Screening tools for early identification of children with developmental delay in low- and middle-income countries: a systematic review

Objective To systematically review, identify and report the screening tools used for early identification of developmental delay in low- and middle-income countries.Design Systematic review.Data sources Four bibliographic databases: Medline (1946 to 13 July 2020), Embase (1974 to 13 July 2020), Scopus (1823 to 11 July 2020) and PsycINFO (1987 to July week 1 2020).Eligibility criteria Peer-reviewed original articles published in English addressing validated culturally sensitive developmental screening tools among children aged <5 years were included in this review.Data extraction and synthesis One author (CK, medical librarian) developed the search strategy. Three authors conducted the database search (phase I: CK; phase II: IJ and MKI). Three authors (TF, IJ and MKI) independently screened the title and abstracts. TF, MKI and GK independently performed the full-text review of the screened articles. During each step of the study selection process, disagreements were resolved through discussion. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement was used to guide the systematic review. Data extraction and analysis were performed using MS Excel. Meta-analysis was not possible due to heterogeneity of the study findings.Results We identified 3349 articles, of which 18 studies from 10 countries, reporting 16 screening tools, were selected for qualitative synthesis. Six cultural contexts were explored. Twelve general, two motor and two speech-language tools were identified. Seven of them found to be parent-completed ones. Five screening tools (American Speech-Language and Hearing Association, Guide for Monitoring Child Development, Infant Neurological International Battery, New Delhi-Development Screening Questionnaire and Woodside Screening Technique) reported relatively higher sensitivity (82.5%–100%) and specificity (83%–98.93%).Conclusions Limited number of culturally sensitive developmental screening tools were validated for children aged <5 years in low- and middle-income countries. Revising existing screening tools in different ethnic and cultural settings and subsequent validation with normative value should be a research priority

Screening tools for early identification of children with developmental delay in low- and middle-income countries: A systematic review

OBJECTIVE: To systematically review, identify and report the screening tools used for early identification of developmental delay in low- and middle-income countries. DESIGN: Systematic review. DATA SOURCES: Four bibliographic databases: Medline (1946 to 13 July 2020), Embase (1974 to 13 July 2020), Scopus (1823 to 11 July 2020) and PsycINFO (1987 to July week 1 2020). ELIGIBILITY CRITERIA: Peer-reviewed original articles published in English addressing validated culturally sensitive developmental screening tools among children aged <5 years were included in this review. DATA EXTRACTION AND SYNTHESIS: One author (CK, medical librarian) developed the search strategy. Three authors conducted the database search (phase I: CK; phase II: IJ and MKI). Three authors (TF, IJ and MKI) independently screened the title and abstracts. TF, MKI and GK independently performed the full-text review of the screened articles. During each step of the study selection process, disagreements were resolved through discussion. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement was used to guide the systematic review. Data extraction and analysis were performed using MS Excel. Meta-analysis was not possible due to heterogeneity of the study findings. RESULTS: We identified 3349 articles, of which 18 studies from 10 countries, reporting 16 screening tools, were selected for qualitative synthesis. Six cultural contexts were explored. Twelve general, two motor and two speech-language tools were identified. Seven of them found to be parent-completed ones. Five screening tools (American Speech-Language and Hearing Association, Guide for Monitoring Child Development, Infant Neurological International Battery, New Delhi-Development Screening Questionnaire and Woodside Screening Technique) reported relatively higher sensitivity (82.5%-100%) and specificity (83%-98.93%). CONCLUSIONS: Limited number of culturally sensitive developmental screening tools were validated for children aged <5 years in low- and middle-income countries. Revising existing screening tools in different ethnic and cultural settings and subsequent validation with normative value should be a research priority

Population-based surveillance of children with cerebral palsy enables early diagnosis and intervention

The 2019 Global Burden of Disease Study suggests that there are an estimated 50 million people with cerebral palsy (CP) globally.1 The burden of CP is substantially higher in low- and middle-income countries (LMICs) compared with high-income countries (HICs).2 Moreover, an overrepresentation of severe functional impairments is observed among children with CP in LMICs, probably due to delayed diagnosis and lack of access to early intervention.2 The limited service availability and shortage of rehabilitation service providers in LMICs is a major challenge.

