The epidemiology of orofacial clefts and other craniofacial anomalies in Trinidad and Tobago and characteristics of multi-disciplinary team cleft care pathways, globally

Juman, Saarah ORCID: https://orcid.org/0000-0002-9434-559X 2024. The epidemiology of orofacial clefts and other craniofacial anomalies in Trinidad and Tobago and characteristics of multi-disciplinary team cleft care pathways, globally. MPhil Thesis, Cardiff University. Item availability restricted.

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Abstract

Introduction Orofacial clefts (OFCs) are the most prevalent craniofacial anomaly (CFA) worldwide, with birth prevalence varying significantly across different geographic regions and ethnic groups. In terms of management, there are also global disparities in the quality and access to comprehensive cleft care through a multidisciplinary (MDT) approach. This is reflected within Small Island Developing States (SIDS) like the Caribbean country, Trinidad and Tobago. Despite its high-income status and universal healthcare system providing free healthcare to its citizens, it faces specific healthcare challenges to other members of the United Nations’ SIDS such as a lack of specialised professionals, infrastructure and quality health services such as a national cleft service. Moreover, the Caribbean, often grouped with Latin America as “Latin America and the Caribbean (LAC)”, is frequently overlooked by international non-profit organisations focused on developing cleft care within low-middle income countries in South and Central America, leaving Caribbean countries with higher income status without similar support. This lack of international influence and interest in developing care for cleft patients within the Caribbean is thought to be at least in part due to the absence of up-to-date epidemiological evidence. Updating the evidence on the epidemiology of orofacial clefts and craniofacial anomalies in Trinidad and Tobago is therefore expected to enhance both local and international efforts aimed at improving cleft care in the country and across the Caribbean region. Additionally, in an ambition to map the characteristics of a cleft care pathway appropriate for introducing a MDT approach to comprehensive cleft care that is sustainable and scalable in low-resource settings such as Trinidad and Tobago and other SIDS, a scoping review approach to systematically reviewing the literature is deemed appropriate. This work supports the United Nation’s Sustainable Development Goals, specifically targets 3.8 and 3.C in achieving access to essential healthcare services and increasing health development in developing countries especially in SIDS. iii Aims and objectives The overall aims of this thesis were to: 1) Explore the epidemiology of orofacial clefts (OFCs) and other craniofacial anomalies in Trinidad and Tobago; and 2) Appraise the literature on MDT care pathways for the management of patients with OFCs globally and use the information found to suggest characteristics of a care pathway appropriate for introducing basic MDT cleft care in low-resource settings that is sustainable and scalable. To achieve this, the objectives were to: 1. Determine the birth prevalence (incidence) of orofacial clefts and other craniofacial anomalies in Trinidad and Tobago and explore associated factors that might be linked to these anomalies through an epidemiological study following a cross-sectional, retrospective, quantitative, observational design. 2. Identify the characteristics of an ideal MDT cleft care pathway and facilitating factors and challenges to its implementation; and 3. Define characteristics of a basic, sustainable MDT cleft care pathway that can be implemented. Methods The aims and objectives were addressed through two pieces of work. The first was an epidemiological study with an observational quantitative cross-sectional design, over a 5-year retrospective period (2018-2022). Birth prevalence was determined using data extracted from Trinidad and Tobago’s digital national birth registry database. The numbers of craniofacial anomalies identified in the database for the country’s different Regional Health Authorities (RHAs) were verified through searching a variety of data sources including hospital files/records and nursing admission notes at each RHA’s Neonatal Intensive Care Unit (NICU) during that period. To explore the evidence-based associated risk factors that might be linked to OFCs, hospital records of babies and their mothers were searched. For each baby iv identified as being born with an OFC, a healthy newborn closely matched (by sex and birth month) was randomly selected within the same RHA to form study and control groups. Birth prevalence and the samples were presented using descriptive statistics. Logistic regression models were used to assess which factors are more strongly linked with the birth of a baby with an OFC. The second was a scoping review conducted following the Joanna Briggs Institute (JBI) guidelines. To identify eligible sources of evidence, multiple electronic databases were searched along with hand-searching for grey literature. Only data sources focused exclusively on the management (which includes both diagnosis and treatment from all clinical specialities) of non-syndromic orofacial cleft were considered. Studies were screened by two independent reviewers using the pre-determined eligibility criteria and any disagreements resolved with a third (experienced) reviewer. Results The birth prevalence of the most common craniofacial anomaly, orofacial clefts, within the population of Trinidad and Tobago was found to be 0.64 per 1000 births. Other craniofacial anomalies were poorly reported as only 3 cases of micrognathia were reported by a single RHA. This study also encountered missing data and inconsistently available and ambiguous data suggesting that record-keeping needs improvement and optimisation in the areas of patient history documentation, craniofacial anomaly surveillance and orofacial cleft classification. Although only a limited number of variables were appropriate for analysis, significant predictors of OFCs were still identified (gestational age; birth weight; birth length; maternal age; maternal medical history within normal limits; gravida; para). This study’s findings suggest that the likelihood of a birth with an OFC decreases with gestational age, birth weight, birth length and if there is a maternal medical history within normal limits. Also, the likelihood of a birth with an OFC increases with maternal age and number of pregnancies. When analysed together, only maternal medical history within normal limits and gravida remain significant suggesting these are the stronger predictors which might themselves v be influencing the other variables. The analysis suggests that a mother with a normal medical history is 91% less likely to give birth to a baby with an OFC. Women with a higher gravida are 74% more likely to give birth to a baby with an OFC. This study also emphasises that development is required to establish a basic cleft MDT care pathway that introduces comprehensive cleft care for these individuals, as an observational finding revealed the absence of a standardised multidisciplinary pathway providing comprehensive cleft care. The scoping review identified 34 sources suitable for synthesis and interpretation. Overarching themes/ characteristics of MDT cleft care pathway were identified within the included sources of evidence as a statement of goals, infrastructure, MDT composition, supporting documents for the delivery of safe, quality cleft care, cleft care timeline and facilitating factors to optimising the care pathways. Using these characteristics, an ideal cleft care pathway was defined and suggestions were proposed for a basic cleft care pathway capable of introducing comprehensive cleft care in low-resource settings. Conclusions In conclusion, this thesis updates the epidemiological evidence on the most common craniofacial anomaly, orofacial clefts, within the population of Trinidad and Tobago. It outlines the steps necessary to support future epidemiological studies on craniofacial anomalies in this population. Additionally, it provides initial suggestions for the characteristics of a care pathway suitable for introducing basic multidisciplinary team (MDT) cleft care that is sustainable and scalable in low-resource settings, while acknowledging that further review and appraisal involving stakeholders is required to develop recommendations.

Item Type:	Thesis (MPhil)
Date Type:	Completion
Status:	Unpublished
Schools:	Schools > Dentistry
Date of First Compliant Deposit:	18 September 2025
Last Modified:	22 Sep 2025 10:03
URI:	https://orca.cardiff.ac.uk/id/eprint/181178

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