Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis

Brian J. Howe; Chandler Pendleton; Miyuraj Harishchandra Hikkaduwa Withanage; Christopher A. Childs; Erliang Zeng; Arjen van Wijk; Ruurd Hermus; Carmencita Padilla; Jacqueline T. Hecht; Fernando A. Poletta; Iêda M. Orioli; Carmen J. Buxó-Martínez; Frederic Deleyiannis; Alexandre R. Vieira; Azeez Butali; Consuelo Valencia-Ramirez; Claudia Restrepo Muñeton; George L. Wehby; Seth M. Weinberg; Mary L. Marazita; Lina M.Moreno Uribe; Xian Jin Xie

doi:10.3390/dj10070128

Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis

Brian J. Howe^*, Chandler Pendleton, Miyuraj Harishchandra Hikkaduwa Withanage, Christopher A. Childs, Erliang Zeng, Arjen van Wijk, Ruurd Hermus, Carmencita Padilla, Jacqueline T. Hecht, Fernando A. Poletta, Iêda M. Orioli, Carmen J. Buxó-Martínez, Frederic Deleyiannis, Alexandre R. Vieira, Azeez Butali, Consuelo Valencia-Ramirez, Claudia Restrepo Muñeton, George L. Wehby, Seth M. Weinberg, Mary L. MarazitaLina M.Moreno Uribe, Xian Jin Xie

^*Corresponding author for this work

Oral Public Health

Research output: Contribution to Journal › Review article › Academic › peer-review

Abstract

Individuals with orofacial clefting (OFC) have a higher prevalence of tooth agenesis (TA) overall. Neither the precise etiology of TA, nor whether TA occurs in patterns that differ by gender or cleft type is yet known. This meta-analysis aims to identify the spectrum of tooth agenesis patterns in subjects with non-syndromic OFC and controls using the Tooth Agenesis Code (TAC) program. An indexed search of databases (PubMed, EMBASE, and CINAHL) along with cross-referencing and hand searches were completed from May to June 2019 and re-run in February 2022. Additionally, unpublished TAC data from 914 individuals with OFC and 932 controls were included. TAC pattern frequencies per study were analyzed using a random effects meta-analysis model. A thorough review of 45 records retrieved resulted in 4 articles meeting eligibility criteria, comprising 2182 subjects with OFC and 3171 controls. No TA (0.0.0.0) was seen in 51% of OFC cases and 97% of controls. TAC patterns 0.2.0.0, 2.0.0.0, and 2.2.0.0 indicating uni-or bi-lateral missing upper laterals, and 16.0.0.0 indicating missing upper right second premolar, were more common in subjects with OFC. Subjects with OFC have unique TA patterns and defining these patterns will help increase our understanding of the complex etiology underlying TA.

Original language	English
Article number	128
Pages (from-to)	1-15
Number of pages	15
Journal	Dentistry Journal
Volume	10
Issue number	7
Early online date	5 Jul 2022
DOIs	https://doi.org/10.3390/dj10070128
Publication status	Published - Jul 2022

Bibliographical note

Special Issue: Dentistry Journal: 10th Anniversary.

Funding Information:
Grant support was provided by: K08 DE028012: “Identifying Phenomic Patterns of Dental Anomalies in Orofacial Clefting” (University of Iowa as Primary Awardee). R01 DE106148: “Ex-tending the Phenotype of Non-Syndromic Orofacial Clefts” (University of Pittsburgh as Primary Awardee). R01 DE014667: “Cleft Lip Genetics: A Multicenter International Consortium” (University of Iowa as Primary Awardee). R37-DE-08559: “Molecular Genetic Epidemiology of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R01 DD000295: “Health Outcomes and Improved Phenotypic Characterization of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R00 DE024571: “Genetic and Environmental Risks for Oral Clefts in Puerto Rican Hispanics” (University of Puerto Rico as primary awardee). S21 MD001830: “Hispanics in Research Capability: SOHP and SOM Partnership” (University of Puerto Rico as primary awardee). U54 MD007587: “Puerto Rico Clinical and Translational Research Consortium” (University of Puerto Rico as primary awardee). R00 DE022378: ”Genetic Studies of Non-Syndromic Clefts in Populations of African De-scent” (University of Iowa as Primary Awardee). R01 DE02830: “Refining the Genetic and Genomic Architecture of Non-Syndromic Orofacial Clefting” (University of Iowa as Primary Awardee). Robert Wood Johnson Foundation 72429: “Genome-Wide Association Studies for Non-Syndromic Clefts in sub-Saharan African Populations”.

