Abstract

To discern environmental factors that, along with genes influencing susceptibility, drive the occurrence of non-syndromic congenital disorders, it is important to identify clusters of these abnormalities. Determine the adjusted prevalence of orofacial clefts in Villa Clara Province, Cuba, and identify and describe their spatiotemporal variability during January 2013-December 2018. Cases were selected from a total of 46,007 births that took place in the province's four maternity hospitals during the study period. Of these, 36 cases of newborns with either prenatal or postnatal non-syndromic orofacial cleft diagnoses were obtained from hospital and community registries. We applied spatial statistical analysis techniques with the aim of identifying areas within the province with the highest prevalence. Adjusted prevalence was 0.78 per 1000 births. The most common non-syndromic orofacial congenital abnormality was cleft lip with or without cleft palate. Frequency of congenital abnormalities increased during the first two years of the study and decreased during the last two years. A primary spatiotemporal cluster was identified in two contiguous municipalities in 2017 and a secondary one in two other neighboring municipalities between 2014 and 2016. Spatiotemporal analysis of non-syndromic orofacial clefts in Villa Clara Province, Cuba, identified two spatiotemporal clusters, constituting an opportunity to better understand the etiology of orofacial clefts.

Highlights

  • To discern environmental factors that, along with genes influencing susceptibility, drive the occurrence of nonsyndromic congenital disorders, it is important to identify clusters of these abnormalities

  • Spatiotemporal analysis of non-syndromic orofacial clefts in Villa Clara Province, Cuba, identified two spatiotemporal clusters, constituting an opportunity to better understand the etiology of orofacial clefts

  • Non-syndromic orofacial cleft (NSOFC) refers to a congenital abnormality of multifactorial origin attributed to a combination of genetic and environmental factors, in which a monogenic or chromosomal cause has not been identified, characterized by a cleft lip with or without palate involvement

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Summary

Introduction

To discern environmental factors that, along with genes influencing susceptibility, drive the occurrence of nonsyndromic congenital disorders, it is important to identify clusters of these abnormalities. Non-syndromic orofacial cleft (NSOFC) refers to a congenital abnormality of multifactorial origin attributed to a combination of genetic and environmental factors, in which a monogenic or chromosomal cause has not been identified, characterized by a cleft lip with or without palate involvement. Its clinical phenotype varies from small abnormalities on the vermillion border of the lip (bilaterally, unilaterally or centrally) to large spreading clefts reaching toward the nostril floor through the maxilla’s alveolar portion, which can cause complete palatal clefts encompassing the secondary or posterior palate, extending over the entire soft palate and reaching the anterior incisive fossa.[4,5]. In the 21st century, NSOFCs were already being classified into two groups: either CL, with or without non-syndromic CP; or non-syndromic CP according to different embryological and environmental factors and genetic studies. Cases with non-syndromic CP show a separation in either the hard palate, soft palate or uvula

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