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Early Detection and Early Intervention Strategies for Cerebral Palsy in...
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Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings

A special issue of Brain Sciences (ISSN 2076-3425). This special issue belongs to the section "Developmental Neuroscience".

Deadline for manuscript submissions: closed (30 May 2022) | Viewed by 27961

Special Issue Editors

Dr. Atul Malhotra

E-Mail Website
Guest Editor
Department of Paediatrics, Monash University, Melbourne, Australia
Interests: neuroscience; neurodevelopment; cell therapies; global health
Prof. Dr. Roslyn N. Boyd

E-Mail Website
Co-Guest Editor
Queensland Cerebral Palsy & Rehabilitation Research Centre, Child Health Research Centre, The University of Queensland, Brisbane, Australia
Interests: cerebral palsy; adverse neurodevelopment; early intervention; motor optimality; neuroplasticity
Dr. Kath Benfer

E-Mail Website
Co-Guest Editor
Queensland Cerebral Palsy & Rehabilitation Research Centre, Child Health Research Centre, The University of Queensland, Brisbane, Australia
Interests: cerebral palsy; pediatrics; early detection and intervention; global health; health service access; oropharyngeal dysphagia

Special Issue Information

Dear Colleagues,

Cerebral Palsy (CP) is the most common physical disability of childhood. A number of antenatal and postnatal factors increase the risk of CP in infants. Early detection of CP is now feasible based on standardized assessment tools and predictive models. Early intervention strategies for CP have evolved over the last few decades and play an important role in shifting the trajectory of development for affected children. In this Special Issue, we invite researchers from all disciplines of CP research—scientists, clinicians, therapists, and policy decision makers—to inform current best practice and future advances in this important field. This issue will showcase and highlight the best research in CP with a view to improving the outcomes for CP patients and their care providers with special emphasis on research and clinical practice in both Low and High Resource settings. New and leading cutting-edge research in detection, prevention, and treatment of CP is welcome.

We are soliciting in the form of, among others, the following kinds of papers:

  • Basic science
  • Clinical science, including implementation science
  • Quantitative and qualitative research

Dr. Atul Malhotra
Guest Editor

Prof. Dr. Roslyn Boyd
Dr. Kath Benfer
Co-Guest Editors

Manuscript Submission Information

Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. Brain Sciences is an international peer-reviewed open access monthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. The Article Processing Charge (APC) for publication in this open access journal is 2200 CHF (Swiss Francs). Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Keywords

  • Cerebral palsy
  • Early detection
  • Physical therapies
  • Early intervention
  • Neuroprotection

Published Papers (9 papers)

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Editorial

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2 pages, 164 KiB  
Editorial
Editorial: Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings
by Atul Malhotra
Brain Sci. 2022, 12(8), 960; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci12080960 - 22 Jul 2022
Cited by 1 | Viewed by 1162
Abstract
Cerebral palsy (CP) is the most common physical disability in childhood [...] Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)