Publisher Copyright:
© 2022 by the authors. Licensee MDPI, Basel, Switzerland.

Funding

Grant support was provided by: K08 DE028012: “Identifying Phenomic Patterns of Dental Anomalies in Orofacial Clefting” (University of Iowa as Primary Awardee). R01 DE106148: “Ex-tending the Phenotype of Non-Syndromic Orofacial Clefts” (University of Pittsburgh as Primary Awardee). R01 DE014667: “Cleft Lip Genetics: A Multicenter International Consortium” (University of Iowa as Primary Awardee). R37-DE-08559: “Molecular Genetic Epidemiology of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R01 DD000295: “Health Outcomes and Improved Phenotypic Characterization of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R00 DE024571: “Genetic and Environmental Risks for Oral Clefts in Puerto Rican Hispanics” (University of Puerto Rico as primary awardee). S21 MD001830: “Hispanics in Research Capability: SOHP and SOM Partnership” (University of Puerto Rico as primary awardee). U54 MD007587: “Puerto Rico Clinical and Translational Research Consortium” (University of Puerto Rico as primary awardee). R00 DE022378: ”Genetic Studies of Non-Syndromic Clefts in Populations of African De-scent” (University of Iowa as Primary Awardee). R01 DE02830: “Refining the Genetic and Genomic Architecture of Non-Syndromic Orofacial Clefting” (University of Iowa as Primary Awardee). Robert Wood Johnson Foundation 72429: “Genome-Wide Association Studies for Non-Syndromic Clefts in sub-Saharan African Populations”.

Funders	Funder number
Puerto Rico Clinical and Translational Research Consortium	R01 DE02830, 72429
Universidad de Puerto Rico	U54 MD007587, S21 MD001830
Universidad de Puerto Rico

Keywords

cleft lip and palate
hypodontia
machine learning
phenotype

Access to Document

10.3390/dj10070128

Persistent URL (handle)

Persistent link

Cite this

Howe, B. J., Pendleton, C., Withanage, M. H. H., Childs, C. A., Zeng, E., van Wijk, A., Hermus, R., Padilla, C., Hecht, J. T., Poletta, F. A., Orioli, I. M., Buxó-Martínez, C. J., Deleyiannis, F., Vieira, A. R., Butali, A., Valencia-Ramirez, C., Muñeton, C. R., Wehby, G. L., Weinberg, S. M., ... Xie, X. J. (2022). Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis. Dentistry Journal, 10(7), 1-15. Article 128. https://doi.org/10.3390/dj10070128

@article{9e97e77407e24513a45cc52f31791131,

title = "Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis",

abstract = "Individuals with orofacial clefting (OFC) have a higher prevalence of tooth agenesis (TA) overall. Neither the precise etiology of TA, nor whether TA occurs in patterns that differ by gender or cleft type is yet known. This meta-analysis aims to identify the spectrum of tooth agenesis patterns in subjects with non-syndromic OFC and controls using the Tooth Agenesis Code (TAC) program. An indexed search of databases (PubMed, EMBASE, and CINAHL) along with cross-referencing and hand searches were completed from May to June 2019 and re-run in February 2022. Additionally, unpublished TAC data from 914 individuals with OFC and 932 controls were included. TAC pattern frequencies per study were analyzed using a random effects meta-analysis model. A thorough review of 45 records retrieved resulted in 4 articles meeting eligibility criteria, comprising 2182 subjects with OFC and 3171 controls. No TA (0.0.0.0) was seen in 51% of OFC cases and 97% of controls. TAC patterns 0.2.0.0, 2.0.0.0, and 2.2.0.0 indicating uni-or bi-lateral missing upper laterals, and 16.0.0.0 indicating missing upper right second premolar, were more common in subjects with OFC. Subjects with OFC have unique TA patterns and defining these patterns will help increase our understanding of the complex etiology underlying TA.",

keywords = "cleft lip and palate, hypodontia, machine learning, phenotype",

author = "Howe, {Brian J.} and Chandler Pendleton and Withanage, {Miyuraj Harishchandra Hikkaduwa} and Childs, {Christopher A.} and Erliang Zeng and {van Wijk}, Arjen and Ruurd Hermus and Carmencita Padilla and Hecht, {Jacqueline T.} and Poletta, {Fernando A.} and Orioli, {I{\^e}da M.} and Bux{\'o}-Mart{\'i}nez, {Carmen J.} and Frederic Deleyiannis and Vieira, {Alexandre R.} and Azeez Butali and Consuelo Valencia-Ramirez and Mu{\~n}eton, {Claudia Restrepo} and Wehby, {George L.} and Weinberg, {Seth M.} and Marazita, {Mary L.} and Uribe, {Lina M.Moreno} and Xie, {Xian Jin}",

