Intra- and Inter-Rater Reliability and Validation of Orofacial Cleft Classification in the Cleft Collective Cohort Study

ObjectiveAssess the level of agreement for classification of orofacial clefts within and between different raters and validate classifications.DesignValidation study.SettingNational longitudinal prospective cohort, United Kingdom.ParticipantsChildren born with orofacial cleft (n=4211), recruited to the Cleft Collective between 2013 and 2024.Main outcome measuresFour cleft classifications of orofacial clefts were explored. Classification one comprised cleft lip, cleft palate, and cleft lip and palate. Classification two added laterality, classification three added sidedness and classification four added completeness using LAHSAL. Data on orofacial cleft classification were collected through seven different sources.ResultsAt least one report of cleft classification was available for 4052 Cleft Collective study children. When assessing intra-rater agreement mothers had the highest level of agreement for the simplest form of cleft classification with a Krippendorf's Alpha of 0.987. When recording LAHSAL for the same child, surgeons reported the same classification for 71% of children (Krippendorf's Alpha = 0.672). When assessing inter-rater agreement across different sources, the simplest cleft classification resulted in the highest level of agreement (Krippendorf's Alpha = 0.957) and the least agreement when using LAHSAL (Krippendorf's Alpha = 0.538).ConclusionOur study found that the more complex a cleft classification becomes the less agreement there is between sources. Differences across sources became most apparent when reporting the sidedness or completeness of a cleft. Validation of cleft classification is important for both clinical care and research. Although LAHSAL is advocated for use both clinically and in research our data show that rigorous training is essential.

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Publication Details

Journal
The Cleft Palate-Craniofacial Journal
Published
2026-05-10
DOI
https://doi.org/10.1177/10556656261443367
Primary Topic
Cleft Lip and Palate Research
Type
article
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article

Intra- and Inter-Rater Reliability and Validation of Orofacial Cleft Classification in the Cleft Collective Cohort Study

Kerry Humphries, Jibby Medina, Yvonne Wren, Amy Davies et al.
The Cleft Palate-Craniofacial Journal
Cleft Lip and Palate Research
article

Intra- and Inter-Rater Reliability and Validation of Orofacial Cleft Classification in the Cleft Collective Cohort Study

Kerry Humphries, Jibby Medina, Yvonne Wren, Amy Davies, Jonathan Sandy, Craig Russell, Scott Deacon, The Cleft Multidisciplinary Collaborative, David Sainsbury, Kate J. Fitzsimons
article en

Abstract

ObjectiveAssess the level of agreement for classification of orofacial clefts within and between different raters and validate classifications.DesignValidation study.SettingNational longitudinal prospective cohort, United Kingdom.ParticipantsChildren born with orofacial cleft (n=4211), recruited to the Cleft Collective between 2013 and 2024.Main outcome measuresFour cleft classifications of orofacial clefts were explored. Classification one comprised cleft lip, cleft palate, and cleft lip and palate. Classification two added laterality, classification three added sidedness and classification four added completeness using LAHSAL. Data on orofacial cleft classification were collected through seven different sources.ResultsAt least one report of cleft classification was available for 4052 Cleft Collective study children. When assessing intra-rater agreement mothers had the highest level of agreement for the simplest form of cleft classification with a Krippendorf's Alpha of 0.987. When recording LAHSAL for the same child, surgeons reported the same classification for 71% of children (Krippendorf's Alpha = 0.672). When assessing inter-rater agreement across different sources, the simplest cleft classification resulted in the highest level of agreement (Krippendorf's Alpha = 0.957) and the least agreement when using LAHSAL (Krippendorf's Alpha = 0.538).ConclusionOur study found that the more complex a cleft classification becomes the less agreement there is between sources. Differences across sources became most apparent when reporting the sidedness or completeness of a cleft. Validation of cleft classification is important for both clinical care and research. Although LAHSAL is advocated for use both clinically and in research our data show that rigorous training is essential.

The Cleft Palate-Craniofacial Journal
North Bristol NHS Trust (GB), Royal College of Surgeons of England (GB), University Of Bristol Dental Hospital (GB), Royal Hospital for Children (GB), University Hospitals Bristol NHS Foundation Trust (GB), University of Bristol (GB), Royal Hospital for Sick Children (GB), Newcastle upon Tyne Hospital (GB), University Hospitals Bristol and Weston NHS Foundation Trust (GB), Cardiff Metropolitan University (GB)
Good health and well-being
Openalex Percentile: Top 5%
Cleft Lip and Palate Research
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