Dental Subphenotypes in Infants With Orofacial Clefts—A Longitudinal Population-Based Retrospective Radiographic Study of the Primary and Secondary Dentitions

2021 ◽  
pp. 105566562199014
Author(s):  
Mimi Yow ◽  
Nuno V. Hermann ◽  
Yuan Wei ◽  
Agneta Karsten ◽  
Sven Kreiborg
Keyword(s):  
Cleft Lip ◽  
Population Based ◽  
Dental Anomalies ◽  
Orofacial Clefts ◽  
Developmental Patterns ◽  
Dental Maturation ◽  
Northern European ◽  
Maxillary Lateral Incisor ◽  
Isolated Cleft Palate ◽  
Historical Controls

Objectives: To determine the developmental patterns of primary and secondary dentitions in infants with orofacial clefts. Design: Retrospective, longitudinal, population-based cohort study. Materials: Longitudinal records and radiographs of 192 nonsyndromic Northern European infants with isolated unilateral cleft lip (UCL, n = 111) and isolated cleft palate (CP, n = 81). Methods: Radiographic assessments of primary and secondary dentition anomalies and dental maturation, by gender and cleft severity for comparisons between the groups and with historical controls. Results: In infants with UCL, the frequencies of dental anomalies were high in both primary (38.7%) and secondary (18.0%) dentitions. Primary and secondary dentition anomalies were not observed in infants with CP and different in the UCL group ( P = .003). Risk differences involved primary supernumerary teeth ( P = .0001) and talon cusp formation ( P = .0001), and secondary tooth agenesis ( P = .001) of the maxillary lateral incisor on the side of the cleft lip. Delayed primary and secondary dental maturation occurred in the UCL and CP groups, greater in infants with UCL ( P < .0001). Primary and secondary dental maturation featured sexual dimorphism with greater delay in males (UCL, P < .0001; CP, .0001 > P = .001). The effect of cleft severity on dental maturation was significant in infants with UCL ( P = .0361) and CP ( P = .0175) in the primary but not in the secondary dentition. Conclusions: There were different dental anomalies in the primary and secondary dentitions in operated infants with UCL and no dental anomalies in unoperated infants with CP. Dental maturation was delayed in infants with UCL and CP with greater delay in males compared to females.

scholarly journals Deep orofacial phenotyping of population-based infants with isolated cleft lip and isolated cleft palate

2020 ◽  
Vol 10 (1) ◽  
Author(s):  
Mimi Yow ◽  
Nuno V. Hermann ◽  
Yuan Wei ◽  
Agneta Karsten ◽  
Sven Kreiborg
Keyword(s):  
Cleft Palate ◽  
Cleft Lip ◽  
Population Based ◽  
Tooth Agenesis ◽  
Orofacial Clefts ◽  
Dental Epithelium ◽  
Pre Treatment ◽  
Isolated Cleft Palate ◽  
Dental Maturity ◽  
Deep Phenotyping

AbstractIsolated orofacial clefts (OFC) are common with poorly understood aetiology. Heterogeneous phenotypes and subphenotypes confound aetiological variant findings. To improve OFC phenome understanding, population-based, consecutive, pre-treatment infants with isolated unilateral cleft lip (UCL, n = 183) and isolated cleft palate (CP, n = 83) of similar ancestry were grouped for deep phenotyping. Subphenotypes stratified by gender and cleft severity were evaluated for primary dental malformations and maturation using radiographs. We found that cleft severity and tooth agenesis were inadequate to distinguish heterogeneity in infants with UCL and CP. Both groups featured slow dental maturity, significantly slower in males and the UCL phenotype. In 32.8% of infants with UCL, supernumerary maxillary lateral incisors were present on the cleft lip side, but not in infants with CP, suggesting a cleft dental epithelium and forme fruste cleft dentoalveolus of the UCL subphenotype. The findings underscored the importance of deep phenotyping to disclose occult OFC subphenotypes.