Research

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12 pages, 1103 KiB  
Article
Assessing the Utility of Neonatal Screening Assessments in Early Diagnosis of Cerebral Palsy in Preterm Infants
by Rebecca Connors, Vathana Sackett, Catherine Machipisa, Kenneth Tan, Pramod Pharande, Lindsay Zhou and Atul Malhotra
Brain Sci. 2022, 12(7), 847; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci12070847 - 28 Jun 2022
Cited by 6 | Viewed by 2408
Abstract
Background: Early diagnosis of cerebral palsy (CP) in high-risk infants is possible at 3–4 months’ corrected age (CA) using standardised assessments. Aim: To assess the utility of neonatal screening assessments—writhing general movements (GMs) and the Hammersmith Neonatal Neurological Examination (HNNE)—to predict CP/high-risk status [...] Read more.
Background: Early diagnosis of cerebral palsy (CP) in high-risk infants is possible at 3–4 months’ corrected age (CA) using standardised assessments. Aim: To assess the utility of neonatal screening assessments—writhing general movements (GMs) and the Hammersmith Neonatal Neurological Examination (HNNE)—to predict CP/high-risk status at 3–4 months’ CA in extremely preterm infants. Methods: Retrospective cohort study of high-risk preterm infants (born < 29 weeks’ gestation and/or birth weight < 1000 g) attending an Early Neurodevelopment Clinic. Data from neonatal assessments were compared with CP/high-risk diagnosis at 3–4 months’ CA, fidgety GMs, and Hammersmith Infant Neurological Examinations (HINE) using logistic regression, linear regression, and Spearman rank correlation. Results: Two hundred and two preterm infants (median gestation age at birth 27.3 (IQR 25.4–28.3) weeks, mean birth weight 870.3 (SD 248.4) grams) were included. A total of 26 (12.8%) infants received early CP/high-risk diagnoses at 3–4 months’ CA. A lower gestational age (GA) (OR = 0.78; p = 0.029, 95% CI [0.26, 0.97]) and abnormal writhing GMs (OR 1.56; p = 0.019, 95% CI [1.07, 2.27]) were predictive of CP/high-risk diagnosis. Although after adjustment for sex, GA, birth weight, and growth restriction, GA (aOR = 0.67; p = 0.068, 95% CI [0.44, 1.03]) and writhing GMs (aOR = 1.44; p = 0.087, 95% CI [0.95, 2.20]) were not significant, a strong trend still persisted. The HNNE scores significantly correlated with both the HINE evaluation (rs = 0.43, p < 0.001, 95% CI [0.31, 0.56]) and fidgety GMs (rs = −0.10, p = 0.012, 95% CI [−0.32, −0.04]). Linear regression confirmed the HNNE was highly predictive of the HINE (correlation coefficient 0.82; p < 0.001, 95% CI [0.48, 1.15]). Writhing GMs did not significantly correlate with either fidgety GMs (p = 0.723, 95% CI [−0.12, 0.17]) or the HINE (p = 0.173, 95% CI [−0.24, 0.04]). Conclusions: Abnormal writhing GMs in the neonatal period were non-significantly associated with early CP/high-risk diagnoses in extremely preterm infants in a multivariate analysis. Additionally, the HNNE significantly correlated with both fidgety GMs and the HINE. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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10 pages, 660 KiB  
Article
A Social Business Model of Early Intervention and Rehabilitation for People with Disability in Rural Bangladesh
by Mahmudul Hassan Al Imam, Manik Chandra Das, Israt Jahan, Mohammad Muhit, Delwar Akbar, Nadia Badawi and Gulam Khandaker
Brain Sci. 2022, 12(2), 264; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci12020264 - 14 Feb 2022
Cited by 3 | Viewed by 2616
Abstract
Background: Despite the high burden of childhood disability in low-and middle-income countries (LMICs), the opportunity for early intervention and rehabilitation is very limited. Studies have found that community-based rehabilitation service is effective for children with cerebral palsy (CP); however, such services are not [...] Read more.
Background: Despite the high burden of childhood disability in low-and middle-income countries (LMICs), the opportunity for early intervention and rehabilitation is very limited. Studies have found that community-based rehabilitation service is effective for children with cerebral palsy (CP); however, such services are not readily available in LMICs, and services run by non-profit organisations on external funding are often not sustainable. In this study, we report the lesson learnt in establishing a social business model of early intervention and rehabilitation services for children with CP and adults with disabilities in a rural subdistrict of Bangladesh. Methods: Case study of a rural early intervention and rehabilitation centre (i.e., the model centre) implemented between May 2018 and September 2019. An economic evaluation incorporating gross margin analysis along with descriptive statistics was performed to assess the social business potentials of the model centre. Results: The establishment of this model centre cost ~5955 USD with an average monthly running cost of ~994 USD. During the 17 months study period, 7038 therapy sessions (average eight sessions per patient) were offered to 862 patients with musculoskeletal and neurological disorders. The most common clinical presentations were low back pain (35.6%; n = 307). Six percent (n = 52) of the attendees were children with CP (mean (SD) age 6.3 (4.0) years; 35.7% (n = 19) were female), who received 1392 sessions, on average 27 sessions per child. The centre reached the break-even point at the 13th month and remained profitable for the next 4 months of the study period. An average session fee of 2.2 USD resulted in a gross margin of -1458 USD and 1940 USD in 2018 and 2019, respectively. Revenue to cost ratios for the 2 years were 0.27:1 and 0.51:1 while average rates of return were −41.4% and 10.1%, respectively. Sensitivity analysis revealed that session numbers including 5000, 6000, 7000, 8000, 9000, and 10,000 were required to break even at the session fees of 3.0, 2.50, 2.0, 2.0, 1.5, and 1.5 USD, respectively. Conclusion: Our social business model of an early intervention and rehabilitation service provides evidence of enhancing access to services for children with CP as well as adults with disabilities while ensuring the sustainability of the services in rural Bangladesh. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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18 pages, 577 KiB  
Article
Outcome of Community-Based Early Intervention and Rehabilitation for Children with Cerebral Palsy in Rural Bangladesh: A Quasi-Experimental Study
by Tasneem Karim, Mohammad Muhit, Israt Jahan, Claire Galea, Catherine Morgan, Hayley Smithers-Sheedy, Nadia Badawi and Gulam Khandaker
Brain Sci. 2021, 11(9), 1189; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci11091189 - 10 Sep 2021
Cited by 13 | Viewed by 3099
Abstract
