost Index, and Edinburgh Visual Gait Score (EVGS) were used to assess treatment, period, carry-over, and follow-up effects in this study. RESULTS Adverse events included joint pain, skin pain, and injury. Heart rate and blood pressure were higher with the AiWalker-K compared to the rest (P < 0.05), but remained within safe ranges. After combined treatment with AiWalker-K and routine rehabilitation treatment, significant improvements in 6MWT, GMFM-88 D and E, PBS, and EVGS were observed compared to routine rehabilitation treatment alone (P < 0.05). CONCLUSIONS Under the guidance of experienced medical personnel, AiWalker-K can be used for rehabilitation in children with CP.
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Affiliation(s)
- Yi Zhang
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Zhichong Hui
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Weihang Qi
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Jiamei Zhang
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Mingmei Wang
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
| | - Dengna Zhu
- Department of Rehabilitation Medicine, the Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
- Henan Key Laboratory of Child Brain Injury and Henan Pediatric Clinical Research Center, Institute of Neuroscience and Third Affiliated Hospital of Zhengzhou University, Zhengzhou, China
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Kukka AJ, Bhattarai P, Sundelin HEK, Gurung R, Brown NJW, Litorp H, Axelin A, Kc A. 'We did everything by phone': a qualitative study of mothers' experience of smartphone-aided screening of cerebral palsy in Kathmandu, Nepal. BMC Pediatr 2024; 24:357. [PMID: 38778316 PMCID: PMC11110401 DOI: 10.1186/s12887-024-04829-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/23/2023] [Accepted: 05/10/2024] [Indexed: 05/25/2024] Open
Abstract
BACKGROUND International guidelines recommend early intervention to all children at risk of cerebral palsy, but targeted screening programs are often lacking in low- and middle-income settings with the highest burden of disease. Smartphone applications have the potential to improve access to early diagnostics by empowering parents to film their children at home followed by centralized evaluation of videos with General Movements Assessment. We explored mothers' perceptions about participating in a smartphone aided cerebral palsy screening program in Kathmandu, Nepal. METHODS This is an explorative qualitative study that used focus group discussions (n = 2) and individual interviews (n = 4) with mothers of term-born infants surviving birth asphyxia or neonatal seizures. Parents used the NeuroMotion™ smartphone app to film their children at home and the videos were analysed using Precthl's General Movements Assessment. Sekhon et al.'s framework on the acceptability of health care interventions guided the design of the group discussions and interviews, and the deductive qualitative content analysis. RESULTS Mothers were interested in engaging with the programme and expressed hope it would benefit their children. Most felt using the app was intuitive. They were, however, unclear about the way the analysis was performed. Support from the research team was often needed to overcome an initial lack of self-confidence in using the technology, and to reduce anxiety related to the follow-up. The intervention was overall perceived as recommendable but should be supplemented by a face-to-face consultation. CONCLUSION Smartphone aided remote screening of cerebral palsy is acceptable in a lower middle-income population but requires additional technical support.
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Affiliation(s)
- Antti J Kukka
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden.
- Department of Pediatrics, Gävle Regional Hospital, Gävle, Region Gävleborg, Sweden.
| | | | - Heléne E K Sundelin
- Department of Biomedical and Clinical Sciences, Division of Children's and Women´S Health, Linköping University, Linköping, Sweden
- Department of Women's and Children's Health, Neuropediatric Unit, Karolinska University Hospital, KarolinskaInstitutet, Stockholm, Sweden
| | - Rejina Gurung
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden
- Golden Community, Lalitpur, Nepal
| | - Nick J W Brown
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden
- Department of Pediatrics, Gävle Regional Hospital, Gävle, Region Gävleborg, Sweden
| | - Helena Litorp
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden
- Department of Global Public Health, Karolinska Institutet, Stockholm, Sweden
| | - Anna Axelin
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden
- Department of Nursing Science, University of Turku, Turku, Finland
| | - Ashish Kc
- Department of Women's and Children's Health, Uppsala University, Uppsala, SE-751 85, Sweden
- School of Public Health and Community Medicine, University of Gothenburg, Gothenburg, Sweden
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Kang X, Huang Y, Zheng Y, Zhang Q, Gong R, Tan J, Ma L, Chen S, Lv X, Shi S. Meta-analysis of the efficacy of Jingjin acupuncture therapy in the treatment of spastic cerebral palsy. Front Neurol 2024; 15:1358732. [PMID: 38784910 PMCID: PMC11111905 DOI: 10.3389/fneur.2024.1358732] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/15/2024] [Accepted: 04/17/2024] [Indexed: 05/25/2024] Open
Abstract
Background This study aimed to systematically evaluate the clinical efficacy of Jingjin (muscle region of the meridian, sinew/tendon/fascia) acupuncture therapy for the treatment of spastic cerebral palsy. Methods Computer searches of the Cochrane Library, Web of Science, PubMed, Embase, Chinese Biomedical Literature (CBM) Database, Wanfang database, Wipu (VIP) database, and China National Knowledge Infrastructure (CNKI) database for published randomized controlled trial (RCT) studies on Jingjin acupuncture treatment of cerebral palsy from the beginning of the database construction until 30 November 2023 were performed, and the quality of the papers was assessed through independent data extraction by two individuals and then meta-analyzed using RevMan5.4 software. A total of 20 RCTs involving 1,453 patients were included. Results The overall effective rate of Jingjin acupuncture therapy was better than that of conventional therapy, with a combined odds ratio (OR) of 4.70 and a 95% confidence interval (CI) of [3.05, 7.24]. The Modified Ashworth Spasticity (MAS) Scale, Gross Motor Function Measure (GMFM), Fine Motor Function Measure (FMFM), and Comprehensive Spasticity Scale (CSS) scores are superior to conventional therapy. Conclusion Jingjin acupuncture therapy is effective in treating spastic cerebral palsy and has better overall efficacy than conventional therapy. Due to the low quality of some of the literature in this study type, more high-quality, well-designed clinical studies are needed to validate it.
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Affiliation(s)
- Xingyu Kang
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Ying Huang
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Yi Zheng
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Qian Zhang
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Rui Gong
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Jinlang Tan
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Le Ma
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Siyu Chen
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Xueyan Lv
- Graduate School, Heilongjiang University of Chinese Medicine, Harbin, China
| | - Shuai Shi
- Rehabilitation Department, The Second Affiliated Hospital of Heilongjiang University of Chinese Medicine, Harbin, Heilongjiang, China
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Gouveia HJCB, Manhães-de-Castro R, Costa-de-Santana BJR, Vasconcelos EEM, Silva ER, Roque A, Torner L, Guzmán-Quevedo O, Toscano AE. Creatine supplementation increases postnatal growth and strength and prevents overexpression of pro-inflammatory interleukin 6 in the hippocampus in an experimental model of cerebral palsy. Nutr Neurosci 2024; 27:425-437. [PMID: 37141266 DOI: 10.1080/1028415x.2023.2206688] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/05/2023]
Abstract
ABSTRACTObjectives: The aim of this study was thus to evaluate the effect of Cr supplementation on morphological changes and expression of pro-inflammatory cytokines in the hippocampus and on developmental parameters. Methods: Male Wistar rat pups were submitted to an experimental model of CP. Cr was administered via gavage from the 21st to the 28th postnatal day, and in water after the 28th, until the end of the experiment. Body weight (BW), food consumption (FC), muscle strength, and locomotion were evaluated. Expression of interleukin-1β (IL-1β), interleukin-6 (IL-6), and tumor necrosis factor α (TNF-α) were assessed in the hippocampus by quantitative real-time polymerase chain reaction. Iba1 immunoreactivity was assessed by immunocytochemistry in the hippocampal hilus. Results: Experimental CP caused increased density and activation of microglial cells, and overexpression of IL-6. The rats with CP also presented abnormal BW development and impairment of strength and locomotion. Cr supplementation was able to reverse the overexpression of IL-6 in the hippocampus and mitigate the impairments observed in BW, strength, and locomotion. Discussion: Future studies should evaluate other neurobiological characteristics, including changes in neural precursor cells and other cytokines, both pro- and anti-inflammatory.
