Barriers to access and utilization of healthcare by children with neurological impairments and disability in low-and middle-income countries: a systematic review

Version Changes

Revised. Amendments from Version 1

Introduction: We have broadened the introduction section to include the International Classification of Functioning, Disability and Health (ICF) framework to improve the justification of the need for this systematic review.  Methods: Following the reviewers' recommendations, we have expanded the search terms to include the terms “OR cerebral palsy” to obtain studies on motor impairments that might have been missed out in the initial searches. We also have explained the checks that were conducted to ensure there was an agreement between the reviewers at the screening stage. In the analysis, more details are provided on the process of identification and classification of barriers limiting access to care by children with neurological impairments and disabilities using data obtained from the primary reports.  Results: The total number of studies included in the Results section of the revised paper is 20 compared to 16 reports analysed in the previous version. We also have a higher number of titles/abstracts (N=3,258) compared to the number (N=3,074) reported in the former draft. We also present the quality of the studies report in this section. We have revised Figure 3 to reflect the effect of the expanded searches in the study selection process. We moved Table 1 from the Methods to Table 3 in the Results, and thereafter, renamed the other tables appropriately both in the text and the captions. We have revised Tables 1, 2 and 4 to include information obtained from the additional reports identified by the expanded searches. As suggested by the reviewers, Figure 4 was removed from the paper in the revised manuscript because it was unnecessary.  Discussion: We have revised the discussion section to incorporate interpretation of all studies identified and included in this systematic review. We have replaced the terms “anti-epileptic drugs” with “antiseizure medications” throughout the paper.

Abstract

Background: Neurological impairments (NI) and disability are common among older children in low-and middle-income countries (LMICs). We conducted a systematic review to examine the barriers limiting access and utilization of biomedical and rehabilitative care by children and adolescents with NI in LMICs.

Methods: We searched PubMed, Latin America and Caribbean Health Sciences Literature, Global Index Medicus, and Google Scholar for studies published between 01/01/1990 and 14/11/2019 to identify relevant studies. We included all studies reporting on barriers limiting access and utilization of preventive, curative, and rehabilitative care for children aged 0-19 years with NI in five domains: epilepsy, and cognitive, auditory, visual, and motor function impairment. Data from primary studies were synthesized using both qualitative and quantitative approaches.

Results: Our literature searches identified 3,258 reports of which 20 were included in the final analysis. Fifteen studies (75.0%) originated from diverse settings in sub-Saharan Africa (SSA). Factors limiting access and utilization of healthcare services in >50% of the studies were: financial constraints (N=17, 85.0%), geographical and physical inaccessibility (N=14, 70.0%), inadequate healthcare resources (N=14, 70.0%), prohibitive culture and beliefs (N=12, 60.0%), and inadequate education/awareness (N=11, 55.0%). Factors reported in <50% of the studies included competing domestic roles (N=4, 20%) and a lack of confidentiality for personal information (N=2, 10.0%). Very few reports were identified from outside Africa preventing a statistical analysis by continent and economic level.

Conclusions: Financial constraints, geographic and physical inaccessibility, and inadequate healthcare resources were the most common barriers limiting access and utilization of healthcare services by children with NI in LMICs.

PROSPERO registration: CRD42020165296 (28/04/2020)

Introduction

Globally, at least 300 million children live with some form of neurological impairment (NI) or disability, of which >90% originate from low- and middle-income countries (LMICs) ( Olusanya et al., 2020). The International Classification of Functioning, Disability and Health, ICF ( WHO, 2001), and the International classification of functioning, disability and health: children and youth version: ICF-CY ( WHO, 2007), provides a common language and framework to guide research and care of children and youth with NI and disabilities, globally. By integrating both the medical and social models of health and disease (biopsychosocial model ( Engel, 1977)), the ICF conceptualizes child functioning as an interaction between the health conditions, environmental factors, and personal factors. Childhood NI and disability not only affects the health and education of the affected individual, but also, directly and indirectly, impacts the family, community, and the society ( Patel et al., 2013).