Additional costs identified by participants in extension and general open-ended questions.

Additional costs identified by participants in extension and general open-ended questions.</p

Epidemiology of cerebral palsy in Sumba Island, Indonesia

Aim: To define the epidemiology, clinical characteristics, and rehabilitation status of children with cerebral palsy (CP) in Sumba Island, Indonesia. Method: A community-based key informant method survey among children (aged <18y) with CP was conducted between March and August 2017. Children with suspected CP underwent detailed neurodevelopmental assessment by a multidisciplinary medical team. Socio-demographic characteristics, aetiology, motor type, motor severity, associated impairments, educational, and rehabilitation status were documented. Results: There were 130 children with clinically confirmed CP. The mean age at assessment was 8 years 11 months and 43.8% (n=57) of the children were female. The mean age at CP diagnosis was 6 years 5 months. Of these children, 46.9% (n=61) had post-neonatally acquired CP, most frequently because of vaccine-preventable infectious encephalopathy (73.8%, n=45). In total, 80.8% (n=105) had a predominantly spastic motor type of CP and 83.8% (n=109) were classified in Gross Motor Functional Classification System levels III to V. A total of 77.7% (n=101) had at least one associated impairment (speech 77.5%, intellectual 29.2%, visual 13.8%, hearing 20.0%, and epilepsy 13.5%). And 66.2% (n=86) had never received rehabilitation services. Interpretation: Post-neonatally acquired CP was common in this setting. Addressing preventable post-neonatally acquired risk factors for CP should be a public health priority. Earlier identification and diagnosis of CP would also provide new opportunities for early intervention and targeted rehabilitation services

Nutritional status of children with cerebral palsy in remote Sumba Island of Indonesia: A community-based key informants study

Aim: To assess the burden and underlying factors of malnutrition among children with cerebral palsy (CP) in the remote Sumba Island, Indonesia. Methods: This is a community-based key informant (KI) method survey of children with CP in the southwest regency of Sumba Island, Indonesia (child population∼152 471). Children with suspected CP identified by trained community volunteers (KIs) underwent a detailed neurodevelopmental assessment by a multidisciplinary medical assessment team to confirm diagnosis of CP. Anthropometric measurements were taken, z scores were calculated, and nutritional status was determined following the World Health Organization guideline. Descriptive analyses were performed, multivariable linear regression model was fitted to identify potential predictors of malnutrition. Results: One hundred and thirty children with CP aged <18 years were included in the study. The majority were severely underweight (78.8%) and severely stunted (85.9%). Severe malnutrition was overrepresented among young children, children who had spastic tri/quadriplegia, Gross Motor Function Classification System levels III–V, had at least one associated impairment, speech impairment and/or swallowing difficulties. Age and presence of visual impairment were found to be significantly associated with weight-for-age z score (i.e., underweight) whereas, epilepsy was significantly associated with the height-for-age z score (i.e., stunting) when adjusted for other covariates. Conclusions: The substantial-high burden of malnutrition among children with CP in remote Sumba Island highlights their vulnerability to poor health-related outcomes. There is an urgent need for nutritional rehabilitation services to avert such consequences among children with CP in low and middle-income countries like Indonesia.Implications for rehabilitation Malnutrition among children with cerebral palsy (CP) is alarmingly high in the remote Sumba island of Indonesia. Children with severe gross motor function limitations or spastic tri/quadriplegia had the most severe malnutrition. There is an urgent need for rehabilitation services including nutrition and feeding interventions for children with CP in low and middle-income countries like Indonesia