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Affiliation(s)
- Henrique J C B Gouveia
- Postgraduate Program in Nutrition, Health Sciences Center, Federal University of Pernambuco, Recife, Brazil
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
- Tecnológico Nacional de México (TECNM) - Instituto Tecnológico Superior de Tacámbaro, Tacámbaro, Michoacán, Mexico
- Centro de Investigación Biomédica de Michoacán, Instituto Mexicano del Seguro Social, Morelia, Michoacán, Mexico
| | - Raul Manhães-de-Castro
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
| | - Bárbara J R Costa-de-Santana
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
- Postgraduate Program in Neuropsychiatry and Behavioral Sciences, Federal University of Pernambuco, Recife, Brazil
| | - Emanuel Ewerton M Vasconcelos
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
| | - Eliesly Roberto Silva
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
| | - Angélica Roque
- Centro de Investigación Biomédica de Michoacán, Instituto Mexicano del Seguro Social, Morelia, Michoacán, Mexico
| | - Luz Torner
- Centro de Investigación Biomédica de Michoacán, Instituto Mexicano del Seguro Social, Morelia, Michoacán, Mexico
| | - Omar Guzmán-Quevedo
- Tecnológico Nacional de México (TECNM) - Instituto Tecnológico Superior de Tacámbaro, Tacámbaro, Michoacán, Mexico
- Centro de Investigación Biomédica de Michoacán, Instituto Mexicano del Seguro Social, Morelia, Michoacán, Mexico
- Postgraduate Program in Neuropsychiatry and Behavioral Sciences, Federal University of Pernambuco, Recife, Brazil
| | - Ana E Toscano
- Studies in Nutrition and Phenotypic Plasticity Unit, Department of Nutrition, Federal University of Pernambuco, Recife, Brazil
- Postgraduate Program in Neuropsychiatry and Behavioral Sciences, Federal University of Pernambuco, Recife, Brazil
- Department of Nursing, CAV, Federal University of Pernambuco, Recife, Brazil
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Pons R, Zoi D. "Deconstructing" upper limb function in dyskinetic cerebral palsy. Eur J Paediatr Neurol 2024; 50:v-vi. [PMID: 38760305 DOI: 10.1016/j.ejpn.2024.05.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 05/19/2024]
Affiliation(s)
- Roser Pons
- First Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Thivon and Levadias, Athens, 11527, Greece.
| | - Dalivigka Zoi
- Pediatric Rehabilitation Unit, Pan & Aglaias Kyriakou Children's Hospital, Leof. Antrea Siggrou 290, Kalithea, 17673, Greece.
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San Luciano M, Oehrn CR, Wang SS, Tolmie JS, Wiltshire A, Graff RE, Zhu J, Starr PA. Protocol for combined N-of-1 trials to assess cerebellar neurostimulation for movement disorders in children and young adults with dyskinetic cerebral palsy. BMC Neurol 2024; 24:145. [PMID: 38684956 PMCID: PMC11057158 DOI: 10.1186/s12883-024-03633-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/11/2024] [Accepted: 04/09/2024] [Indexed: 05/02/2024] Open
Abstract
BACKGROUND Movement and tone disorders in children and young adults with cerebral palsy are a great source of disability. Deep brain stimulation (DBS) of basal ganglia targets has a major role in the treatment of isolated dystonias, but its efficacy in dyskinetic cerebral palsy (DCP) is lower, due to structural basal ganglia and thalamic damage and lack of improvement of comorbid choreoathetosis and spasticity. The cerebellum is an attractive target for DBS in DCP since it is frequently spared from hypoxic ischemic damage, it has a significant role in dystonia network models, and small studies have shown promise of dentate stimulation in improving CP-related movement and tone disorders. METHODS Ten children and young adults with DCP and disabling movement disorders with or without spasticity will undergo bilateral DBS in the dorsal dentate nucleus, with the most distal contact ending in the superior cerebellar peduncle. We will implant Medtronic Percept, a bidirectional neurostimulator that can sense and store brain activity and deliver DBS therapy. The efficacy of cerebellar DBS in improving quality of life and motor outcomes will be tested by a series of N-of-1 clinical trials. Each N-of-1 trial will consist of three blocks, each consisting of one month of effective stimulation and one month of sham stimulation in a random order with weekly motor and quality of life scales as primary and secondary outcomes. In addition, we will characterize abnormal patterns of cerebellar oscillatory activity measured by local field potentials from the intracranial electrodes related to clinical assessments and wearable monitors. Pre- and 12-month postoperative volumetric structural and functional MRI and diffusion tensor imaging will be used to identify candidate imaging markers of baseline disease severity and response to DBS. DISCUSSION Our goal is to test a cerebellar neuromodulation therapy that produces meaningful changes in function and well-being for people with CP, obtain a mechanistic understanding of the underlying brain network disorder, and identify physiological and imaging-based predictors of outcomes useful in planning further studies. TRIAL REGISTRATION ClinicalTrials.gov NCT06122675, first registered November 7, 2023.
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Affiliation(s)
- M San Luciano
- Movement Disorders and Neuromodulation Center, Department of Neurology, University of California San Francisco, Weill Institute for Neurosciences, 1651 4th Street Level 3 SW Academic Offices, Box #1838, 94158, San Francisco, CA, USA.