Considering the medical component of the ICF framework, known risk factors of NI and disability in children include adverse pregnancy and birth events such as prematurity and neonatal encephalopathy ( Abuga et al., 2021a), infections of the brain including malaria and meningitis, malnutrition, trauma, and poverty ( Banks et al., 2017; Scherzer et al., 2012). The affected children, especially those with moderate, severe or multiple disabilities, are vulnerable to infections and accidents which might necessitate hospitalization and subsequent rehabilitation ( Moreau et al., 2013). Unfortunately, there is a high treatment gap for NI and disability in LMICs ranging between 50–90% ( Patel et al., 2013). The most significant factors limiting the implementation of effective interventions in LMICs include (i) insufficient evidence on the delivery of interventions (ii) inadequate identification of the affected children, and (iii) a shortage of skilled professionals ( Patel et al., 2009). Besides, numerous factors limit access or utilization of available preventive, curative, and rehabilitative services by the affected children ( WHO, 2011).

Primary preventive services such as nutritional supplementation and immunization have an established role in the prevention of NI ( Groce et al., 2014). Vitamin A supplementation and early childhood immunization against measles, rubella, and poliomyelitis substantially reduce the risk of developing NI ( Maulik & Darmastadt, 2007), but also reduce morbidity in those that have NI. Secondary prevention involves early screening to identify those already with NI for treatment (or management) to alter the prognosis. Diagnostic tests such as the electroencephalogram (EEG) may help classify seizures and determine treatment ( Bassili et al., 2002). Tertiary and quaternary prevention includes treatment and rehabilitation, respectively, to prevent premature mortality, improve functioning, and quality of life. Corrective surgery for children with hearing impairments ( Roland et al., 2016) and antiseizure medications for children with epilepsy ( Mbuba et al., 2012) are widely documented curative/management options. Rehabilitative services to reduce activity limitation and to improve participation in respective communities include physiotherapy and occupational therapy. However, children from LMICs lack adequate access to the aforementioned continuum of healthcare due to multifarious barriers such as geographical inaccessibility, societal stigma, and financial constraints ( WHO, 2011).

A range of barriers, both from the consumer’s and provider’s perspectives, may hinder uptake of biomedical services by affected children in LMICs ( Figure 1). Poverty in families of children with NI and inadequate government funding limits the prioritization, availability, access, and quality of rehabilitative care ( Bright et al., 2018). Besides, prevailing cultural beliefs and societal perceptions shape caregivers’ perceptions especially on the aetiology of NI, which affects decisions about the alternatives of care, and ultimately the prognosis of neurodisability ( Zuurmond et al., 2019). A 2015 report published by the World Health Organization (WHO) further highlights that people with NI usually experience discrimination at the point of care, which may discourage subsequent seeking of appropriate services ( WHO, 2015). Measures such as decentralizing health systems have been proposed to bring services closer to the people and to reduce the geographical distance (distance decay) especially for rural-dwelling populations ( Saltman et al., 2007).

Figure 1. A modified socio-ecological model with layers of barriers limiting access/utilization of healthcare by children with neurodisability.

Some studies from LMICs have separately identified contextual factors that limit access and utilization of existing biomedical services by children with NI. The available evidence on barriers preventing the use of preventive, curative, and rehabilitative services by children with NI in LMICs remains fragmented. We, therefore, conducted a systematic review to collate and classify barriers limiting access and utilization of biomedical services by children and adolescents with NI in LMICs. Synthesized evidence from LMICs is required to inform policy and public health action to ensure equity in access and utilization of healthcare as enshrined in the agenda of the United Nation’s sustainable development goals ( Pettigrew et al., 2015; Tangcharoensathien et al., 2015).

Methods

Reporting guidelines

We used the National Health Service Centre for Review and Dissemination (CRD) recommendations ( Booth et al., 2010) and the preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines ( Abuga et al., 2021b; Moher et al., 2009) to conduct this systematic review. We registered a protocol with the International Prospective Register for Systematic Reviews (PROSPERO), registration ID CRD42020165296 (28 ^th April 2020).

Search strategy and inclusion criteria

We searched PubMed, Latin America and Caribbean Health Sciences Literature ( LILACs), Global Index Medicus, and Google Scholar databases for reports published between 01/01/1990 and 14/11/2019 to identify relevant reports. These searches covered a period through which the burden of NI and disability has significantly increased, globally ( Global Research on Developmental Disabilities Collaborators, 2018). The key search terms were ‘neurological impairment’ and ‘access’ or ‘utilization’ and ‘healthcare services’ as shown in Figure 2, with limits to human studies only.