note = "Special Issue: Dentistry Journal: 10th Anniversary. Funding Information: Grant support was provided by: K08 DE028012: “Identifying Phenomic Patterns of Dental Anomalies in Orofacial Clefting” (University of Iowa as Primary Awardee). R01 DE106148: “Ex-tending the Phenotype of Non-Syndromic Orofacial Clefts” (University of Pittsburgh as Primary Awardee). R01 DE014667: “Cleft Lip Genetics: A Multicenter International Consortium” (University of Iowa as Primary Awardee). R37-DE-08559: “Molecular Genetic Epidemiology of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R01 DD000295: “Health Outcomes and Improved Phenotypic Characterization of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R00 DE024571: “Genetic and Environmental Risks for Oral Clefts in Puerto Rican Hispanics” (University of Puerto Rico as primary awardee). S21 MD001830: “Hispanics in Research Capability: SOHP and SOM Partnership” (University of Puerto Rico as primary awardee). U54 MD007587: “Puerto Rico Clinical and Translational Research Consortium” (University of Puerto Rico as primary awardee). R00 DE022378: ”Genetic Studies of Non-Syndromic Clefts in Populations of African De-scent” (University of Iowa as Primary Awardee). R01 DE02830: “Refining the Genetic and Genomic Architecture of Non-Syndromic Orofacial Clefting” (University of Iowa as Primary Awardee). Robert Wood Johnson Foundation 72429: “Genome-Wide Association Studies for Non-Syndromic Clefts in sub-Saharan African Populations”. Publisher Copyright: {\textcopyright} 2022 by the authors. Licensee MDPI, Basel, Switzerland.",

year = "2022",

month = jul,

doi = "10.3390/dj10070128",

language = "English",

volume = "10",

pages = "1--15",

journal = "Dentistry Journal",

issn = "2304-6767",

publisher = "Multidisciplinary Digital Publishing Institute (MDPI)",

number = "7",

}

Howe, BJ, Pendleton, C, Withanage, MHH, Childs, CA, Zeng, E, van Wijk, A, Hermus, R, Padilla, C, Hecht, JT, Poletta, FA, Orioli, IM, Buxó-Martínez, CJ, Deleyiannis, F, Vieira, AR, Butali, A, Valencia-Ramirez, C, Muñeton, CR, Wehby, GL, Weinberg, SM, Marazita, ML, Uribe, LMM & Xie, XJ 2022, 'Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis', Dentistry Journal, vol. 10, no. 7, 128, pp. 1-15. https://doi.org/10.3390/dj10070128

TY - JOUR

T1 - Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code

T2 - A Meta-Analysis

AU - Howe, Brian J.

AU - Pendleton, Chandler

AU - Withanage, Miyuraj Harishchandra Hikkaduwa

AU - Childs, Christopher A.

AU - Zeng, Erliang

AU - van Wijk, Arjen

AU - Hermus, Ruurd

AU - Padilla, Carmencita

AU - Hecht, Jacqueline T.

AU - Poletta, Fernando A.

AU - Orioli, Iêda M.

AU - Buxó-Martínez, Carmen J.

AU - Deleyiannis, Frederic

AU - Vieira, Alexandre R.

AU - Butali, Azeez

AU - Valencia-Ramirez, Consuelo

AU - Muñeton, Claudia Restrepo

AU - Wehby, George L.

AU - Weinberg, Seth M.

AU - Marazita, Mary L.