We evaluated the outcome of a community-based early intervention and habilitation for children with cerebral palsy (CP) in Bangladesh. Children registered on the Bangladesh CP Register (BCPR) were recruited in two groups for this study: Group A received a comprehensive six-month long community-based [...] Read more.
We evaluated the outcome of a community-based early intervention and habilitation for children with cerebral palsy (CP) in Bangladesh. Children registered on the Bangladesh CP Register (BCPR) were recruited in two groups for this study: Group A received a comprehensive six-month long community-based caregiver-led intervention program at the “Shishu Shorgo” (Bengali title, which translates to ‘Children’s Heaven’) Early Intervention and Rehabilitation Centres developed to support participants from the BCPR. Group B received standard care. A quasi-experimental study was conducted. Data were obtained at baseline, at the end of the program (i.e., 6 months), and at a 12-month follow-up. Outcome measures for children included gross motor functional measure (GMFM-66), Communication Function Classification System (CFCS), and Viking Speech Scale (VSS) and, for adult caregivers, the depression, anxiety, and stress scale (DASS 21). Between October 2016 and March 2017, 156 children with CP were recruited (77 in Group A and 79 in Group B). The total score of GMFM-66, CFCS level, and VSS level significantly improved statistically in Group A (p < 0.05 for all) and deteriorated in Group B (p < 0.001, p = 0.095, p = 0.232). The intervention showed promising outcomes particularly for children with CP under five years of age. There is a need for caregiver-led community-based programs for children with CP in LMICs. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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14 pages, 463 KiB  
Article
Age of Diagnosis, Fidelity and Acceptability of an Early Diagnosis Clinic for Cerebral Palsy: A Single Site Implementation Study
by Anna te Velde, Esther Tantsis, Iona Novak, Nadia Badawi, Jane Berry, Prue Golland, Johanna Korkalainen, Robyn McMurdo, Ronda Shehata and Catherine Morgan
Brain Sci. 2021, 11(8), 1074; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci11081074 - 16 Aug 2021
Cited by 15 | Viewed by 3524
Abstract
Cerebral palsy (CP) diagnosis is historically late, at between 12 and 24 months. We aimed to determine diagnosis age, fidelity to recommended tests and acceptability to parents and referrers of an early diagnosis clinic to implement a recent evidence-based clinical guideline for the [...] Read more.
Cerebral palsy (CP) diagnosis is historically late, at between 12 and 24 months. We aimed to determine diagnosis age, fidelity to recommended tests and acceptability to parents and referrers of an early diagnosis clinic to implement a recent evidence-based clinical guideline for the early diagnosis of CP. A prospective observational case series of infants <12 months with detectable risks for CP attending our clinic was completed with data analysed cross-sectionally. Infants had a high risk of CP diagnosis at a mean age of 4.4 (standard deviation [SD] 2.3) months and CP diagnosis at 8.5 [4.1] months. Of the 109 infants seen, 57% had a diagnosis of CP or high risk of CP, showing high specificity to our inclusion criteria. Parent and referrer acceptability of the clinic was high. Paediatricians had the highest rate of referral (39%) followed by allied health (31%), primary carer (14%) and other health workers (16%). Fidelity to the guideline was also high. All infants referred <5 mths had the General Movements Assessment (GMA) and all except one had the Hammersmith Infant Neurological Examination (HINE) administered. N = 92 (84%) of infants seen had neuroimaging, including n = 53 (49%) who had magnetic resonance imaging (MRI), showing recommended tests are feasible. Referral to CP-specific interventions was at 4.7 [3.0] months, sometimes before referral to clinic. Clinicians can be confident CP can be diagnosed well under 12 months using recommended tools. This clinic model is acceptable to parents and referrers and supports access to CP-specific early interventions when they are likely to be most effective. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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15 pages, 266 KiB  
Article
Predictors of Rehabilitation Service Utilisation among Children with Cerebral Palsy (CP) in Low- and Middle-Income Countries (LMIC): Findings from the Global LMIC CP Register
by Mahmudul Hassan Al Imam, Israt Jahan, Mohammad Muhit, Denny Hardianto, Francis Laryea, Amir Banjara Chhetri, Hayley Smithers-Sheedy, Sarah McIntyre, Nadia Badawi and Gulam Khandaker
Brain Sci. 2021, 11(7), 848; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci11070848 - 25 Jun 2021
Cited by 16 | Viewed by 2926
Abstract
Background: We assessed the rehabilitation status and predictors of rehabilitation service utilisation among children with cerebral palsy (CP) in selected low- and middle-income countries (LMICs). Methods: Data from the Global LMIC CP Register (GLM-CPR), a multi-country register of children with CP aged <18 [...] Read more.
Background: We assessed the rehabilitation status and predictors of rehabilitation service utilisation among children with cerebral palsy (CP) in selected low- and middle-income countries (LMICs). Methods: Data from the Global LMIC CP Register (GLM-CPR), a multi-country register of children with CP aged <18 years in selected countries, were used. Descriptive and inferential statistics (e.g., adjusted odds ratios) were reported. Results: Between January 2015 and December 2019, 3441 children were registered from Bangladesh (n = 2852), Indonesia (n = 130), Nepal (n = 182), and Ghana (n = 277). The proportion of children who never received rehabilitation was 49.8% (n = 1411) in Bangladesh, 45.8% (n = 82) in Nepal, 66.2% (n = 86) in Indonesia, and 26.7% (n = 74) in Ghana. The mean (Standard Deviation) age of commencing rehabilitation services was relatively delayed in Nepal (3.9 (3.1) year). Lack of awareness was the most frequently reported reason for not receiving rehabilitation in all four countries. Common predictors of not receiving rehabilitation were older age at assessment (i.e., age of children at the time of the data collection), low parental education and family income, mild functional limitation, and associated impairments (i.e., hearing and/or intellectual impairments). Additionally, gender of the children significantly influenced rehabilitation service utilisation in Bangladesh. Conclusions: Child’s age, functional limitation and associated impairments, and parental education and economic status influenced the rehabilitation utilisation among children with CP in LMICs. Policymakers and service providers could use these findings to increase access to rehabilitation and improve equity in rehabilitation service utilisation for better functional outcome of children with CP. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)