Figure 2. Search terms used in the systematic review.

Eligible studies included those of: (i) children and adolescents aged 0–19 years; (ii) children or adolescents with NI in five domains namely, epilepsy, and impairments in cognitive, hearing, visual, and motor functions; (iii) assessing access and utilization of healthcare services including preventive, curative, and rehabilitative care; and (iv) LMICs as defined by the World Bank ( World-Bank, 2019). We excluded systematic reviews, reports, studies on adults, studies conducted in HICs, commentaries, and studies published in languages other than English.

Defining neurological impairment and disability

Neurological impairment was defined as a deficit of the central nervous system (CNS) resulting in functional limitation in five domains namely: epilepsy, and cognitive, hearing, visual, and motor impairments ( Mung'ala-Odera et al., 2006). Epilepsy was defined according to the International League Against Epilepsy’s (ILAE) guidelines as the presence of two or more unprovoked seizures occurring more than 24 hours apart within the previous 12 months ( Fisher et al., 2014; Thurman et al., 2011). A child with moderate or severe cognitive impairment refers to a child with a z-score below-2SD or -3SD, respectively based on neuropsychological scores standardized to the normal population. Moderate motor impairment is defined as a difficulty in holding objects, dressing and sitting upright, or ambulant only with help, while severely impaired children include those unable to walk or have no functional use of the hands ( WHO, 2011). Moderate hearing impairment refers to a 41–70dB loss in the best ear or difficulty in hearing with a hearing aid; severe impairment refers to greater than 70dB hearing loss or complete loss of hearing in the best ear ( WHO, 2020). A child with moderate vision impairment has a visual acuity poorer than 6/18 while those with visual acuity poorer than 6/60 meters are classified as having severe vision impairment ( WHO, 2019). However, there was a variation in the actual definitions used in the individual studies and most reports lacked information on NI severity. We, therefore, used these definitions as a formal guideline to verify the actual definitions used in the included reports.

Study selection, data extraction, and quality appraisal

Study selection was done in two phases. In the first phase, two reviewers (LM and JA) independently screened the reports identified by the searches by title and abstract for eligibility. The two reviewers compared the lists of the identified reports as to whether they met the predefined inclusion criteria and disagreements were resolved through consensus. In the second phase, both reviewers (LM and JA) examined the full-text of articles obtained from the first phase against the inclusion and exclusion criteria. Disagreements were resolved through consensus in discussions involving three reviewers (LM, JA and EC). We extracted data relevant for analysis using a pretested data extraction tool designed by the reviewers using guidelines from the PRISMA checklist. Extracted data included author details, study setting, study population, participant characteristics, the type of healthcare services sought, and barriers hindering access or utilization of the services by children with NI or disability. We assessed the quality of each study using the Joanna Briggs Institute (JBI) critical appraisal tools, which are distinct for cross-sectional, cohort, and qualitative studies ( Munn et al., 2014).

Synthesis of included reports

Eligible reports identified by the searches and the selection processes were both qualitative and quantitative in design. We, therefore, used a mixed methods review process to synthesize evidence from the eligible studies. The qualitative component of the analytic phase involved the identification and classification of the barriers limiting access to healthcare as described in the primary studies. We grouped all eligible studies based on the domain of NI investigated, and then classified the barriers identified in the primary studies in a three-step process. In step I, three reviewers (LM, EC and JA) independently identified the emerging categories in all the data retrieved from the primary articles. Single or multiple quotes in the primary articles were sufficient because the articles themselves did not similarly present these barriers. In stage II, the three reviewers held a joint meeting to present and discuss all the identified categories, each at a time. Some categories were merged, and others were created to include reviewers’ suggestions logically and comprehensively. In stage III, the reviewers held the final joint consensus meeting to review the previously created and discussed categories before writing the final report. This three-step process ensured that the classification process for the barriers identified in the primary articles was valid and reliable. We also identified verbatim excerpts from the eligible primary qualitative studies to represent the voices of the caregivers about their perceptions of the barriers to care for their disabled children. The quantitative component of the analytic phase primarily entailed determining the frequencies and proportions (percentages) of the identified barriers depending on the continent and economic level of the country of origin of the included primary studies. Services sought by children with NI were classified as preventive, curative, or rehabilitative.