| | - C R Oehrn
- Department of Neurological Surgery, University of California San Francisco, San Francisco, CA, USA
| | - S S Wang
- Movement Disorders and Neuromodulation Center, Department of Neurology, University of California San Francisco, Weill Institute for Neurosciences, 1651 4th Street Level 3 SW Academic Offices, Box #1838, 94158, San Francisco, CA, USA
| | - J S Tolmie
- Movement Disorders and Neuromodulation Center, Department of Neurology, University of California San Francisco, Weill Institute for Neurosciences, 1651 4th Street Level 3 SW Academic Offices, Box #1838, 94158, San Francisco, CA, USA
| | - A Wiltshire
- Movement Disorders and Neuromodulation Center, Department of Neurology, University of California San Francisco, Weill Institute for Neurosciences, 1651 4th Street Level 3 SW Academic Offices, Box #1838, 94158, San Francisco, CA, USA
| | - R E Graff
- Department of Epidemiology and Biostatistics, University of California San Francisco, San Francisco, CA, USA
| | - J Zhu
- Movement Disorders and Neuromodulation Center, Department of Neurology, University of California San Francisco, Weill Institute for Neurosciences, 1651 4th Street Level 3 SW Academic Offices, Box #1838, 94158, San Francisco, CA, USA
| | - P A Starr
- Department of Neurological Surgery, University of California San Francisco, San Francisco, CA, USA
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San Luciano M, Oehrn CR, Wang SS, Tolmie JS, Wiltshire A, Graff RE, Zhu J, Starr PA. Protocol for combined N-of-1 trials to assess cerebellar neurostimulation for movement disorders in children and young adults with dyskinetic cerebral palsy. RESEARCH SQUARE 2024:rs.3.rs-4077387. [PMID: 38645256 PMCID: PMC11030503 DOI: 10.21203/rs.3.rs-4077387/v1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 04/23/2024]
Abstract
Background Movement and tone disorders in children and young adults with cerebral palsy are a great source of disability. Deep brain stimulation (DBS) of basal ganglia targets has a major role in the treatment of isolated dystonias, but its efficacy in dyskinetic cerebral palsy (DCP) is lower, due to structural basal ganglia and thalamic damage and lack of improvement of comorbid choreoathetosis and spasticity. The cerebellum is an attractive target for DBS in DCP since it is frequently spared from hypoxic ischemic damage, it has a significant role in dystonia network models, and small studies have shown promise of dentate stimulation in improving CP-related movement and tone disorders. Methods Ten children and young adults with DCP and disabling movement disorders with or without spasticity will undergo bilateral DBS in the dorsal dentate nucleus, with the most distal contact ending in the superior cerebellar peduncle. We will implant Medtronic Percept, a bidirectional neurostimulator that can sense and store brain activity and deliver DBS therapy. The efficacy of cerebellar DBS in improving quality of life and motor outcomes will be tested by a series of N-of-1 clinical trials. Each N-of-1 trial will consist of three blocks, each consisting of one month of effective stimulation and one month of sham stimulation in a random order with weekly motor and quality of life scales as primary and secondary outcomes. In addition, we will characterize abnormal patterns of cerebellar oscillatory activity measured by local field potentials from the intracranial electrodes related to clinical assessments and wearable monitors. Pre- and 12-month postoperative volumetric structural and functional MRI and diffusion tensor imaging will be used to identify candidate imaging markers of baseline disease severity and response to DBS. Discussion Our goal is to test a cerebellar neuromodulation therapy that produces meaningful changes in function and well-being for people with CP, obtain a mechanistic understanding of the underlying brain network disorder, and identify physiological and imaging-based predictors of outcomes useful in planning further studies. Trial registration ClinicalTrials.gov NCT06122675, first registered November 7, 2023.
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Affiliation(s)
- Marta San Luciano
- University of California, San Francisco, Weill Institute for Neurosciences
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Trinh A, Lim A, Wong P, Brown J, Pitkin J, Wollenhoven B, Ebeling P, Fuller P, Milat F, Zacharin M. A pilot study proposing an algorithm for pubertal induction in cerebral palsy. J Pediatr Endocrinol Metab 2024; 37:222-227. [PMID: 38374118 DOI: 10.1515/jpem-2024-0013] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/06/2024] [Accepted: 01/18/2024] [Indexed: 02/21/2024]
Abstract
OBJECTIVES To explore delayed puberty in cerebral palsy (CP) and to test the acceptability of an interventional puberty induction algorithm. METHODS A two phase cohort study in children and adolescents diagnosed with CP who have delayed puberty. Phase 1: Retrospective review of clinical records and interviews with patients who have been treated with sex-steroids and Phase 2: Prospective interventional trial of pubertal induction with a proposed algorithm of transdermal testosterone (males) or oestrogen (females). Phase 1 examined experiences with sex-steroid treatment. Phase 2 collected data on height adjusted bone mineral density (BMAD), fractures, adverse effects, mobility and quality of life over two years during the induction. RESULTS Phase 1, treatment was well tolerated in 11/20 treated with sex-steroids; phase 2, using the proposed induction algorithm, 7/10 treated reached Tanner stage 3 by nine months. One participant reached Tanner stage 5 in 24 months. Mean change in BMAD Z-scores was +0.27 % (SD 0.002) in those who could be scanned by dual-energy X-ray absorptiometry (DXA). CONCLUSIONS Delayed puberty may be diagnosed late. Treatment was beneficial and well tolerated, suggesting all patients with severe pubertal delay or arrest should be considered for sex hormone supplementation.
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Affiliation(s)
- Anne Trinh
- Monash Health, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Angelina Lim
- Murdoch Children's Research Institute, Parkville, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Phillip Wong
- Hudson Institute of Medical Research, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Justin Brown
- Monash Health, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Janne Pitkin
- Murdoch Children's Research Institute, Parkville, VIC, Australia
| | - Beverley Wollenhoven
- Monash Health, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Peter Ebeling
- Monash Health, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Peter Fuller
- Hudson Institute of Medical Research, Clayton, VIC, Australia
| | - Frances Milat
- Hudson Institute of Medical Research, Clayton, VIC, Australia
- Monash University, Melbourne, VIC, Australia
| | - Margaret Zacharin
- Murdoch Children's Research Institute, Parkville, VIC, Australia
- Royal Children's Hospital, Melbourne, Australia
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Kleeren L, Mailleux L, McLean B, Elliott C, Dequeker G, Van Campenhout A, de Xivry JJO, Verheyden G, Ortibus E, Klingels K, Feys H. Does somatosensory discrimination therapy alter sensorimotor upper limb function differently compared to motor therapy in children and adolescents with unilateral cerebral palsy: study protocol for a randomized controlled trial. Trials 2024; 25:147. [PMID: 38409060 PMCID: PMC10895830 DOI: 10.1186/s13063-024-07967-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/26/2023] [Accepted: 02/05/2024] [Indexed: 02/28/2024] Open
Abstract
BACKGROUND Besides motor impairments, up to 90% of the children and adolescents with unilateral cerebral palsy (uCP) present with somatosensory impairments in the upper limb. As somatosensory information is of utmost importance for coordinated movements and motor learning, somatosensory impairments can further compromise the effective use of the impaired upper limb in daily life activities. Yet, intervention approaches specifically designated to target these somatosensory impairments are insufficiently investigated in children and adolescents with uCP. Therefore, the aim of this randomized controlled trial (RCT) is to compare the effectiveness of somatosensory discrimination therapy and dose-matched motor therapy to improve sensorimotor upper limb function in children and adolescents with uCP, who experience somatosensory impairments in the upper limb. We will further explore potential behavioral and neurological predictors of therapy response. METHODS A parallel group, evaluator-blinded, phase-II, single-center RCT will be conducted for which 50 children and adolescents with uCP, aged 7 to 15 years, will be recruited. Participants will be randomized to receive 3 weekly sessions of 45 minutes of either somatosensory discrimination therapy or upper limb motor therapy for a period of 8 weeks. Stratification will be performed based on age, manual ability, and severity of tactile impairment at baseline. Sensorimotor upper limb function will be evaluated at baseline, immediately after the intervention and after 6 months follow-up. The primary outcome measure will be bimanual performance as measured with the Assisting Hand Assessment. Secondary outcomes include a comprehensive test battery to objectify somatosensory function and measures of bimanual coordination, unimanual motor function, and goal attainment. Brain imaging will be performed at baseline to investigate structural brain lesion characteristics and structural connectivity of the white matter tracts. DISCUSSION This protocol describes the design of an RCT comparing the effectiveness of somatosensory discrimination therapy and dose-matched motor therapy to improve sensorimotor upper limb function in children and adolescents with uCP. The results of this study may aid in the selection of the most effective upper limb therapy, specifically for children and adolescents with tactile impairments. TRIAL REGISTRATION ClinicalTrials.gov (NCT06006065). Registered on August 8, 2023.