Review

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18 pages, 1309 KiB  
Review
Epidemiology of Cerebral Palsy among Children and Adolescents in Arabic-Speaking Countries: A Systematic Review and Meta-Analysis
by Sami Mukhdari Mushta, Catherine King, Shona Goldsmith, Hayley Smithers-Sheedy, Al-Mamoon Badahdah, Harunor Rashid, Nadia Badawi, Gulam Khandaker and Sarah McIntyre
Brain Sci. 2022, 12(7), 859; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci12070859 - 29 Jun 2022
Cited by 11 | Viewed by 3404
Abstract
Background: Studies on cerebral palsy among children and adolescents in Arabic-speaking countries are scarce. In this systematic review, we aimed to describe the epidemiology of cerebral palsy among children and adolescents in Arabic-speaking countries in terms of prevalence, risk factors, motor types, and [...] Read more.
Background: Studies on cerebral palsy among children and adolescents in Arabic-speaking countries are scarce. In this systematic review, we aimed to describe the epidemiology of cerebral palsy among children and adolescents in Arabic-speaking countries in terms of prevalence, risk factors, motor types, and rehabilitation. Methods: Six key bibliographic databases were searched for relevant literature published to 17 July 2021. Titles and abstracts were screened for potential inclusion and two independent reviewers screened the full texts of potential articles following pre-defined inclusion/exclusion criteria. The included studies were evaluated independently by three reviewers. The risk of bias was assessed, and data were extracted and analysed. Results: A total of 32 studies from 7 countries met our inclusion criteria. The prevalence of cerebral palsy in Arabic-speaking countries was 1.8/1000 live births (95% CI: 1.2–2.5). Spastic cerebral palsy was the most common motor type, representing 59.8% (95% CI: 46.2–72.7) of pooled estimates. This included children with spastic quadriplegia, diplegia, and hemiplegia; 25.1% (95% CI: 18.2–32.8), 16.2% (95% CI: 11.4–23.3), and 10.4% (95% CI: 7.3–13.8), respectively. Consanguinity was high and represented 37.7% (95% CI: 29.3–46.6). Only one included study reported the types of rehabilitation received (e.g., physiotherapy and assistance devices). Conclusions: This paper provides a summary of the epidemiology of cerebral palsy in Arabic-speaking countries and highlights areas for future research. There is still a substantial knowledge gap on the epidemiology of cerebral palsy in these regions. Countries in the Arab region should follow examples of countries that have successfully established cerebral palsy registries to generate evidence on epidemiology of cerebral palsy and opportunities for prevention. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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13 pages, 582 KiB  
Review
Early Diagnosis of Cerebral Palsy in Low- and Middle-Income Countries
by Arrabella R. King, Mahmudul Hassan Al Imam, Sarah McIntyre, Catherine Morgan, Gulam Khandaker, Nadia Badawi and Atul Malhotra
Brain Sci. 2022, 12(5), 539; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci12050539 - 23 Apr 2022
Cited by 3 | Viewed by 5128
Abstract
Cerebral palsy describes a group of permanent disorders of movement, motor function and posture that occur due to non-progressive insults to the developing brain. Most of the information concerning the early diagnosis of cerebral palsy originates from studies conducted in high-income countries. In [...] Read more.
Cerebral palsy describes a group of permanent disorders of movement, motor function and posture that occur due to non-progressive insults to the developing brain. Most of the information concerning the early diagnosis of cerebral palsy originates from studies conducted in high-income countries. In this scoping review, we aimed to explore the tools used in low- and middle-income countries for the early diagnosis of cerebral palsy. A systematic search was conducted using OVID Medline and PubMed databases. “Early diagnosis” was defined as diagnosis prior to 12 months of age, and low- and middle-income countries were classified according to the World Bank classification system. We identified nine studies on the early diagnosis of cerebral palsy from low- and middle-income countries. The tools featured (n = number of studies) were: General Movement Assessment (6), neonatal magnetic resonance imaging (3), Hammersmith Neonatal Neurological Examination (2), Hammersmith Infant Neurological Examination (1) and cranial ultrasound (1). We found a paucity of published literature on the early diagnosis of cerebral palsy from low- and middle-income countries. Further research is needed to determine the tools that are accurate and feasible for use in low-resource settings, particularly since cerebral palsy is more prevalent in these areas. Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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Other