Results

Search results

The database searches yielded 3,258 reports, of which 20 were eligible for the final analysis ( Figure 3). Most (75.0%) studies were conducted in Africa while the rest originated from Asia. Over 60% of these studies originated from lower-middle-income countries (LMIs) while the remainder (<40%) came from low-income countries (LIs) and upper-middle-income countries (UMICs), respectively. There were more community-based studies (55.0%) compared with hospital-based reports (45.0%), and more qualitative studies (50.0%) than quantitative studies (45.0%) ( Table 1). There were six (30.0%) studies on epilepsy, four (20.0%) studies on motor impairment/cerebral palsy, three (15.0%) studies on hearing impairment, two (10.0%) studies on cognitive impairment/intellectual disability, one (5.0%) study on vision impairment, and four (20.0%) studies in more than one domain of NI ( Table 2). The median quality score for quantitative studies (N=9) was 90% (range 37.5–90.0). Similarly, the median score for qualitative studies (N=10) was 90% (range 69.0–90.0).

Figure 3. PRISMA flow diagram of the study selection process.

Table 1. Classification of the studies included by economy level and study setting.

Classification		Number of Studies (n=20)	The total sample size of participants in the study (n=7,607)
Continent	Asia	5 (25.0%)	5,095
	Africa	15 (75.0%)	2,512
World Bank country income classification	Low-income	3 (15.0%)	631
	Lower-middle-income	13 (65.0%)	1,561
	Upper-middle-income	4 (20.0%)	5,415
Study design	Cohort	2 (10.0%)	5,137
	Cross-sectional	7 (35.0%)	1,794
	Qualitative	10 (50.0%)	629
	Mixed methods	1 (5.0%)	47
Setting	Community	11 (55.0%)	6,085
	Hospital	9 (45.0%)	1,522

Healthcare for children with neurological impairment

Rehabilitative services were sought in nine (45.0%) studies, treatment/management reported in seven (35.0%) studies, and preventive care reported in four (20.0%) studies ( Table 2). Children with motor impairments/cerebral palsy or intellectual disabilities sought strength training, ambulation and speech therapy care from occupational therapy, physical therapy and mental health departments ( Gobrial, 2012; He et al., 2017) while rehabilitation for hearing impairment included the provision of assistive hearing devices ( Merugumala et al., 2017). Antiseizure medication used in the management of seizures included phenobarbital ( Bassili et al., 2002; Mbuba et al., 2012), phenytoin ( Bassili et al., 2002), sodium valproate ( Bassili et al., 2002), and carbamazepine ( Bassili et al., 2002; Mbuba et al., 2012). Bassili and colleagues also identified the use of electroencephalogram (EEG) and computed tomography for the diagnosis or classification of epilepsy ( Bassili et al., 2002). None of the epilepsy reports identified the use of surgical services. Preventive services reported in three studies included the screening of HIV/AIDS ( Yousafzai et al., 2005), the provision of contraceptives for young adults with disabilities ( Burke et al., 2017), and screening services for children with hearing impairment ( Bright et al., 2017).

Table 2. General characteristics of studies eligible for inclusion in the systematic review.

