Oral health is a critical health concern with 43.3% of children in India suffering from oral health issues such as dental caries [1]. Children with intellectual and developmental disabilities (IDD), including cerebral palsy (CP), Down syndrome (DS), intellectual disability (ID), autism spectrum disorder (ASD), and attention deficit hyperactivity disorder (ADHD), have a notably greater prevalence of oral health problems [2]. While IDD is not a risk factor for poor oral health in itself, however, IDD decreases a person’s capacity to take care of themselves, which tangentially raises the likelihood of oral health diseases [3]. These children have high rates of bruxism, traumatic dental injuries, and gingival infections, which can be brought on by a variety of factors including medication side effects, behavioural abnormalities, and impaired motor skills [4]. This increases the risk of tooth loss, caries, inflammation, and plaque development in persons with IDD compared to those without it [5]. The consequences of poor oral health are not limited to discomfort and aesthetic issues; they have been linked to general medical conditions such as aspiration pneumonia, cardiovascular disease, diabetes, and respiratory problems [6,7,8,9,10].
According to National Family Health Survey (NFHS-5), there are 5,229 children aged 0–14 years with intellectual disabilities in India [11]. Children with IDD face unique hurdles in achieving optimal physical, psychological, and social health. Oral disorders can cause toothache, stress, difficulties with routine activities, social disengagement, and reduced food intake, emphasising the need for specialised oral healthcare practices for children with IDD [12, 13]. Nevertheless, children with IDD may experience a variety of obstacles to obtaining appropriate oral health treatment. These involve various challenges that carers face, such as low income and insufficient education, living in rural regions, and a lack of access to appropriate dental treatment [5, 13]. Anxiety and refusal to cooperate during dental treatments compound these issues, necessitating the use of sedatives and unique communication skills to alleviate the patient’s suffering and treat the conditions. Furthermore, carers and non-trained dentists’ unwillingness to provide dental care to children with IDD also contribute to oral health disparities [14, 15].
To address this health disparity, United Nations (UN) members, including India, have committed to the Sustainable Development Goals (SDGs) 2030 principle of “Leave No One Behind”, which seeks to reduce disparities and vulnerabilities [16]. Nonetheless, the lack of data on the oral health condition of children with IDD in India is a significant problem. This knowledge gap underlines the necessity for a systematic review aimed at collating and synthesising the data on the oral health status of these children, which might provide the data required to develop future preventive and intervention efforts focused on reducing oral health disparities. This systematic review and meta-analysis was designed with a specific focus to chart the oral health of children with IDD in the Indian subcontinent by collating available evidence. The research objective is restricted to India as the subcontinent presents with a diverse demographic. Analysing one country in detail serves as a foundation for a later comparison with other regions. The results of this study will outline the necessary steps for implementing appropriate oral healthcare strategies that align with the SDGs’ commitment to inclusivity and equality.
Research question
What is the oral health status of the children with Intellectual and Development Disabilities?
The research Question followed the Population, Intervention, Comparator and Outcome (PICO) format as follows:
Population: Children with Intellectual and Development Disabilities.
Intervention: None.
Comparator: None.
Outcome: Oral health habits and oral health related issues.
Methods
We preregistered a protocol for the study before initiating it [PROSPERO Registration: CRD42024512646]. The latest edition of the Preferred Reporting Items for Systematic Reviews and Meta-analysis Statement (PRISMA) [17] was adopted to guide the study’s methodology. Supplementary Table 2 depicts PRISMA guidelines.
Search strategy
An exhaustive literature search was conducted for the following databases: PubMed-MEDLINE, Scopus, and Embase from the earliest available date till 31st March 2024. Additional Additional sources like Google Scholar, unpublished studies, conference proceedings, and cross-references were explored. Medical Subject Headings (MeSH) terms, keywords, phrase searching, truncation and other free terms combined with boolean operators (OR, AND) were used to search articles. Identical keywords were used for all the search platforms following the syntax rules of each database. The keywords utilised are presented in Supplementary Table 1. The search results were downloaded to a bibliographic database (EndNote 20) to facilitate the removal of duplicate entries. Authors were contacted for any unpublished studies.
Eligibility criteria
The eligibility critera was as follows: (1) Population: individuals below 18 years of age with IDDs in India. Any significant deficit in adaptive behaviour and intellectual functions, including learning, thinking, and problem-solving, that starts in childhood and affects one’s ability to function independently is referred to as an intellectual or developmental disability (IDD) [18]. Articles that solely consisted of children with physical disabilities were excluded from the analysis. (2) Intervention/Comparator: None; (3) Outcome: Oral health status indices such as brushing habits, and dental issues such as dental caries, gingivitis, periodontitis, or any other indices measured by the authors. IDD patients with oral health issue secondary to any systemic illness were excluded; (4) Study Design: Cross-sectional studies were included. qualitative studies or secondary articles such as systematic reviews, editorials, opinions, viewpoints, conference proceedings, and research without complete text were eliminated. Articles published in non-English language were also not considered eligible for inclusion.
Assessment and data selection
All the information obtained through screening of the articles were imported into EndNote 20, and all the titles and abstracts were reimported into the Excel Workbook. The articles were independently scanned by two reviewers (VM and ST), initially by title and abstract. The articles were selected for full-text reading, if the search keywords were present in the title and the abstract. Articles without abstracts but with titles related to our objectives were also selected to screen the full-text for inclusion. After selection, both the reviewers (VM and ST) read the full-text articles in detail. The articles that fulfilled the inclusion criteria were processed for data extraction. Both the reviewers (VM and ST) searched the reference lists of all the selected articles for additional relevant articles. The level of agreement between the two reviewers, calculated by Cohen’s kappa (k) index, was 0.92 for titles and abstracts and 0.90 for full texts. Disagreements between the two reviewers were resolved through discussions. If a conflict persisted, the judgement of a third reviewer (AM) was considered decisive.
Quality assessment
Two reviewers (VM and ST) used the Joanna Briggs Institute 2017 critical evaluation checklist for prevalence studies [19] to evaluate the included studies’ quality based on the eight criteria specified in the checklist. Any disagreement between the two reviewers was resolved by discussion until consensus.
Data extraction
Two reviewers (VM and ST) independently extracted data using specially designed data extraction forms utilising Microsoft Excel software. Any disagreement was resolved by discussion between the authors. Information curated for data extraction included author/year of publication, study setting, disability type, age, number of males and females, total sample size, data collection tool, and major findings.
Statistical analysis
We believe that all the studies were comparable as the assessment of oral health status followed worldwide guidelines (World Health Organisation Oral health assessment form, 1997; community periodontal index (CPI); Oral hygiene index-simplified). However, considering the variation in age group and diagnosis of participants across the included studies, we performed a meta-analysis using a random-effects model for variables reported from four or more studies (with a significance level set at < 0.05) and a prediction interval was calculated. Heterogeneity among studies was investigated using Cochrane’s Q, I2 and tau2 statistics. The analyses were done using the meta package in R version 4.3.0.
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