A Population-Based Evaluation of Antenatal Diagnosis of Orofacial Clefts

2008 ◽  
Vol 45 (2) ◽  
pp. 148-153 ◽  
Author(s):  
Kathleen A. Russell ◽  
Victoria M. Allen ◽  
Mary E. MacDonald ◽  
Kirsten Smith ◽  
Linda Dodds
Keyword(s):  
Nova Scotia ◽  
Cleft Palate ◽  
Cleft Lip ◽  
Chromosomal Abnormalities ◽  
Antenatal Diagnosis ◽  
Population Based ◽  
Orofacial Clefts ◽  
Antenatal Detection ◽  
Type Mode ◽  
Isolated Cleft Palate

Objectives: To evaluate the changes in prevalence and antenatal detection of cleft lip with or without cleft palate and isolated cleft palate and to describe the association between anomalies and rates of antenatal diagnosis in Nova Scotia from 1992 to 2002. Design: This population-based cohort study employed the Nova Scotia Atlee Perinatal Database, the Fetal Anomaly Database, and IWK Cleft Palate Database in Halifax, Nova Scotia, Canada. Outcome Measures: Cleft type, mode of diagnosis, and associated abnormalities of orofacial clefts for liveborn infants, stillbirths, and second trimester terminations of pregnancy between 1992 and 2002 were determined. Results: There were 225 fetuses identified as having orofacial clefts. The overall prevalence of clefts was 2.1 in 1000 live births, and this prevalence did not change with time. The overall antenatal detection of cleft lip with or without cleft palate was 23%; however, there was improvement in detection of cleft lip with or without cleft palate from the years 1992 to 1996 (14%) to the years 1997 to 2002 (30%, p  =  .02). No isolated cleft palates were detected antenatally. Associated structural anomalies were seen in 34.2% of cases with orofacial clefts, and chromosomal abnormalities were associated with 9.8%. Conclusions: The prevalence of orofacial clefts in Nova Scotia has not changed from 1992 to 2002. The proportion of antenatally diagnosed cleft lip with or without cleft palate in Nova Scotia is consistent with rates reported in the literature and has increased from 1992 to 2002.

Socioeconomic Status and Orofacial Clefts in Scotland, 1989 to 1998

2003 ◽  
Vol 40 (5) ◽  
pp. 481-485 ◽  
Author(s):  
J. D. Clark ◽  
P. A. Mossey ◽  
L. Sharp ◽  
J. Little
Keyword(s):  
Risk Factors ◽  
Socioeconomic Status ◽  
Cleft Lip ◽  
Population Based ◽  
Orofacial Clefts ◽  
Smoking During Pregnancy ◽  
Live Births ◽  
Strong Positive Relationship ◽  
The Relationship ◽  
Deprived Areas

Objective The purpose of this study was to investigate the association between socioeconomic status and orofacial clefts (OFC) in Scotland. Design Study of prevalence at birth over a 10-year period using an area-based measure of material deprivation. Setting Population-based study throughout Scotland. Participants Eight hundred thirty-four live births with OFC born between January 1, 1989, and December 31, 1998, ascertained from the nationwide register of the Cleft Service in Scotland, compared with the total 603,825 live births in Scotland in this period. Results There was a strong positive relationship whereby the prevalence of OFC at birth increased with increasing deprivation. This trend was statistically significant for cleft lip and/or palate (CL[P]: p = .016) but not for cleft palate (CP; p = .078). For each type of cleft, the relative risk among those resident in the most deprived areas, compared with those resident in the least deprived areas, was 2.33. Conclusions The association between OFC and socioeconomic status is consistent with a report for an earlier period in a smaller part of Scotland. Unlike the earlier study, this pattern appears to be stronger for CL(P) than for CP. These observations do not appear to be an artifact of recording. It is possible that they reflect the association between deprivation and risk factors for OFC, most likely tobacco smoking during pregnancy. Because the relationship between OFC and socioeconomic status appears to have been virtually unstudied in other populations, it would be valuable to investigate this relationship elsewhere and determine whether known risk factors account for the relationship.

Timing of Exchange of the Maxillary Deciduous and Permanent Teeth in Boys with Three Types of Orofacial Clefts

1996 ◽  
Vol 33 (4) ◽  
pp. 318-323 ◽  
Author(s):  
Miroslav Peterka ◽  
Renata Peterková ◽  
Zbyněk Likovský
Keyword(s):  
Permanent Teeth ◽  
Control Group ◽  
Orofacial Clefts ◽  
Maxillary Canine ◽  
Second Molar ◽  
Developmental Disturbances ◽  
Maxillary Lateral Incisor ◽  
Maxillary Premolars ◽  
Dental Plaster ◽  
Isolated Cleft Palate