First Author & year of Publication	Country	Study design	Domain studied	Type of services discussed
Alloh et al., 2009	Cote d’Ivore	Cross-sectional	Disabilities	Rehabilitative
Alrasheed et al., 2018	Sudan	Qualitative	Vision	Preventive
Bassili et al., 2002	Egypt	Cross-sectional	Epilepsy	Treatment/management
Burke et al., 2017	Senegal	Qualitative	Disabilities	Preventive
Bright et al., 2017	Malawi	Cohort	Hearing	Treatment/management
Carter et al., 2012	Kenya	Qualitative	Epilepsy	Treatment/management
Gobrial, 2012	Egypt	Cross-sectional	Intellectual	Rehabilitative
He et al., 2017	China	Retrospective cohort	Intellectual	Rehabilitative
( Jindal et al., 2018)	India	Qualitative	Motor impairments/Cerebral palsy	Rehabilitative
Kirabira et al., 2018	Uganda	Cross-sectional	Epilepsy	Treatment/management
Khoza-Shangase, 2019	South Africa	Qualitative	Hearing	Rehabilitative
Mbuba et al., 2012	Kenya	Cross-sectional	Epilepsy	Treatment/management
( McConachie et al., 2001)	Bangladesh	Mixed methods	Motor impairments/cerebral palsy	Rehabilitative
Merugumala et al., 2017	India	Qualitative	Hearing	Rehabilitative
( Nuri et al., 2019)	Bangladesh	Qualitative	Motor impairments/cerebral palsy	Rehabilitative
( Patel et al., 2017)	Botswana	Qualitative	Motor impairments/cerebral palsy	Rehabilitative
El Sharkawy et al., 2006	Kenya	Qualitative	Epilepsy	Treatment/management
Tataryn et al., 2017	Malawi	Cross-sectional	Disabilities	Preventive
Wagner et al., 2016	South Africa	Cross-sectional	Epilepsy	Treatment/management
Yousafzai et al., 2005	Uganda, Rwanda	Qualitative	Disabilities	Preventive

Barriers for healthcare (quantitative analysis)

The barriers identified from all eligible studies were classified, analysed, and reported based on seven all-inclusive themes namely: financial constraints, geographical inaccessibility and physical barriers, the inadequacy of healthcare resources, inadequate education/awareness, prohibitive culture/beliefs, competing domestic roles, and a lack of confidentiality/anonymity ( Table 3).

Table 3. Classification of barriers preventing access/utilization of healthcare by children with neurological impairments/disability.

Theme (category) of barrier	Criteria for inclusion
1. Financial constraints	Determined by whether the patient/caregiver was able to pay for biomedical services and all indirect costs incurred while seeking care.
2. Education/ awareness	Lack of/inadequate information or awareness from healthcare facilities about the availability of care services; or caregiver/patient unable to seek services due to limited education/information/health illiteracy.
3. Culture/beliefs	Caregivers or patients’ values and perceptions inhibiting seeking biomedical services or societal attitude influencing health-seeking behaviour
4. Geographical inaccessibility and physical barriers	Geographical inaccessibility described as the proximity of the healthcare facilities from the patient’s/caregiver residence. Inaccessibility also included the inability to access healthcare services due to physical disabilities and/or unfavourable infrastructural design of healthcare facilities or transport systems.
5. Inadequate healthcare resources and quality of care	The attitude of healthcare workers and availability of appropriate services/equipment such as expert consultation, clinical assessment, supply of critical drugs, laboratory equipment, and testing in the healthcare facilities.
6. Confidentiality/anonymity	Comprising privacy and protection of patient information of those seeking healthcare.
7. Childcare/competing domestic roles	Domestic roles such as childcare or taking care of the sick and elderly at home as competing roles for the caregivers.

The main factors limiting accessing/utilization of healthcare services in >50% of the studies were financial constraints (N=17, 85.0%), geographical inaccessibility (N=14, 70.0%), inadequacy of healthcare resources (N=14, 70.0%), prohibitive culture/beliefs (N=12, 60.0%), and inadequate education/awareness (N=11, 55.0%). Factors reported in less than half of the studies were competing domestic roles (N=4, 20.0%), and the lack of confidentiality/anonymity (N=2, 10.0%). We identified very few reports originating from outside Africa which prevented statistical comparisons of individual reports by continents and economic level of the countries contributing primary studies in the analysis ( Table 4).

Table 4. Barriers limiting healthcare access classified by continents and levels of income.

Barriers	Continent		Economic level
	Africa	Asia	LMI	LI	UMIC
Financial constraints	13 (76.5%)	4 (23.5%)	10 (58.8%)	3 (17.6%)	4 (23.5%)
Geographical accessibility	12 (85.7%)	2 (14,3%)	9 (64.3%)	3 (21.4%)	2 (14.3%)
Inadequate healthcare resources	11 (78.6%)	3 (21.4%)	10 (71.4%)	1 (7.1%)	3 (21.4%)
Inadequate education/awareness	8 (72.7%)	3 (27.3%)	8 (72.7%)	2 (18.2%)	1 (9.1%)
Prohibitive culture/beliefs	9 (75.0%)	3 (25.0%)	7 (58.3%)	3 (25.0%)	2 (16.7%)
Competing domestic roles	2 (50.0%)	2 (50.0%)	4 (100%)	-	-
Confidentiality/anonymity	2 (100%)	-	2 (100%)	-	-