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Affiliation(s)
- Lize Kleeren
- KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation, Leuven, B-3001, Belgium.
- KU Leuven, Child and Youth Institute, Leuven, B-3000, Belgium.
- Hasselt University, Rehabilitation Research Centre, Faculty of Rehabilitation Sciences, Diepenbeek, B-3590, Belgium.
| | - Lisa Mailleux
- KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation, Leuven, B-3001, Belgium
- KU Leuven, Child and Youth Institute, Leuven, B-3000, Belgium
| | - Belinda McLean
- Curtin School of Allied Health, Faculty of Health Sciences, Curtin University, Perth, Australia
- Kids Rehab WA, Telethon Kids Institute, Perth, Australia
| | - Catherine Elliott
- Curtin School of Allied Health, Faculty of Health Sciences, Curtin University, Perth, Australia
- Kids Rehab WA, Telethon Kids Institute, Perth, Australia
| | - Griet Dequeker
- University Hospitals Leuven, Cerebral Palsy Reference Centre, Leuven, B-3000, Belgium
| | - Anja Van Campenhout
- KU Leuven, Child and Youth Institute, Leuven, B-3000, Belgium
- University Hospitals Leuven, Cerebral Palsy Reference Centre, Leuven, B-3000, Belgium
- KU Leuven, Department of Development and Regeneration, Leuven, B-3000, Belgium
| | - Jean-Jacques Orban de Xivry
- KU Leuven, Leuven Brain Institute, Leuven, B-3000, Belgium
- KU Leuven, Department of Movement Sciences, Research Group of Motor Control and Neuroplasticity, Leuven, B-3000, Belgium
| | - Geert Verheyden
- KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation, Leuven, B-3001, Belgium
| | - Els Ortibus
- KU Leuven, Child and Youth Institute, Leuven, B-3000, Belgium
- University Hospitals Leuven, Cerebral Palsy Reference Centre, Leuven, B-3000, Belgium
- KU Leuven, Department of Development and Regeneration, Leuven, B-3000, Belgium
| | - Katrijn Klingels
- KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation, Leuven, B-3001, Belgium
- Hasselt University, Rehabilitation Research Centre, Faculty of Rehabilitation Sciences, Diepenbeek, B-3590, Belgium
| | - Hilde Feys
- KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation, Leuven, B-3001, Belgium
- KU Leuven, Child and Youth Institute, Leuven, B-3000, Belgium
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Santana Almansa A, Gable DL, Frazier Z, Sveden A, Quinlan A, Chopra M, Lewis SA, Kruer M, Poduri A, Srivastava S. Clinical utility of a genetic diagnosis in individuals with cerebral palsy and related motor disorders. Ann Clin Transl Neurol 2024; 11:251-262. [PMID: 38168508 PMCID: PMC10863912 DOI: 10.1002/acn3.51942] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/20/2023] [Revised: 10/07/2023] [Accepted: 10/19/2023] [Indexed: 01/05/2024] Open
Abstract
OBJECTIVE Evaluation of the clinical utility of a genetic diagnosis in CP remains limited. We aimed to characterize the clinical utility of a genetic diagnosis by exome sequencing (ES) in patients with CP and related motor disorders. METHODS We enrolled participants with CP and "CP masquerading" conditions in an institutional ES initiative. In those with genetic diagnoses who had clinical visits to discuss results, we retrospectively reviewed medical charts, evaluating recommendations based on the genetic diagnosis pertaining to medication intervention, surveillance initiation, variant-specific testing, and patient education. RESULTS We included 30 individuals with a molecular diagnosis and clinical follow-up. Nearly all (28 out of 30) had clinical impact resulting from the genetic diagnosis. Medication interventions included recommendation of mitochondrial multivitamin supplementation (6.67%, n = 2), ketogenic diet (3.33%, n = 1), and fasting avoidance (3.33%, n = 1). Surveillance-related actions included recommendations for investigating systemic complications (40%, n = 12); referral to new specialists to screen for systemic manifestations (33%, n = 10); continued follow-up with established specialists to focus on specific manifestations (16.67%, n = 5); referral to clinical genetics (16.67%, n = 5) to oversee surveillance recommendations. Variant-specific actions included carrier testing (10%, n = 3) and testing of potentially affected relatives (3.33%, n = 1). Patient education-specific actions included referral to experts in the genetic disorder (30%, n = 9); and counseling about possible changes in prognosis, including recognition of disease progression and early mortality (36.67%, n = 11). INTERPRETATION This study highlights the clinical utility of a genetic diagnosis for CP and "CP masquerading" conditions, evident by medication interventions, surveillance impact, family member testing, and patient education, including possible prognostic changes.
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Affiliation(s)
- Alexandra Santana Almansa
- Child Neurology Residency Training ProgramBoston Children's HospitalBostonMassachusettsUSA
- Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
| | - Dustin L. Gable
- Child Neurology Residency Training ProgramBoston Children's HospitalBostonMassachusettsUSA
- Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
| | - Zoë Frazier
- Rosamund Stone Zander Translational Neuroscience Center, Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
| | - Abigail Sveden
- Rosamund Stone Zander Translational Neuroscience Center, Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
| | - Aisling Quinlan
- Rosamund Stone Zander Translational Neuroscience Center, Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
| | - Maya Chopra
- Rosamund Stone Zander Translational Neuroscience Center, Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
- Harvard Medical SchoolBoston Children's HospitalBostonMassachusettsUSA
| | - Sara A. Lewis
- Department of Neurology and PediatricsPhoenix Children's HospitalPhoenixArizonaUSA
| | - Michael Kruer
- Department of Neurology and PediatricsPhoenix Children's HospitalPhoenixArizonaUSA
| | - Annapurna Poduri
- Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
- Harvard Medical SchoolBoston Children's HospitalBostonMassachusettsUSA
- Neurogenetics Program and Epilepsy Genetics ProgramBoston Children's HospitalBostonMassachusettsUSA
| | - Siddharth Srivastava
- Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
- Rosamund Stone Zander Translational Neuroscience Center, Department of NeurologyBoston Children's HospitalBostonMassachusettsUSA
- Harvard Medical SchoolBoston Children's HospitalBostonMassachusettsUSA
- Cerebral Palsy and Spasticity CenterBoston Children's HospitalBostonMassachusettsUSA
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Wang B, Wang F, Yang L, Jiang J, Zhu J, Duan J, Yuan J, Tang J, Wu D. Plasma neuron specific enolase (NSE), tumour necrosis factor-alpha (TNF-α) and soluble IL-2 receptor alpha (sIL-2Rα) levels in children with developmental delay (DD): Use of combined ROC curves to increase their diagnostic value. Cytokine 2024; 174:156469. [PMID: 38101168 DOI: 10.1016/j.cyto.2023.156469] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/06/2023] [Revised: 11/23/2023] [Accepted: 12/04/2023] [Indexed: 12/17/2023]
Abstract
BACKGROUND Developmental delay (DD) occurs when children fail to reach developmental milestones in comparison to peers of the same age range. However, there are no valuable biomarkers for the early diagnosis of DD. Since there is no specific marker for screening the disease, we evaluated plasma NSE, TNF-α and sIL2-Rα as potential markers for this purpose. METHODS In this cross-sectional randomized case-control study, a total of 174 DD patients and 49 matched elderly controls aged between 2 months and 60 months were recruited. A sensitive enzyme-linked immunosorbent assay and an immunoradiometric assay were used to evaluate the levels of plasma IL-1, IL-6, IL-8, IL-10, sIL2-Rα, TNF-α, and NSE. Statistical analyses using t test, χ2, ANOVA, ROC curves and binary logistic regression models were performed. RESULTS In comparison to the control group, the DD group had greater levels of NSE, TNF-α, and sIL2-Rα(p < 0.05). In the binary logistic regression analysis of DD, NSE had an odds ratio (OR) of 1.783 (95 % CI 1.297 to 2.451, p = 0.000), indicating that NSE was an independent risk factor for DD. The plasma TNF-α level was positively correlated with plasma NSE and sIL2-Rα levels in the DD group (r = 0.366 and 0.433, respectively), and the DQ score and plasma sIL2-Rα level in the DD group were positively correlated. The ROC curve revealed that the respective areas under the NSE, TNF-α, and sIL2-Rα ROC curves were 0.9797, 0.9365, and 0.8533, respectively. Moreover, a significant increase in AUC was observed using combined ROC curve analysis. CONCLUSIONS Children with DD have significantly altered plasma concentrations of sIL2-Rα, NSE, and TNF-α. NSE, TNF-α and sIL2-Rα can be used as DD blood biomarkers. This information may be helpful in early diagnosis and intervention.