Epidemiology of cerebral palsy in low- and middle-income countries: Preliminary findings from an international multi-centre cerebral palsy register

Aim: To describe the epidemiology of cerebral palsy (CP) in children from low- and middle-income countries (LMICs) using data from the Global Low- and Middle-Income Country CP register (GLM-CPR). Method: The GLM-CPR is a multi-country initiative that combines and compares data from children with CP (<18y) in LMICs. Children with CP are registered after detailed neurodevelopmental assessment by a multidisciplinary medical team using a harmonized protocol. Data are collected on agreed core variables. Descriptive analyses are completed to report findings from participating countries. Results: Between January 2015 and May 2019, 2664 children were recruited from Bangladesh, Nepal, Indonesia, and Ghana (mean age [SD] at assessment: 7y 8mo [4y 8mo], 95% confidence interval 7y 6mo−7y 11mo; male [n=1615] 60.6%, female [n=1049] 39.4%). Overall, 86.6% children acquired CP prenatally and perinatally (e.g. preterm birth, birth asphyxia, neonatal encephalopathy). Median age at CP diagnosis was 3 years. Moreover, 79.2% children had spastic CP and 73.3% were classified in Gross Motor Function Classification System levels III to V. Notably, 47.3% of children never received rehabilitation services (median age at receiving rehabilitation services was 3y; 12.7% received assistive devices) and 75.6% of school-age children had no access to education. Interpretation: Population-based data show that the proportion of severe cases of CP is very high in LMICs. Children with CP in LMICs lack access to rehabilitation and educational services and a large proportion of children have potentially preventable risk factors, for example, birth asphyxia and neonatal infections. Delayed diagnosis, severe motor impairments, and lack of rehabilitation in most children call for urgent action to identify preventive opportunities and promote early diagnosis and intervention for children with CP in LMICs. What this paper adds The proportion of severe cases of cerebral palsy (CP) is very high in rural low- and middle-income countries (LMICs). Children with CP in LMICs lack access to rehabilitation and educational services. A large proportion of children with CP in LMICs have potentially preventable risk factors

Adult sociodemographic characteristics.

The most recent cost estimates of cerebral palsy (CP) in Australia did not include out-of-pocket costs for families. This study aimed to: 1) describe and estimate out-of-pocket costs for people with CP and their families by age and gross motor function classification system (GMFCS) level; 2) measure financial distress. A cross-sectional quantitative survey design was used with qualitative approaches to analyse open-ended questions. A CP-specific out-of-pocket costs survey was co-designed with people with lived experience. Adults with CP and carers were recruited from Australian population-based CP Registers and via social media. Sociodemographic variables were analysed descriptively and median (IQR) expenses for health, assistive technology, personal care, housing, occupation, transport, leisure, respite and holidays, by age (0–6; 7–17; 18 years +) and gross motor function [GMFCS level I-II vs III-V] were calculated. The In Charge Financial Distress/Financial Wellbeing Scale measured financial distress. Regression analyses were conducted to investigate costs and financial distress. Additional out-of-pocket costs itemised in open-ended questions were charted. Comments were thematically analysed using the framework approach. 271 surveys were completed for children 0–6 years (n = 47), children/adolescents 7–17 years (n = 124) and adults (n = 100). 94% of participants had out-of-pocket costs associated with CP, with an overall annual median of $4,460 Australian dollars (IQR$11,955). After controlling for income, private insurance and disability funding, the GMFCS III-V group had costs two times higher than the GMFCS I-II group (2.01; 95% CI 1.15–3.51). Age was not significantly associated with costs. 36% of participants had high to overwhelming financial distress; this was not associated with age or GMFCS level after controlling for financial factors. Families had several additional disability costs. Open-ended responses revealed experiences of financial concern were influenced by funding scheme experiences, reduced income, uncertainty, access to support networks and an inability to afford CP-related costs. Cost estimates and financial distress indicators should inform policy, funding and clinical decisions when planning interventions to support people with CP and their families.</div

Annual costs across sub-types.

