Dentition patterns in bilateral cleft lip subphenotypes: multicenter study

M. J. Faaij; N. C.W. van der Kaaij; M. A. Disse; J. P.W. Don Griot; C. Vermeij-Keers; E. M. Bronkhorst; E. M. Ongkosuwito

doi:10.1007/s00784-022-04431-y

Dentition patterns in bilateral cleft lip subphenotypes: multicenter study

M. J. Faaij^*, N. C.W. van der Kaaij, M. A. Disse, J. P.W. Don Griot, C. Vermeij-Keers, E. M. Bronkhorst, E. M. Ongkosuwito

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

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.

Original language	English
Pages (from-to)	4623-4632
Number of pages	10
Journal	Clinical Oral Investigations
Volume	26
Issue number	6
Early online date	22 Mar 2022
DOIs	https://doi.org/10.1007/s00784-022-04431-y
Publication status	Published - Jun 2022

Bibliographical note

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

Funding Information:
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.

Publisher Copyright:
© 2022, The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

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@article{b495a6abc98d41a2bee14ce161d6300a,

title = "Dentition patterns in bilateral cleft lip subphenotypes: multicenter study",

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.",

author = "Faaij, {M. J.} and {van der Kaaij}, {N. C.W.} and Disse, {M. A.} and {Don Griot}, {J. P.W.} and C. Vermeij-Keers and Bronkhorst, {E. M.} and Ongkosuwito, {E. M.}",

note = "Funding Information: The work was supported by the Section of Orthodontics and Craniofacial Biology, Department of Dentistry, Radboud Medical Center Nijmegen, the Netherlands. Funding Information: 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. Publisher Copyright: {\textcopyright} 2022, The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.",

year = "2022",

month = jun,

doi = "10.1007/s00784-022-04431-y",

language = "English",

volume = "26",

pages = "4623--4632",

journal = "Clinical Oral Investigations",

issn = "1432-6981",

publisher = "Springer-Verlag",

number = "6",

}

TY - JOUR

T1 - Dentition patterns in bilateral cleft lip subphenotypes

T2 - multicenter study

AU - Faaij, M. J.

AU - van der Kaaij, N. C.W.

AU - Disse, M. A.

AU - Don Griot, J. P.W.

AU - Vermeij-Keers, C.

AU - Bronkhorst, E. M.

AU - Ongkosuwito, E. M.

N1 - Funding Information: The work was supported by the Section of Orthodontics and Craniofacial Biology, Department of Dentistry, Radboud Medical Center Nijmegen, the Netherlands. Funding Information: 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. Publisher Copyright: © 2022, The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

PY - 2022/6

Y1 - 2022/6

N2 - 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.

AB - 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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DO - 10.1007/s00784-022-04431-y

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SN - 1432-6981

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JO - Clinical Oral Investigations

JF - Clinical Oral Investigations

IS - 6

ER -

Dentition patterns in bilateral cleft lip subphenotypes: multicenter study

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