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Affiliation(s)
- Baotian Wang
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - Fan Wang
- Department of Outpatient, the First Affiliated Hospital of Anhui Medical University, China.
| | - Li Yang
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China
| | - Junhong Jiang
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - Jing Zhu
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - Jun Duan
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - Jinjing Yuan
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - Jiulai Tang
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
| | - De Wu
- Department of Pediatrics, the First Affiliated Hospital of Anhui Medical University, Hefei, Anhui 230022, PR China.
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Prosser LA, Pierce SR, Skorup JA, Paremski AC, Alcott M, Bochnak M, Ruwaih N, Jawad AF. Motor training for young children with cerebral palsy: A single-blind randomized controlled trial. Dev Med Child Neurol 2024; 66:233-243. [PMID: 37550991 DOI: 10.1111/dmcn.15729] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/10/2022] [Revised: 07/07/2023] [Accepted: 07/12/2023] [Indexed: 08/09/2023]
Abstract
AIM To compare the effect of iMOVE (Intensive Mobility training with Variability and Error) therapy with dose-matched conventional therapy on gross motor development and secondary outcomes in young children with cerebral palsy. METHOD This single-blind, randomized controlled trial included repeated assessments of gross motor function (using the Gross Motor Function Measure) and secondary outcomes during a 12- to 24-week intervention phase and at three follow-up points after treatment. Treatment was delivered three times per week in both groups. Forty-two children aged 12 to 36 months were stratified by age and motor function to ensure equivalence between groups at baseline. RESULTS Thirty-six children completed treatment and follow-up phases. Treatment fidelity was high and adherence was equivalent between groups (77.3% conventional therapy, 76.2% iMOVE). There were no group differences on the primary (gross motor function after 12 weeks p = 0.18; after 24 weeks p = 0.94) or any secondary (postural control p = 0.88, caregiver satisfaction p = 0.52, child engagement p = 0.98) measure after treatment or at the follow-up points. However, one-third of total participants exceeded predicted change after 12 weeks and 77% exceeded predicted change after 24 weeks of treatment. INTERPRETATION Our observations indicate a potential dose-response effect of rehabilitation therapy. We further demonstrated that individual therapeutic ingredients can be manipulated. When delivered consistently, both iMOVE and conventional therapy interventions might both be more effective than standard care. WHAT THIS PAPER ADDS Those receiving iMOVE therapy demonstrated more independent practice time, error, and child-initiation than those receiving the dose-matched control. iMOVE therapy was not superior to the control (conventional physical) therapy. Most participants exceeded predicted change after 24 weeks of treatment.
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Affiliation(s)
- Laura A Prosser
- Division of Rehabilitation Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA
- Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
| | - Samuel R Pierce
- Department of Physical Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA
| | - Julie A Skorup
- Department of Physical Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA
| | - Athylia C Paremski
- Division of Rehabilitation Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA
| | - Morgan Alcott
- Department of Physical Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA
| | - Meghan Bochnak
- Department of Physical Therapy, Rady Children's Hospital, San Diego, CA, USA
| | - Noor Ruwaih
- Division of Rehabilitation Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA
| | - Abbas F Jawad
- Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
- Division of General Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA, USA
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Herron MS, Wang L, von Bartheld CS. Prevalence and types of strabismus in cerebral palsy: A global and historical perspective based on a systematic review and meta-analysis. MEDRXIV : THE PREPRINT SERVER FOR HEALTH SCIENCES 2024:2024.01.23.24301684. [PMID: 38343841 PMCID: PMC10854329 DOI: 10.1101/2024.01.23.24301684] [Citation(s) in RCA: 2] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Indexed: 02/18/2024]
Abstract
Purpose Strabismus is more frequent in cerebral palsy (CP) than in the normal population, but reports differ how much it is increased. We here examined the global prevalence and types of strabismus in CP, whether esotropia or exotropia is more frequent, and whether the prevalence differs between ethnicities and/or country income levels, and between generations. Methods We compiled in a systematic review and meta-analysis the results of 147 CP studies that report the prevalence of strabismus or the ratio of esotropia to exotropia, and we conducted subgroup analyses for region (income level) and ethnicity. We performed a pooled analysis for the CP strabismus prevalence, and estimated the global number of CP cases with strabismus. Results The pooled prevalence of strabismus in CP is 49.8% in high-income countries and 39.8% in lower-income countries. We estimate the global number of strabismus cases in CP as 12.2 million, with 7.6 million males and 4.6 million females, based on current estimates of 29.6 million global CP cases. Esotropia is more frequent than exotropia in Caucasians, while exotropia is more frequent than esotropia in Hispanic and in some Asian and African populations. The strabismus prevalence in CP increases with increasing country income levels. Conclusion Generational changes in strabismus prevalence appear to reflect a transition of CP types and an increase in prevalence as countries attain higher income and more effective maternal health care. The distribution of esotropia and exotropia in CP patients largely reflects the horizontal strabismus type that is predominant in the subject's ethnicity.
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Affiliation(s)
- Michael S. Herron
- Center of Biomedical Research Excellence in Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
| | - Lingchen Wang
- School of Public Health, University of Nevada, Reno, Nevada, USA
| | - Christopher S. von Bartheld
- Center of Biomedical Research Excellence in Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
- Department of Physiology and Cell Biology, University of Nevada, Reno School of Medicine, Reno, Nevada, USA
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Sachdeva R, Girshin K, Shirkhani Y, Gad P. Beyond pediatrics: noninvasive spinal neuromodulation improves motor function in an adult with cerebral palsy. Bioelectron Med 2024; 10:1. [PMID: 38167312 PMCID: PMC10762938 DOI: 10.1186/s42234-023-00133-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/04/2023] [Accepted: 11/09/2023] [Indexed: 01/05/2024] Open
Abstract
Regaining motor function in individuals with cerebral palsy (CP) has been predominantly studied in children, resulting in an underrepresentation of adults in research efforts. We tested the efficacy of noninvasive spinal neuromodulation with neurorehabilitation (Spinal Cord Innovation in Pediatrics; SCiP™ therapy). A 60-year-old CP participant underwent 8 weeks of SCiP™ therapy, resulting in significant motor recovery measured by 14.2-points increase in gross motor function measure (GMFM-88) score, ~ three times the Minimal Clinically Important Difference (MCID) of 5-points. This represented gains in kneeling, sitting, and walking functions. The improvement in GMFM-88 score was maintained above the MCID at the follow up visit (10.3 points above the baseline), twenty weeks following the last therapy session, indicating a persistent effect of the therapy. Our preliminary findings support the therapeutic promise of SCiP™ therapy for enhancing motor function in CP adults. Broader investigations are needed to establish its wider applicability.