Timing of exchange of the deciduous and permanent maxillary teeth was investigated using dental plaster casts of 163 boys with total unilateral cleft (UCLP), 82 boys with bilateral cleft (BCLP), and 97 boys with isolated cleft palate (CP). All patients were treated at the Prague Plastic Surgery Clinic. The results were compared with a control group of 294 schoolboys. To evaluate the course of eruption, the proportion of each erupted teeth in each year of age was employed. In boys with UCLP, eruption of the permanent maxillary lateral incisors and the permanent maxillary second molar was retarded on the cleft side. On the non-affected side, no delay of eruption was observed, but earlier eruption was found in the permanent maxillary canine and in the permanent maxillary first and second premolars. In boys with BCLP, the highest retardation of eruption was found in the permanent maxillary lateral incisor and in the permanent maxillary first molar. The permanent maxillary canine and both permanent maxillary premolars erupted earlier than in the control group. In boys with CP, only the permanent maxillary central incisors erupted earlier. The maxillary deciduous canines and the second molars were both lost early. We conclude that the developmental disturbances of the maxillary jaw and teeth in patients with orofacial clefts are also associated with alteration of timing of dental exchange.

Maternal Smoking during Early Pregnancy, GSTP1 and EPHX1 Variants, and Risk of Isolated Orofacial Clefts

2007 ◽  
Vol 44 (4) ◽  
pp. 366-373 ◽  
Author(s):  
Dorian Ramirez ◽  
Edward J. Lammer ◽  
David M. Iovannisci ◽  
Cecile Laurent ◽  
Richard H. Finnell ◽  
...  
Keyword(s):  
Cleft Palate ◽  
Maternal Smoking ◽  
Cleft Lip ◽  
Detoxification Enzymes ◽  
Glutathione S Transferase ◽  
Orofacial Clefts ◽  
Functional Polymorphisms ◽  
Increased Risk ◽  
Cleft Lip Palate ◽  
Isolated Cleft Palate

Objective: To examine the interactions between four fetal xenobiotic metabolizing gene polymorphisms, maternal cigarette smoking, and risk for oral cleft defects. Design and Participants: California population–based case-control study of 431 infants born with isolated orofacial clefts and 299 nonmalformed controls. Main Outcome Measures: Infants were genotyped for functional polymorphisms of the detoxification enzymes microsomal epoxide hydrolase-1 (EPHX1 T→C [Tyr113His], and A→G [His139Arg]), and glutathione-S transferase Pi-1 (GSTP1 A→G [Ile105Val] and C→T [Ala114Val]), and risks for cleft outcomes were measured for gene only and gene-maternal smoking effects. Results: Although smoking was associated with an increased risk for isolated cleft lip ± palate, we found no independent associations of genotypes of EPHX1-codon 113 or GSTP1-codon 105 polymorphisms for either isolated cleft lip ± palate or isolated cleft palate. The heterozygote genotype for the EPHX1-codon 139 polymorphism was associated with an increased risk of isolated cleft palate (odds ratio = 1.6 [95% confidence interval, 1.0 to 2.6]). Infant EPHX1 and GTSP1 polymorphic variants did not appreciably alter the risks for clefts associated with maternal smoking, nor were any EPHX1 combined genotype-specific risks found. Infant genotypes of the GSTP1-codon 105 polymorphism, combined with glutathione-S-transferase-μ-1 null genotypes, did not appreciably alter the risk of orofacial clefts. Conclusions: Our results suggest that genetic variation of the detoxification enzymes EPHX1 and GSTP1 did not increase the risks of orofacial clefting, nor do they influence the risks associated with maternal smoking.

Birth Defect Co-Occurrence Patterns Among Infants With Cleft Lip and/or Palate

2021 ◽  
pp. 105566562110100
Author(s):  
Maria Luisa Navarro Sanchez ◽  
Renata H. Benjamin ◽  
Laura E. Mitchell ◽  
Peter H. Langlois ◽  
Mark A. Canfield ◽  
...  
Keyword(s):  
Cleft Palate ◽  
Birth Defects ◽  
Birth Defect ◽  
Cleft Lip ◽  
Population Based ◽  
Orofacial Clefts ◽  
Developmental Mechanisms ◽  
Using Data ◽  
Occurrence Patterns ◽  
Population Based Registry

Objective: To investigate 2- to 5-way patterns of defects co-occurring with orofacial clefts using data from a population-based registry. Design: We used data from the Texas Birth Defects Registry for deliveries between 1999 and 2014 to Texas residents, including 1884 cases with cleft palate (CP) and 5289 cases with cleft lip with or without cleft palate (CL±P) without a known syndrome. We identified patterns of defects co-occurring with CP and with CL±P observed more frequently than would be expected if these defects occurred independently. We calculated adjusted observed-to-expected ( O/ E) ratios to account for the known tendency of birth defects to cluster nonspecifically. Results: Among infants without a syndrome, 23% with CP and 21% with CL±P had at least 1 additional congenital anomaly. Several combinations of defects were observed much more often than expected. For example, the combination of CL±P, congenital hydrocephaly, anophthalmia, and other nose anomalies had an O/ E ratio of 605. For both CP and CL±P, co-occurrence patterns with the highest O/ E ratios involved craniofacial and brain abnormalities, and many included the skeletal, cardiovascular, and renal systems. Conclusions: The patterns of defects we observed co-occurring with clefts more often than expected may help improve our understanding of the relationships between multiple defects. Further work to better understand some of the top defect combinations could reveal new phenotypic subgroups and increase our knowledge of the developmental mechanisms that underlie the respective defects.