The most recent cost estimates of cerebral palsy (CP) in Australia did not include out-of-pocket costs for families. This study aimed to: 1) describe and estimate out-of-pocket costs for people with CP and their families by age and gross motor function classification system (GMFCS) level; 2) measure financial distress. A cross-sectional quantitative survey design was used with qualitative approaches to analyse open-ended questions. A CP-specific out-of-pocket costs survey was co-designed with people with lived experience. Adults with CP and carers were recruited from Australian population-based CP Registers and via social media. Sociodemographic variables were analysed descriptively and median (IQR) expenses for health, assistive technology, personal care, housing, occupation, transport, leisure, respite and holidays, by age (0–6; 7–17; 18 years +) and gross motor function [GMFCS level I-II vs III-V] were calculated. The In Charge Financial Distress/Financial Wellbeing Scale measured financial distress. Regression analyses were conducted to investigate costs and financial distress. Additional out-of-pocket costs itemised in open-ended questions were charted. Comments were thematically analysed using the framework approach. 271 surveys were completed for children 0–6 years (n = 47), children/adolescents 7–17 years (n = 124) and adults (n = 100). 94% of participants had out-of-pocket costs associated with CP, with an overall annual median of $4,460 Australian dollars (IQR$11,955). After controlling for income, private insurance and disability funding, the GMFCS III-V group had costs two times higher than the GMFCS I-II group (2.01; 95% CI 1.15–3.51). Age was not significantly associated with costs. 36% of participants had high to overwhelming financial distress; this was not associated with age or GMFCS level after controlling for financial factors. Families had several additional disability costs. Open-ended responses revealed experiences of financial concern were influenced by funding scheme experiences, reduced income, uncertainty, access to support networks and an inability to afford CP-related costs. Cost estimates and financial distress indicators should inform policy, funding and clinical decisions when planning interventions to support people with CP and their families.</div

Annual costs across cost type.

The most recent cost estimates of cerebral palsy (CP) in Australia did not include out-of-pocket costs for families. This study aimed to: 1) describe and estimate out-of-pocket costs for people with CP and their families by age and gross motor function classification system (GMFCS) level; 2) measure financial distress. A cross-sectional quantitative survey design was used with qualitative approaches to analyse open-ended questions. A CP-specific out-of-pocket costs survey was co-designed with people with lived experience. Adults with CP and carers were recruited from Australian population-based CP Registers and via social media. Sociodemographic variables were analysed descriptively and median (IQR) expenses for health, assistive technology, personal care, housing, occupation, transport, leisure, respite and holidays, by age (0–6; 7–17; 18 years +) and gross motor function [GMFCS level I-II vs III-V] were calculated. The In Charge Financial Distress/Financial Wellbeing Scale measured financial distress. Regression analyses were conducted to investigate costs and financial distress. Additional out-of-pocket costs itemised in open-ended questions were charted. Comments were thematically analysed using the framework approach. 271 surveys were completed for children 0–6 years (n = 47), children/adolescents 7–17 years (n = 124) and adults (n = 100). 94% of participants had out-of-pocket costs associated with CP, with an overall annual median of $4,460 Australian dollars (IQR$11,955). After controlling for income, private insurance and disability funding, the GMFCS III-V group had costs two times higher than the GMFCS I-II group (2.01; 95% CI 1.15–3.51). Age was not significantly associated with costs. 36% of participants had high to overwhelming financial distress; this was not associated with age or GMFCS level after controlling for financial factors. Families had several additional disability costs. Open-ended responses revealed experiences of financial concern were influenced by funding scheme experiences, reduced income, uncertainty, access to support networks and an inability to afford CP-related costs. Cost estimates and financial distress indicators should inform policy, funding and clinical decisions when planning interventions to support people with CP and their families.</div