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Affiliation(s)
- Rahul Sachdeva
- SpineX Inc, Los Angeles, CA, 91324, USA.
- Department of Medicine, International Collaboration On Repair Discoveries (ICORD), University of British Columbia, Vancouver, BC, V5Z1M9, Canada.
| | - Kristin Girshin
- SpineX Inc, Los Angeles, CA, 91324, USA
- GirshinPT Rancho, Cucamunga, CA, 91701, USA
| | | | - Parag Gad
- SpineX Inc, Los Angeles, CA, 91324, USA
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Leal-Martinez F, Jimenez Ramirez G, Ibarra A. Nutritional Support System (NSS) as a New Therapeutic Strategy for Cerebral Palsy. CNS & NEUROLOGICAL DISORDERS DRUG TARGETS 2024; 23:271-277. [PMID: 37005522 DOI: 10.2174/1871527322666230330124124] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 08/16/2022] [Revised: 02/24/2023] [Accepted: 02/26/2023] [Indexed: 04/04/2023]
Abstract
Cerebral palsy (CP) is part of a group of nonprogressive motor disorders. The disease affects movement and posture and constitutes the most frequent cause of motor disability in childhood. CP is characterized by spasticity, reflecting lesions in the pyramidal pathway. Treatment is currently focused on physical rehabilitation, and the annual progression of the disease is 2-3%. About 60% of these patients present severe degrees of malnutrition associated with dysphagia, gastrointestinal abnormalities, malabsorption, increased metabolism, and depression. These alterations promote sarcopenia functional dependence and affect the quality of life and delay the evolution of motor skills. Currently, there is evidence that the supplementation of several nutrients, dietary correction, and probiotics can improve neurological response by stimulating neuroplasticity, neuroregeneration, neurogenesis, and myelination. This therapeutic strategy could shorten the response period to treatment and increase both gross and fine motor skills. The interaction of nutrients and functional foods integrating a Nutritional Support System (NSS) has shown greater efficiency in neurological stimulation than when nutrients are supplied separately. The most studied elements in the neurological response are glutamine, arginine, zinc, selenium, cholecalciferol, nicotinic acid, thiamine, pyridoxine, folate, cobalamin, Spirulina, omega-3 fatty acids, ascorbic acid, glycine, tryptophan, and probiotics. The NSS represents a therapeutic alternative that will restore neurological function in patients with spasticity and pyramidal pathway lesions, both characteristics of patients with CP.
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Affiliation(s)
- Fernando Leal-Martinez
- Centro de Investigación en Ciencias de la Salud (CICSA), FCS, Universidad Anáhuac México Campus Norte. Av. Universidad Anáhuac No. 46, Col. Lomas Anáhuac, Huixquilucan Edo. de México; CP 52786; México
| | - Guadalupe Jimenez Ramirez
- Centro de Investigación en Ciencias de la Salud (CICSA), FCS, Universidad Anáhuac México Campus Norte. Av. Universidad Anáhuac No. 46, Col. Lomas Anáhuac, Huixquilucan Edo. de México; CP 52786; México
| | - Antonio Ibarra
- Centro de Investigación en Ciencias de la Salud (CICSA), FCS, Universidad Anáhuac México Campus Norte. Av. Universidad Anáhuac No. 46, Col. Lomas Anáhuac, Huixquilucan Edo. de México; CP 52786; México
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Gao Q, Yao S, Tian Y, Zhang C, Zhao T, Wu D, Yu G, Lu H. Automating General Movements Assessment with quantitative deep learning to facilitate early screening of cerebral palsy. Nat Commun 2023; 14:8294. [PMID: 38097602 PMCID: PMC10721621 DOI: 10.1038/s41467-023-44141-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/27/2023] [Accepted: 12/01/2023] [Indexed: 12/17/2023] Open
Abstract
The Prechtl General Movements Assessment (GMA) is increasingly recognized for its role in evaluating the integrity of the developing nervous system and predicting motor dysfunctions, particularly in conditions such as cerebral palsy (CP). However, the necessity for highly trained professionals has hindered the adoption of GMA as an early screening tool in some countries. In this study, we propose a deep learning-based motor assessment model (MAM) that combines infant videos and basic characteristics, with the aim of automating GMA at the fidgety movements (FMs) stage. MAM demonstrates strong performance, achieving an Area Under the Curve (AUC) of 0.967 during external validation. Importantly, it adheres closely to the principles of GMA and exhibits robust interpretability, as it can accurately identify FMs within videos, showing substantial agreement with expert assessments. Leveraging the predicted FMs frequency, a quantitative GMA method is introduced, which achieves an AUC of 0.956 and enhances the diagnostic accuracy of GMA beginners by 11.0%. The development of MAM holds the potential to significantly streamline early CP screening and revolutionize the field of video-based quantitative medical diagnostics.
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Affiliation(s)
- Qiang Gao
- State Key Laboratory of Microbial Metabolism, Joint International Research Laboratory of Metabolic and Developmental Sciences, Department of Bioinformatics and Biostatistics, School of Life Sciences and Biotechnology, Shanghai Jiao Tong University, Shanghai, China
| | - Siqiong Yao
- State Key Laboratory of Microbial Metabolism, Joint International Research Laboratory of Metabolic and Developmental Sciences, Department of Bioinformatics and Biostatistics, School of Life Sciences and Biotechnology, Shanghai Jiao Tong University, Shanghai, China
- SJTU-Yale Joint Center of Biostatistics and Data Science, National Center for Translational Medicine, MoE Key Lab of Artificial Intelligence, AI Institute, Shanghai Jiao Tong University, Shanghai, China
| | - Yuan Tian
- Department of Health Management, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Chuncao Zhang
- Department of Health Management, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
| | - Tingting Zhao
- Shanghai Engineering Research Center for Big Data in Pediatric Precision Medicine, NHC Key Laboratory of Medical Embryogenesis and Developmental Molecular Biology & Shanghai Key Laboratory of Embryo and Reproduction Engineering, Shanghai Jiao Tong University, Shanghai, China
| | - Dan Wu
- Shanghai Engineering Research Center for Big Data in Pediatric Precision Medicine, NHC Key Laboratory of Medical Embryogenesis and Developmental Molecular Biology & Shanghai Key Laboratory of Embryo and Reproduction Engineering, Shanghai Jiao Tong University, Shanghai, China
| | - Guangjun Yu
- Shanghai Engineering Research Center for Big Data in Pediatric Precision Medicine, NHC Key Laboratory of Medical Embryogenesis and Developmental Molecular Biology & Shanghai Key Laboratory of Embryo and Reproduction Engineering, Shanghai Jiao Tong University, Shanghai, China.