Abbreviations: LMI (low-middle-income), LI (low-income), UMIC (upper-middle-income)

Barriers for healthcare (qualitative analyses)

Financial constraints. Most caregivers could not afford the recurrent costs for the antiseizure medication ( Carter et al., 2012; El Sharkawy et al., 2006; Kirabira et al., 2018). "This hospital is good but sometimes you go there, get examined and prescribed for drugs and you need money for those drugs. So if you don't have money, then you just remain with the illness", reported a caregiver for a patient with epilepsy from a rural village in Kenya ( Carter et al., 2012). Secondly, transportation costs hindered poorer families from accessing healthcare ( Carter et al., 2012; Merugumala et al., 2017; Tataryn et al., 2017). A caregiver from India explained, “I know the center didn't ask for money, but it is the effort to get here right? We have to pay for bus fare and that's a lot for me. So, I left it even when they called us back” ( Merugumala et al., 2017). Indirect costs of healthcare included loss of work-time ( Wagner et al., 2016), loss of income ( Bright et al., 2017), and diversion of limited family resources for treatment ( Carter et al., 2012).

Geographical access. The main geographical factors limiting access were unfavourable terrain ( Bright et al., 2017), and distance decay ( Burke et al., 2017; Carter et al., 2012; El Sharkawy et al., 2006; Mbuba et al., 2012; Tataryn et al., 2017). Unsuitable infrastructural design such as the absence of ramps, unfavourable public transport systems, and a lack of wheelchairs was also identified ( Merugumala et al., 2017). “The journey itself is difficult because my son cannot walk yet, so if I don't find an auto-rickshaw, I have to carry him all the way down the long road to the center. That is very tiring especially in the summer heat, but what else can I do?”, complained a caregiver from India ( Merugumala et al., 2017).

Availability of healthcare resources. Seventy percent of the reports cited either an inadequate number of healthcare facilities, understaffing, or a lack of equipment and medication. Patients had difficulties in getting an appointment in public hospitals as there were few trained experts in neurology ( Alloh et al., 2009; Alrasheed et al., 2018) and rehabilitation. Poor diagnostic equipment and the unavailability of antiseizure medication in healthcare facilities were common for epilepsy patients ( Carter et al., 2012). Screening programs for children with hearing impairment were unavailable and rehabilitation facilities for motor impairment/cerebral palsy and intellectual disability were few and were located in urban centres ( Gobrial, 2012; Khoza-Shangase, 2019; Merugumala et al., 2017).

Education/awareness. Some caregivers lacked information on the causes and treatment of epilepsy ( Carter et al., 2012), and about the existence of healthcare services ( Alrasheed et al., 2018; Burke et al., 2017; El Sharkawy et al., 2006; Merugumala et al., 2017; Tataryn et al., 2017). Lack of health education programs was reported by caregivers for patients with epilepsy and visual impairments, respectively ( Alrasheed et al., 2018; Bassili et al., 2002). Health illiteracy limited caregivers’ awareness about the availability of specialist services and delayed or hindered the diagnosis of hearing impairment and management of children with intellectual disability ( He et al., 2017; Merugumala et al., 2017). Parents and health workers could not communicate with deaf adolescents using sign language and print material in health campaigns was not adapted for blind individuals ( Yousafzai et al., 2005).

Culture/beliefs. There were misconceptions about the cause of epilepsy where animistic beliefs were strongly held. “It is said that it is witchcraft. She was bewitched, that is according to our customs. That is when you will go to a mganga [traditional health practitioner] because you want to untrap them”, explained a grandmother to a child with epilepsy in Kenya ( Carter et al., 2012). Alternative care including consultation of traditional health practitioners (THP) was preferred to biomedical care for childhood eye diseases ( Alrasheed et al., 2018). Societal stigma hindered access to (i) antiseizure medication by children with epilepsy ( Kirabira et al., 2018), rehabilitation for those with cerebral palsy ( Jindal et al., 2018; McConachie et al., 2001; Patel et al., 2017) (ii) access to contraception by adolescents with disabilities ( Yousafzai et al., 2005), and (iii) HIV/AIDS testing among young adults with physical impairments ( Burke et al., 2017). The dominant role of family elders in health-related decisions played a pivotal role where the grandmothers believed that deafness would resolve spontaneously ( Merugumala et al., 2017).