AU - Uribe, Lina M.Moreno

AU - Xie, Xian Jin

N1 - Special Issue: Dentistry Journal: 10th Anniversary. Funding Information: Grant support was provided by: K08 DE028012: “Identifying Phenomic Patterns of Dental Anomalies in Orofacial Clefting” (University of Iowa as Primary Awardee). R01 DE106148: “Ex-tending the Phenotype of Non-Syndromic Orofacial Clefts” (University of Pittsburgh as Primary Awardee). R01 DE014667: “Cleft Lip Genetics: A Multicenter International Consortium” (University of Iowa as Primary Awardee). R37-DE-08559: “Molecular Genetic Epidemiology of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R01 DD000295: “Health Outcomes and Improved Phenotypic Characterization of Cleft Lip and Palate” (University of Iowa as Primary Awardee). R00 DE024571: “Genetic and Environmental Risks for Oral Clefts in Puerto Rican Hispanics” (University of Puerto Rico as primary awardee). S21 MD001830: “Hispanics in Research Capability: SOHP and SOM Partnership” (University of Puerto Rico as primary awardee). U54 MD007587: “Puerto Rico Clinical and Translational Research Consortium” (University of Puerto Rico as primary awardee). R00 DE022378: ”Genetic Studies of Non-Syndromic Clefts in Populations of African De-scent” (University of Iowa as Primary Awardee). R01 DE02830: “Refining the Genetic and Genomic Architecture of Non-Syndromic Orofacial Clefting” (University of Iowa as Primary Awardee). Robert Wood Johnson Foundation 72429: “Genome-Wide Association Studies for Non-Syndromic Clefts in sub-Saharan African Populations”. Publisher Copyright: © 2022 by the authors. Licensee MDPI, Basel, Switzerland.

PY - 2022/7

Y1 - 2022/7

N2 - Individuals with orofacial clefting (OFC) have a higher prevalence of tooth agenesis (TA) overall. Neither the precise etiology of TA, nor whether TA occurs in patterns that differ by gender or cleft type is yet known. This meta-analysis aims to identify the spectrum of tooth agenesis patterns in subjects with non-syndromic OFC and controls using the Tooth Agenesis Code (TAC) program. An indexed search of databases (PubMed, EMBASE, and CINAHL) along with cross-referencing and hand searches were completed from May to June 2019 and re-run in February 2022. Additionally, unpublished TAC data from 914 individuals with OFC and 932 controls were included. TAC pattern frequencies per study were analyzed using a random effects meta-analysis model. A thorough review of 45 records retrieved resulted in 4 articles meeting eligibility criteria, comprising 2182 subjects with OFC and 3171 controls. No TA (0.0.0.0) was seen in 51% of OFC cases and 97% of controls. TAC patterns 0.2.0.0, 2.0.0.0, and 2.2.0.0 indicating uni-or bi-lateral missing upper laterals, and 16.0.0.0 indicating missing upper right second premolar, were more common in subjects with OFC. Subjects with OFC have unique TA patterns and defining these patterns will help increase our understanding of the complex etiology underlying TA.

AB - Individuals with orofacial clefting (OFC) have a higher prevalence of tooth agenesis (TA) overall. Neither the precise etiology of TA, nor whether TA occurs in patterns that differ by gender or cleft type is yet known. This meta-analysis aims to identify the spectrum of tooth agenesis patterns in subjects with non-syndromic OFC and controls using the Tooth Agenesis Code (TAC) program. An indexed search of databases (PubMed, EMBASE, and CINAHL) along with cross-referencing and hand searches were completed from May to June 2019 and re-run in February 2022. Additionally, unpublished TAC data from 914 individuals with OFC and 932 controls were included. TAC pattern frequencies per study were analyzed using a random effects meta-analysis model. A thorough review of 45 records retrieved resulted in 4 articles meeting eligibility criteria, comprising 2182 subjects with OFC and 3171 controls. No TA (0.0.0.0) was seen in 51% of OFC cases and 97% of controls. TAC patterns 0.2.0.0, 2.0.0.0, and 2.2.0.0 indicating uni-or bi-lateral missing upper laterals, and 16.0.0.0 indicating missing upper right second premolar, were more common in subjects with OFC. Subjects with OFC have unique TA patterns and defining these patterns will help increase our understanding of the complex etiology underlying TA.

KW - cleft lip and palate

KW - hypodontia

KW - machine learning

KW - phenotype

UR - http://www.scopus.com/inward/record.url?scp=85133844939&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85133844939&partnerID=8YFLogxK

U2 - 10.3390/dj10070128

DO - 10.3390/dj10070128

M3 - Review article

AN - SCOPUS:85133844939

SN - 2304-6767

VL - 10

SP - 1

EP - 15

JO - Dentistry Journal

JF - Dentistry Journal

IS - 7

M1 - 128

ER -

Tooth Agenesis Patterns in Orofacial Clefting Using Tooth Agenesis Code: A Meta-Analysis

Abstract

Bibliographical note

Funding

Keywords

Access to Document

Persistent URL (handle)

Other files and links

Fingerprint

Cite this