- School of Medicine, The Chinese University of Hong Kong, Shenzhen, Guangdong, China.
| | - Hui Lu
- State Key Laboratory of Microbial Metabolism, Joint International Research Laboratory of Metabolic and Developmental Sciences, Department of Bioinformatics and Biostatistics, School of Life Sciences and Biotechnology, Shanghai Jiao Tong University, Shanghai, China.
- SJTU-Yale Joint Center of Biostatistics and Data Science, National Center for Translational Medicine, MoE Key Lab of Artificial Intelligence, AI Institute, Shanghai Jiao Tong University, Shanghai, China.
- Shanghai Engineering Research Center for Big Data in Pediatric Precision Medicine, NHC Key Laboratory of Medical Embryogenesis and Developmental Molecular Biology & Shanghai Key Laboratory of Embryo and Reproduction Engineering, Shanghai Jiao Tong University, Shanghai, China.
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Suh MR, Min K, Cho KH, Kim J, Lim I, Park M, Noh EM, Kim MY. Maintenance of the synergistic effects of cord blood cells and erythropoietin combination therapy after additional cord blood infusion in children with cerebral palsy: 1-year open-label extension study of randomized placebo-controlled trial. Stem Cell Res Ther 2023; 14:362. [PMID: 38087394 PMCID: PMC10717973 DOI: 10.1186/s13287-023-03600-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/06/2022] [Accepted: 12/05/2023] [Indexed: 12/18/2023] Open
Abstract
BACKGROUND This 1-year open-label extension study aimed to identify the persistent synergistic effects of allogeneic umbilical cord blood (UCB) cells and erythropoietin (EPO) in children with cerebral palsy (CP) for up to 2 years. METHODS This open-label extension study followed children with CP who were enrolled in the previous randomized, double blind, placebo-controlled trial. The following groups from the first trial were maintained: (A) UCB + EPO, (B) UCB, (C) EPO, and (D) only placebo, and all the participants had continued active rehabilitation. This extended study started 3 months after termination of the first trial, which had a 1-year follow-up duration. All subjects received single additional UCB intravenous infusion at the extension baseline regardless of their initial allocation. Outcome measures were the gross motor performance measure (GMPM), gross motor function measure-66 (GMFM-66), and Bayley scales of infant development-II (BSID-II), which were followed at 3, 6, and 12 months after the extension baseline. Changes in the outcome scores from the baseline values of the previous trial and this study were analysed. RESULTS Sixty-nine children (4.29 ± 1.28 years, M:F = 34:35) were included in this study. Each group showed improvements in the outcome measures at 12 months after additional UCB infusion compared to the baseline scores, except for GMFM and GMPM in Group C which were elevated at 3 and 6 months post-therapy. Total subject analyses did not show significant differences in the outcome measures between the four different groups at 3, 6 and 12 months after additional UCB therapy. However, patients with severe dysfunction, whose GMFCS levels were IV and V, revealed a larger improvement of the GMPM score in Group A than in Group D (Ps < 0.05) from the baseline value of the previous trial. The changes in BSID-II mental scale scores were positively correlated with the number of administered total nucleated cells per unit body weight during this one-year extension study period (r = 0.536, P = 0.001). CONCLUSIONS These results suggest that when administering UCB to treat patients with CP, combination therapy with EPO is more effective, and the effect might last as long as 2 years, especially in patients with severe impairments. TRIAL REGISTRATION CHA Bundang Medical Center IRB, No. 2015-06-093, approved on July 29, 2015, ( https://www.e-irb.com:3443/devlpg/nlpgS200.jsp ), ClinicalTrials.gov, NCT03130816, retrospectively registered on April 27, 2017 ( https://clinicaltrials.gov/ct2/show/NCT03130816?term=NCT03130816&draw=2&rank=1 ).
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Affiliation(s)
- Mi Ri Suh
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea
| | - Kyunghoon Min
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea
| | - Kye Hee Cho
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea
- Department of Rehabilitation Medicine, CHA Ilsan Medical Center, CHA University School of Medicine, Goyang, Republic of Korea
| | - Jongwook Kim
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea
| | - Ikhyun Lim
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea
| | - Mijin Park
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
| | - Eun-Min Noh
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea
| | - Min Young Kim
- Department of Rehabilitation Medicine, CHA Bundang Medical Center, CHA University School of Medicine, 59 Yatap-Ro, Bundang-Gu, Seongnam, Gyeonggi-Do, Republic of Korea.
- Rehabilitation and Regeneration Research Center, CHA University, Pocheon, Republic of Korea.
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Lyster AE, Hansen SL, Andersen CT, Nielsen JB, Westerterp K, Wouters L, Kiens B, Ritterband-Rosenbaum A. Oral Motor Impairments Contribute to Weight Status of Adults with Severe Cerebral Palsy. Nutrients 2023; 15:5042. [PMID: 38140301 PMCID: PMC10745858 DOI: 10.3390/nu15245042] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/01/2023] [Revised: 11/29/2023] [Accepted: 12/02/2023] [Indexed: 12/24/2023] Open
Abstract
Adults with severe cerebral palsy (CP) are susceptible to malnutrition and metabolic disorders due to limited daily physical activity and challenges related to eating. We hypothesized that the condition of being underweight arises from inadequate energy intake due to difficulties in eating, rather than heightened total energy expenditure or an elevated resting metabolic rate. The present study encompassed 17 adults with severe CP (classified as GMFSC III-V). Energy intake, utilization, and expenditure were gauged via thorough dietary recordings and double-labeled water (DLW) analyses. Resting metabolic rates were assessed through indirect calorimetry, and metabolic health was investigated via blood samples. Oral motor function, eating assessment during meals, and weight fluctuations throughout the experimental period were also evaluated. We found significant correlations between weight, oral impairments (p < 0.01), and eating difficulties (p < 0.05). While total energy expenditure and daily consumption were similar between underweight (UW) and overweight (OW) individuals, significant variability in both expenditure and intake was evident within the UW group. Particularly, those with lower BMIs experienced heightened mealtime impairments and complications. Our present findings indicate that eating difficulties are the central concern for UW status in this population.
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Affiliation(s)
- Aslak Emil Lyster
- The August Krogh Section for Molecular Physiology, Department of Nutrition, Exercise and Sports, University of Copenhagen, Universitetsparken 13, 2100 Copenhagen, Denmark; (A.E.L.); (B.K.)
| | - Solvejg Lis Hansen
- Elsass Foundation, Holmegaardsvej 28, 2920 Charlottelund, Denmark; (S.L.H.); (C.T.A.); (J.B.N.)
| | | | - Jens Bo Nielsen
- Elsass Foundation, Holmegaardsvej 28, 2920 Charlottelund, Denmark; (S.L.H.); (C.T.A.); (J.B.N.)
- Department of Neuroscience, University of Copenhagen, Blegdamsvej 3, 2200 Copenhagen, Denmark
| | - Klaas Westerterp
- Department of Nutrition and Movement Sciences, The Maastricht University, P.O. Box 616, 6200 MD Maastricht, The Netherlands; (K.W.); (L.W.)
| | - Loek Wouters
- Department of Nutrition and Movement Sciences, The Maastricht University, P.O. Box 616, 6200 MD Maastricht, The Netherlands; (K.W.); (L.W.)
| | - Bente Kiens
- The August Krogh Section for Molecular Physiology, Department of Nutrition, Exercise and Sports, University of Copenhagen, Universitetsparken 13, 2100 Copenhagen, Denmark; (A.E.L.); (B.K.)
| | - Anina Ritterband-Rosenbaum
- Elsass Foundation, Holmegaardsvej 28, 2920 Charlottelund, Denmark; (S.L.H.); (C.T.A.); (J.B.N.)
