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Dentition patterns in bilateral cleft lip subphenotypes: multicenter study

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Abstract

Objectives

Here, we retrospectively investigated cases of bilateral oral clefts (OCs) to determine the clinical relevance of detailed distinction of incomplete cleft lip subphenotypes, based on morphological severity of the cleft, within the categories cleft lip with or without alveolus (CL ± A) and cleft lip, alveolus, and palate (CLAP). We further assessed possible associations between CL subphenotypes (complete vs different incomplete types) and different dentition patterns of the lateral incisor.

Materials and methods

Our analysis included 151 non-syndromic Caucasian bilateral OC-patients (8–20 years old) from the Dutch Association for Cleft Palate and Craniofacial Anomalies registry. Six different deciduous and permanent lateral incisor patterns were distinguished: normal position (z/Z), supernumerary lateral incisor (n/N), presence in the anterior (x/X) or posterior (y/Y) segment of the cleft, one in each cleft segment (xy/XY), and agenesis (ab/AB). Logistic regression was performed to show the associations between the CL subphenotypes and dentition patterns of the lateral incisor.

Results

One hundred three had complete, while 48 had incomplete CLs. Patterns z/Z and n/N were associated with a submucous/vermillion notch, incomplete CL, and intact alveolus. Patterns x/X, y/Y, and xy/XY were most common in patients with two-thirds to subtotal CL and complete CL. The most severe pattern, ab/AB, was most commonly associated with complete CL.

Conclusions

Based on the morphological severity of the CLs, it can be stated that the more severe the CL in bilateral CL ± A and CLAP, the more severe the abnormal pattern of the dentition.

Clinical relevance

Further distinction of incomplete cleft lip subphenotypes (submucous/vermillion notch, one-third to two-thirds CL, two-thirds to subtotal CL) in bilateral CL ± A and CLAP has clinical relevance.

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Acknowledgements

The authors thank the members of the cleft palate teams from the Erasmus University Medical Center Rotterdam, Radboud University Medical Center Nijmegen, and Amsterdam UMC location VU University Medical Center for their support with data acquisition. Furthermore, we want to thank A.H. Trenning for the data acquisition from the NVSCA registry.

Funding

The work was supported by the Section of Orthodontics and Craniofacial Biology, Department of Dentistry, Radboud Medical Center Nijmegen, the Netherlands.

Author information

Authors and Affiliations

  1. Department of Dentistry, Section of Orthodontics and Craniofacial Biology, Radboud University Nijmegen Medical Center, 309 Dentistry, PO Box 9101, 6500, HB, Nijmegen, the Netherlands

    M. J. Faaij & E. M. Ongkosuwito

  2. Department of Orthodontics, Erasmus University Medical Center Rotterdam, Dr. Molewaterplein 40, 3015, GD, Rotterdam, the Netherlands

    N. C. W. van der Kaaij

  3. Department of Dentistry, Section of Orthodontics, Amsterdam University Medical Centers, Location VUmc, Amsterdam, Gustav Mahlerlaan 3004, 1081, LA, Amsterdam, the Netherlands

    M. A. Disse

  4. Department of Plastic Surgery, Amsterdam University Medical Centers, location VUmc, Amsterdam, De Boelelaan 1117, 1081, HV, Amsterdam, the Netherlands

    J. P. W. Don Griot

  5. Department of Plastic, Reconstructive and Hand Surgery, Erasmus University Medical Center, Rotterdam, Dr. Molewaterplein 40, 3015, GD, Rotterdam, the Netherlands

    C. Vermeij-Keers

  6. Department of Dentistry, Section of Preventive and Restorative Dentistry, Radboud University Medical Center Nijmegen, 309 Dentistry, PO Box 9101, 6500, HB, Nijmegen, the Netherlands

    E. M. Bronkhorst

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  1. M. J. Faaij
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  7. E. M. Ongkosuwito
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Contributions

M.J. Faaij and E.M. Ongkosuwito contributed to the study conception and design; data acquisition, analysis, and interpretation; and the drafting and critical revision of the manuscript. N.C.W. van der Kaaij, M.A. Disse, J.P.W. Don Griot, and C. Vermeij-Keers contributed to the data acquisition, and critically revised the manuscript. E.M. Bronkhorst performed all statistical analyses and critically revised the manuscript. All authors gave their final approval, and have agreed to be accountable for all aspects of the work.

Corresponding author

Correspondence to M. J. Faaij.

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Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee (Erasmus MC: 2019-078, Radboudumc: 2018-4208, Amsterdam VUmc: 2019-3479) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

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For this type of study, formal consent is not required.

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The authors declare no competing interests.

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Faaij, M.J., van der Kaaij, N.C.W., Disse, M.A. et al. Dentition patterns in bilateral cleft lip subphenotypes: multicenter study. Clin Oral Invest 26, 4623–4632 (2022). https://doi.org/10.1007/s00784-022-04431-y

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