scholarly journals Frequency of Additional Congenital Dental Anomalies in Children with Cleft Lip, Alveolar and Palate

2020 ◽  
Vol 9 (12) ◽  
pp. 3813
Author(s):  
Przemysław Pastuszak ◽  
Izabella Dunin-Wilczyńska ◽  
Agnieszka Lasota
Keyword(s):  
Cleft Lip ◽  
Alveolar Bone ◽  
Dental Anomalies ◽  
Maxillary Expansion ◽  
Supernumerary Teeth ◽  
Maxillary Canine ◽  
Panoramic Radiographs ◽  
Maxillary Lateral Incisor ◽  
Applied Approach ◽  
Medical University

The aim of the study was to assess the risk of incidence of additional congenital dental anomalies in children with nonsyndromic cleft lip, alveolar and/without palate (CL/P). Hypodontia, hyperdontia and canines impaction was recognized. From patients with CL/P treated at the Clinic of Congenital Facial Deformities in Chair and Department of Jaw Orthopedics Medical University of Lublin, 56 subjects were randomly selected. On the panoramic radiographs taken at the age of 8–12 years, the angle of upper unerupted canines was measured using Westerlund’s recommendations. The supernumerary teeth and hypodontia were checked. The procedures: maxillary expansion, secondary alveolar bone graft (SABG) and extraction of primary canines were noted. The frequency of canines impaction was 5.36%. Hypodontia was found in 37.5% of patients; hyperdontia was present in 23.21% of patients. No influence of procedures (expansion of the maxilla, SABG, deciduous canines extraction) on permanent maxillary canine eruption was proved. Patients with CL/P are exposed to the unfavorable position of unerupted maxillary canines most frequently in the cleft area of complete cleft. Maxillary lateral incisor on the cleft side is most frequently affected with congenital anomaly. Hypodontia and hyperdontia do not influence maxillary canine impaction. Good clinical result was achieved with an applied approach, which should be widely introduced.

scholarly journals Refractive Errors and Strabismus In Patients with Orofacial Clefts

2021 ◽  
Author(s):  
Camille Catarina Artuso ◽  
Ana Kelly Fernandes ◽  
Luisa Moreira Hopker ◽  
Ricardo Mokross Fernandes ◽  
Renato da Silva Freitas ◽  
...  
Keyword(s):  
Refractive Error ◽  
Medical Records ◽  
Cleft Lip ◽  
Cleft Lip And Palate ◽  
Refractive Errors ◽  
Orofacial Clefts ◽  
Significant Difference ◽  
Eye Examination ◽  
The Mean ◽  
Isolated Cleft Palate

Abstract Purpose: To evaluate the prevalence of refractive errors and strabismus in patients with orofacial clefts. Methods: This retrospective study analyzed the medical records of 54 patients with orofacial clefts between August 2018 and March 2020. A complete eye examination was performed, including visual acuity assessment on a logMAR scale, anterior biomicroscopy, cycloplegic refraction, eye motility examination, and indirect ophthalmoscopy. Results: The mean age of the patients at presentation was 9.47 years. Twentythree (42.59%) patients had isolated cleft palate (CP), 10 (18.52%) had cleft lip (CL), and 21 (38.89%) had cleft lip and palate (CLP). The mean spherical equivalent was 1.30D (±1.56) in CL, 0.32D (±2.24) in CLP, and 0.62D (±3.76) in CP. The prevalence of refractive error, either spherical or cylinder >0.5 was 88%. The most common refractive error was hyperopia (60%), followed by astigmatism (54%) and myopia (16%). Overall, 52.63% of the patients were prescribed glasses. No statistically significant difference was observed between the groups with respect to the need for prescription of glasses (p=0.6753). There were 15 patients with some type of strabismus, and other ophthalmological changes were observed in 13 patients. Conclusion: In this population with orofacial clefts, the prevalence of refractive errors and strabismus was 88% and 22%, respectively.

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