6 pages, 192 KiB  
Protocol
Neurophysiological Assessments of Brain and Spinal Cord Associated with Lower Limb Functions in Children with Cerebral Palsy: A Protocol for Systematic Review and Meta-Analysis
by Leonard Ubalde and Jing-Nong Liang
Brain Sci. 2021, 11(5), 628; https://0-doi-org.brum.beds.ac.uk/10.3390/brainsci11050628 - 13 May 2021
Viewed by 1917
Abstract
Background: Task-dependent neurophysiological adaptations in people with cerebral palsy have been examined using various techniques such as functional magnetic resonance imaging, peripheral nerve stimulation in order to assess H-reflexes, and transcranial magnetic stimulation. This activity-dependent plasticity is hypothesized to improve specific gross motor [...] Read more.
Background: Task-dependent neurophysiological adaptations in people with cerebral palsy have been examined using various techniques such as functional magnetic resonance imaging, peripheral nerve stimulation in order to assess H-reflexes, and transcranial magnetic stimulation. This activity-dependent plasticity is hypothesized to improve specific gross motor function in individuals with cerebral palsy. Although these adaptations have been examined extensively, most studies examined tasks utilizing the upper limbs. The aim of this review is to assess the neurophysiological adaptations of the central nervous system in individuals with cerebral palsy during lower limb functional tasks. Methods: A systematic review and meta-analysis will be conducted to evaluate the neurophysiological changes in the brain and spinal cord associated with lower extremity tasks in individuals with cerebral palsy. We will search within PubMed, MEDLINE, Embase, PsychINFO, and CINAHL using a predetermined search string to identify and evaluate relevant studies. Two independent reviewers will screen these studies against our inclusion criteria and risks of bias, and will extract the data from each study. A third reviewer will be used to resolve any disagreement regarding the inclusion of a study between reviewers. Randomized controlled trials as well as cross-sectional studies published in English 10 years before May 2021 that investigate the neurophysiological adaptations in the brain and spinal cord in people with cerebral palsy will be included if they meet the eligibility criteria. Primary outcomes will include scalar values of fractional anisotropy (FA), H-reflex gains or measures of amplitude, as well as motor cortex (M1) cortical excitability as measured by transcranial magnetic stimulation. Discussion: Since no identifiable data will be involved in this study, no ethical approval is required. Our results will provide insight into the neurophysiological adaptations in children with cerebral palsy, which will be useful in guiding directions for clinical decision making and future development of targeted interventions in pediatrics rehabilitation for children with cerebral palsy. Systematic review registration: The protocol for this systematic review is registered with the International Prospective Register of Systematic Reviews (PROSPERO; registration number: CRD42020215902). Full article
(This article belongs to the Special Issue Early Detection and Early Intervention Strategies for Cerebral Palsy in Low and High Resource Settings)
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