- Department of Neuroscience, University of Copenhagen, Blegdamsvej 3, 2200 Copenhagen, Denmark
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49
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Baker AH, Leland SB, Freiman E, Herigon JC, Eisenberg MA. Characteristics and Outcomes of Culture-Positive and Culture-Negative Pediatric Sepsis. J Pediatr 2023; 263:113718. [PMID: 37659590 DOI: 10.1016/j.jpeds.2023.113718] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/31/2023] [Revised: 08/21/2023] [Accepted: 08/29/2023] [Indexed: 09/04/2023]
Abstract
OBJECTIVES To compare the outcomes of pediatric severe sepsis and septic shock among patients with culture-positive and culture-negative sepsis and to determine if there are differentiating markers of disease severity between these 2 populations during their initial presentation and emergency department (ED) stay. STUDY DESIGN Retrospective cohort study of patients ≤21 years of age who presented to the ED of a single children's hospital with severe sepsis or septic shock from June 1, 2017 to June 5, 2019. RESULTS There were 235 patients who met criteria for severe sepsis or septic shock. Of these, 139 (59.1%) had culture-negative sepsis and 96 (40.9%) had culture-positive sepsis. In the adjusted multivariable model, children with culture-negative sepsis had more intensive care unit (ICU)-free days than those with culture-positive sepsis (27.3 vs 24.1; adjusted median differences [aMD] -2.6 [-4.4, -0.8]). There were no differences in mortality or hospital-free days. On initial presentation, there were no differences in fever, hypothermia, tachycardia, tachypnea, or hypotension between the 2 groups. There were no differences in proportion of patients receiving the following interventions: intravenous (IV) antibiotics, IV fluids, vasoactive medications, CPR, intubation, or time from arrival to provision of these interventions. CONCLUSIONS Culture-negative sepsis constitutes a substantial proportion of pediatric severe sepsis and septic shock. In this study, patients with culture-negative and culture-positive sepsis presented similarly on arrival to the ED and received similar treatments while there. Patients with culture-negative sepsis had more ICU-free days than those with culture-positive sepsis, although differences in hospital length of stay (LOS) and mortality were not observed.
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Affiliation(s)
- Alexandra H Baker
- Division of Emergency Medicine, Department of Pediatrics, Boston Children's Hospital, Boston, MA; Department of Pediatrics, Harvard Medical School, Harvard University, Boston, MA.
| | - Shannon B Leland
- Department of Pediatrics, Harvard Medical School, Harvard University, Boston, MA; Department of Anesthesiology, Critical Care, and Pain Medicine, Boston Children's Hospital, Boston, MA
| | - Eli Freiman
- Department of Emergency Medicine, Newton Wellesley Hospital, Newton, MA
| | - Joshua C Herigon
- Division of Infectious Diseases, Department of Pediatrics, Children's Mercy Hospital, Kansas City, MO; Department of Pediatrics, University of Missouri - Kansas City School of Medicine, University of Missouri, Kansas City, MO
| | - Matthew A Eisenberg
- Division of Emergency Medicine, Department of Pediatrics, Boston Children's Hospital, Boston, MA; Department of Pediatrics, Harvard Medical School, Harvard University, Boston, MA
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50
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Pulay MÁ, Nagy R, Kói T, Harnos A, Zimonyi N, Garami M, Gasparics Á, Hegyi P, Túri I, Feketéné Szabó É. The Effect of Additional Whole-Body Vibration on Musculoskeletal System in Children with Cerebral Palsy: A Systematic Review and Meta-Analysis of Randomized Clinical Trials. J Clin Med 2023; 12:6759. [PMID: 37959224 PMCID: PMC10648882 DOI: 10.3390/jcm12216759] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/25/2023] [Revised: 10/20/2023] [Accepted: 10/23/2023] [Indexed: 11/15/2023] Open
Abstract
Nowadays, whole-body vibration (WBV) has become increasingly popular as an additional therapy in the intervention of patients with cerebral palsy (CP). However, the impact of WBV remains a subject of debate. Consequently, a systematic review and meta-analysis were undertaken to evaluate the effects of WBV on the musculoskeletal system in children with CP. Randomized controlled trials (RCTs) were sought in the most frequent databases. The intervention studied was WBV combined with conventional physiotherapy (PT) compared with conventional PT as the control; the main outcomes were changes in the musculoskeletal system. Weighted mean differences with 95%CIs were calculated. A random-effects model was applied, and the publication bias was checked using funnel plots. On the basis of the inclusion and exclusion criteria, 16 articles, including 414 patients, were considered in the final analysis. The improvement in walking performance (speed and step length) was statistically significant (p < 0.05), and although there were no significant differences in the further outcomes, a clear positive tendency was visible in the case of improved muscle strength, decreased spasticity, enhanced gross motor functions, and overall stability. Based on the findings, a clear assessment of the usefulness of this intervention cannot be made; nonetheless, due to the promising results, it would be worthwhile to conduct additional RCTs to enhance the available evidence in this field. Due to the wide range of vibration configurations, including varying durations and intensities, it is suggested to establish guidelines and a strategy for the incorporation of this additional treatment.
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Affiliation(s)
- Márk Ágoston Pulay
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- András Pető Faculty, Semmelweis University, 1125 Budapest, Hungary; (I.T.); (É.F.S.)
- Department of Ergonomics and Psychology, Faculty of Economic and Social Sciences, Budapest University of Technology and Economics, 1111 Budapest, Hungary
| | - Rita Nagy
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- Heim Pál National Pediatric Institute, 1089 Budapest, Hungary
- Institute for Translational Medicine, Medical School, University of Pécs, 7622 Pécs, Hungary
| | - Tamás Kói
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- Department of Stochastics, Institute of Mathematics, Budapest University of Technology and Economics, 1111 Budapest, Hungary
| | - Andrea Harnos
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- Department of Biostatistics, University of Veterinary Medicine, 1078 Budapest, Hungary
| | - Nóra Zimonyi
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- András Pető Faculty, Semmelweis University, 1125 Budapest, Hungary; (I.T.); (É.F.S.)
| | - Miklós Garami
- Pediatric Center, Semmelweis University, 1085 Budapest, Hungary;
| | - Ákos Gasparics
- Department of Obstetrics and Gynecology, Semmelweis University, 1088 Budapest, Hungary;
- Division of Neonatology, Department of Pediatrics, Semmelweis University, 1083 Budapest, Hungary
| | - Péter Hegyi
- Centre for Translational Medicine, Semmelweis University, 1085 Budapest, Hungary; (R.N.); (T.K.); (A.H.); (N.Z.); (P.H.)
- Institute for Translational Medicine, Medical School, University of Pécs, 7622 Pécs, Hungary
- Institute of Pancreatic Diseases, Semmelweis University, 1085 Budapest, Hungary
| | - Ibolya Túri
- András Pető Faculty, Semmelweis University, 1125 Budapest, Hungary; (I.T.); (É.F.S.)
| | - Éva Feketéné Szabó
- András Pető Faculty, Semmelweis University, 1125 Budapest, Hungary; (I.T.); (É.F.S.)
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