Competing domestic roles, and lack of confidentiality. Four papers reported that childcare and other competing roles such as home care for a sick relative were given a higher priority over the healthcare for a child with a disability ( Bright et al., 2017; McConachie et al., 2001; Merugumala et al., 2017; Tataryn et al., 2017). Two studies ( Burke et al., 2017; Yousafzai et al., 2005) identified the lack of confidentiality and privacy of personal information as patients with disabilities needed to be accompanied by caregivers during consultations.

Discussion

Overall, the main factors hindering access to healthcare by children with NI were financial constraints, geographical and physical inaccessibility, inadequate healthcare resources, prohibitive culture and beliefs, and lack of education or awareness, respectively. Other important but less frequently reported factors were competing for domestic roles for caregivers, and a lack of confidentiality for personal information. Our searches identified more studies in epilepsy, and we obtained fewer reports for the other domains of NI such as hearing impairments. The main form of care reported for epilepsy patients was provision of antiseizure medications; however, there was a lack of newer antiseizure medication and other treatment options such as epilepsy surgery. Rehabilitative services were the most common form of care for those with motor impairments/cerebral palsy and intellectual disability. Most studies originated from Africa and fewer studies were identified from other LMICs complicating valid comparison by continent and level of economic development.

Children with NI from impoverished families could not afford out-of-pocket payments for healthcare, a problem compounded by a lack of health insurance. Many families were forced to neglect healthcare to meet more pressing basic needs such as food and shelter. These can be addressed by expanding the scope of health insurance coverage to reduce out-of-pocket payments for healthcare in LMICs. Children with epilepsy could not regularly access antiseizure medication, a common challenge in developing countries that can be solved by establishing community-based services, outreach programs and reducing the prices of antiseizure medication in settings where they are costly. Indirect costs such as loss of work time have a significant economic impact on these families as time intended to be spent earning an income is used to care for their children. A cycle of poverty and disability might explain the inability of most caregivers to afford expensive primary care for their children ( Banks et al., 2017). Wilmshurst and colleagues ( Wilmshurst et al., 2014), affirm the finding that management of children with NI is expensive, and the cost of healthcare is unaffordable for many impoverished families. Additional expenses such as public transport and indirect costs such as loss of income were observed in previous studies ( Eide et al., 2015).

Inadequate healthcare resources especially for rehabilitation were common in most African studies. Few rehabilitation experts were working in poorly resourced rehabilitation centres predominantly located in urban centres. The lack of specialist services for visually impaired children reflects a previously reported shortage of 3.7 ophthalmologists per one million people in LMICs, a figure which is substantially low compared to 76.2 per million people in high-income countries ( Resnikoff et al., 2020). These shortages can be resolved by investing in training and employing more rehabilitation experts. Lack of information and inadequate communication is a great challenge in LMICs. Ineffective communication between healthcare providers and patient/caregivers has previously been studied ( Maloni et al., 2010), and patients and caregivers with NI would benefit from effective and clear communication from service providers, including the use of sign language for the deaf. Proper information packaging, effective doctor-patient communication, and further investment in health promotion campaigns might create and sustain awareness about neurodisability and healthcare ( WHO, 2018). It is also well-established that maternal education is strongly associated with the use of health services ( Armar-Klemesu et al., 2000), while illiteracy of caregivers presents difficulties in understanding instructions from care providers ( Crabtree, 2007). Also, health education might play a critical role in supporting the previously suggested community-based outreach programs.

As seen in a previous study ( Eide et al., 2015) poor terrain and long geographical distance significantly reduced the likelihood that children with NI from remote areas were able to access healthcare. Also, a lack of environmental modification to cater to those with physical impairment limited access to healthcare in some studies. For instance, there was a shortage of wheelchairs, and public transport systems and hospital facilities lacked provisions for people with physical disabilities. Distance decay, a phenomenon where service utilization reduces with increasing geographical distances from the healthcare facilities can be addressed through decentralizing healthcare and equipping rural health facilities with appropriate healthcare and rehabilitation resources. Additionally, community-based rehabilitation has been recommended to complement the care provided by the existing healthcare systems ( Iemmi et al., 2015). While the convention for the rights of people with disabilities, based on the ICF framework, advocates for modification of the environment for the welfare of those with disabilities ( WHO, 2001; WHO, 2011), these recommendations have not been implemented in most LMICs. Governments must ensure disability mainstreaming in existing and future infrastructural development (such as the construction of ramps) and partnership with non-governmental/private entities in providing other forms of support (e.g. wheelchairs) for children with disabilities.

In terms of culture and beliefs, our results are similar to those from a Turkish study ( Diken, 2006), where mothers who perceived child disability to be a result of curses were more likely to seek traditional interventions. Misconceptions and animistic beliefs on the cause of NI were strongly associated with visiting THP. There is a need to integrate THP into formal healthcare alongside strengthening community-based rehabilitation ( Krah et al., 2018). Attitudes from the patient/caregiver, healthcare provider, or societal perspectives were associated with decisions made by primary caregivers or children with NI regarding healthcare. For example, a lack of altruism and discrimination by healthcare providers, and a lack of privacy was observed in sexual and reproductive care for adolescents with disability ( Banks et al., 2017; Eide et al., 2015). Education and training of healthcare providers on equality and diversity are imperative to address the discrimination in the context of physician-patient-caregiver relationships. There is also a need to train non-existent specialities ( Bunning et al., 2014) and capacity building of existing healthcare staff to be sustained ( Maloni et al., 2010).

Strengths and limitations

There were few studies identified by our searches, with most reports from Africa and none from South America. Logistical constraints e.g. subscription requirements limited the number of databases we could search as well as the translation of reports identified in other languages than English. Also, we acknowledge that our search strategy might have missed some studies because a range of commonly used etiological and disability terms may have changed over time in the existing literature. This will affect the generalizability of our findings, and specific studies are needed from unrepresented settings. Both quantitative and qualitative studies were eligible for analysis, but study design variability prevented the utility of purely qualitative or quantitative methods in this review. While children and adolescents represent a broad range of age groups possibly with different factors influencing the utility of healthcare, there was no standard reporting of barriers in the primary studies reviewed, which should be standardized in future working groups by expert panels or task forces. However, to the best of our knowledge, this is the first systematic review to synthesize evidence of barriers limiting access and utilization of preventive, curative, and rehabilitative care by children and adolescents with NI in multiple domains in LMICs.

Conclusion

Financial constraints, geographic inaccessibility, inadequate healthcare resources, poor communication/awareness, and cultural barriers were the most ubiquitous barriers limiting access and utilization of healthcare services by children with NI. There were more studies from sub-Saharan Africa where the use of rehabilitative care was more common. Expanding health insurance coverage, improving infrastructure with the decentralization of healthcare, and adequate training and staffing of care facilities, combined with investment in structured health promotion are fundamental steps towards addressing these challenges.

Data availability

All data underlying the results are available as part of the article and no additional source data are required.

Reporting guidelines

Harvard Dataverse: PRISMA checklist for ‘Barriers to access and utilization of healthcare by children with neurological impairments and disability in low-and middle-income countries: A systematic review’. https://doi.org/10.7910/DVN/H2V167 ( Abuga et al., 2021b).

Data are available under the terms of the Creative Commons Attribution 4.0 International license (CC-BY 4.0).

Funding Statement

This work was supported through the DELTAS Africa Initiative Grant No. 107754/Z/15/Z-DELTAS Africa SSACAB and from DELTAS Africa Initiative [DEL-15-003]. The DELTAS Africa Initiative is an independent funding scheme of the African Academy of Sciences (AAS)’s Alliance for Accelerating Excellence in Science in Africa (AESA) and supported by the New Partnership for Africa's Development Planning and Coordinating Agency (NEPAD Agency) with funding from the Wellcome Trust [107769/Z/10/Z] and the UK government. The views expressed in this publication are those of the author(s) and not necessarily those of AAS, NEPAD Agency, Wellcome Trust or the UK government. For the purpose of Open Access, the author has applied a CC-BY public copyright license to any author accepted manuscript version arising from this submission.

The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.