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Akıncı O, Oğuz Eİ, Bozkurt P, Kadıoğlu MB, Ocak M, Orhan K, Kişnişçi RŞ. Effect of cleft palate type and manufacturing method on feeding plate adaptation: A volumetric micro-computed tomography analysis. J Prosthodont 2025. [PMID: 39871456 DOI: 10.1111/jopr.14027] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/15/2024] [Accepted: 01/10/2025] [Indexed: 01/29/2025] Open
Abstract
PURPOSE Feeding plates for cleft palate patients have been used by clinicians for many years to temporarily close the oro-nasal communication until definitive treatment with surgical techniques. The current in vitro study aimed to evaluate the adaptation of the feeding plates manufactured by two different techniques for three cleft types. MATERIALS AND METHODS Feeding plates were manufactured with conventional compression molding (CM) and 3-dimensional (3D) additive manufacturing on main models representing bilateral cleft, unilateral right, and unilateral left cleft types (n = 10). The 3D volumetric space between the feeding plate and the corresponding main model was measured by micro-CT to evaluate the adaptation. The adaptation of the plates was assessed based on three different measurement regions: anterior, left, and right. Repeated measure analysis of variance (ANOVA), three factorial ANOVA, and post hoc Bonferroni tests were used as statistical analysis (α = 0.05). RESULTS CM groups showed higher volumetric space measurements between the base and master model than 3D groups regardless of measurement region and cleft type, which refers to misfit (p ˂ 0.05). Cleft type differed in the adaptation of 3D groups yet not in CM groups (p ˂ 0.05). The volumetric space evaluation for the right measurement region resulted in higher values regardless of manufacturing method and cleft type (p ˂ 0.05). CONCLUSION Considering that 3D-printed feeding plates showed better adaptation compared to conventionally manufactured plates for all cleft types, 3D printing can be suggested as the manufacturing method of choice for feeding plates.
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Affiliation(s)
- Osman Akıncı
- Graduate Oral and Maxillofacial Surgeon, Antalya, Turkey
| | - Ece İrem Oğuz
- Department of Prosthodontics, Faculty of Dentistry, Ankara University, Ankara, Turkey
| | | | | | - Mert Ocak
- Department of Anatomy, Faculty of Dentistry, Ankara University, Ankara, Turkey
| | - Kaan Orhan
- Department of Dentomaxillofacial Radiology, Ankara University, Ankara, Turkey
| | - Reha Şükrü Kişnişçi
- Department of Oral and Maxillofacial Surgery, Lokman Hekim University, Ankara, Turkey
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Beyeler J, Lauener A, Katsaros C, La Scala GC, Degen M. Epidemiologic trends of cleft lip and/or palate in Switzerland. BMC Oral Health 2025; 25:114. [PMID: 39844198 PMCID: PMC11756186 DOI: 10.1186/s12903-025-05500-w] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/13/2024] [Accepted: 01/17/2025] [Indexed: 01/24/2025] Open
Abstract
BACKGROUND Epidemiologic data on the number of cleft lip and/or palate (orofacial cleft (OFC)) births in Switzerland are currently sparse. However, this knowledge is essential for better understanding the etiologies underlying the various cleft phenotypes and providing expectant parents with the best possible healthcare planning and counseling. METHODS This is the first descriptive study to report data on the prevalence of the various cleft types, their sex, and regional distributions in Switzerland. Data for the years 1998-2021 were obtained from the Swiss Federal Statistical Office. Due to the notable initial underreporting of cleft cases from 1998 to 2006, this period was omitted from the final analyses. RESULTS Between 2007 and 2021, the prevalence of all Swiss OFC cases per 10,000 live births was 12.5, with a stable trend. Cleft lip was the least common anomaly. Except for cleft palate, which was more common in females, males were generally more affected by OFC than females. There was no discernible regional trend for any of the malformations, even though the prevalence differed throughout the seven Swiss regions. CONCLUSIONS This study presents the first descriptive epidemiologic profiles for OFCs in Switzerland and emphasizes the importance of nationwide OFC registries with an accurate and reliable reporting system for the benefit of current and future patients with clefts, their parents or caregivers, and society as a whole.
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Affiliation(s)
- Joël Beyeler
- Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Bern, Switzerland
- Department of Restorative, Preventive and Pediatric Dentistry, School of Dental Medicine, University of Bern, Bern, Switzerland
| | - Anic Lauener
- Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Bern, Switzerland
| | - Christos Katsaros
- Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Bern, Switzerland
- Laboratory for Oral Molecular Biology, Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Freiburgstrasse 3, Bern, 3010, Switzerland
| | - Giorgio C La Scala
- Pediatric Plastic Surgery, Division of Pediatric Surgery, Department of Pediatrics, University Hospital of Geneva, Geneva, Switzerland
| | - Martin Degen
- Laboratory for Oral Molecular Biology, Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Freiburgstrasse 3, Bern, 3010, Switzerland.
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Sofi-Mahmudi A, Shamsoddin E, Khademioore S, Khazaei Y, Vahdati A, Tovani-Palone MR. Global, regional, and national survey on burden and Quality of Care Index (QCI) of orofacial clefts: Global burden of disease systematic analysis 1990-2019. PLoS One 2025; 20:e0317267. [PMID: 39774532 PMCID: PMC11709310 DOI: 10.1371/journal.pone.0317267] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2024] [Accepted: 12/25/2024] [Indexed: 01/11/2025] Open
Abstract
BACKGROUND Orofacial clefts are the most common craniofacial anomalies that include a variety of conditions affecting the lips and oral cavity. They remain a significant global public health challenge. Despite this, the quality of care for orofacial clefts has not been investigated at global and country levels. OBJECTIVE We aimed to measure the quality-of-care index (QCI) for orofacial clefts worldwide. METHODS We used the 2019 Global Burden of Disease data to create a multifactorial index (QCI) to assess orofacial clefts globally and nationally. By utilizing data on incidence, prevalence, years of life lost, and years lived with disability, we defined four ratios to indirectly reflect the quality of healthcare. Subsequently, we conducted a principal component analysis to identify the most critical variables that could account for the observed variability. The outcome of this analysis was defined as the QCI for orofacial clefts. Following this, we tracked the QCI trends among males and females worldwide across various regions and countries, considering factors such as the socio-demographic index and World Bank classifications. RESULTS Globally, the QCI for orofacial clefts exhibited a consistent upward trend from 1990 to 2019 (66.4 to 90.2) overall and for females (82.9 to 94.3) and males (72.8 to 93.6). In the year 2019, the top five countries with the highest QCI scores were as follows: Norway (QCI = 99.9), Ireland (99.4), France (99.4), Germany (99.3), the Netherlands (99.3), and Malta (99.3). Conversely, the five countries with the lowest QCI scores on a global scale in 2019 were Somalia (59.1), Niger (67.6), Burkina Faso (72.6), Ethiopia (73.0), and Mali (74.4). Gender difference showed a converging trend from 1990 to 2019 (optimal gender disparity ratio (GDR): 123 vs. 163 countries), and the GDR showed a move toward optimization (between 0.95 and 1.05) in the better and worse parts of the world. CONCLUSION Despite the positive results regarding the QCI for orofacial clefts worldwide, some countries showed a slight negative trend.
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Affiliation(s)
- Ahmad Sofi-Mahmudi
- National Pain Centre, Department of Anesthesia, McMaster University, Hamilton, ON, Canada
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
- Seqiz Health Network, Kurdistan University of Medical Sciences, Seqiz, Kurdistan
| | - Erfan Shamsoddin
- Cochrane Iran Associate Centre, National Institute for Medical Research Development (NIMAD), Tehran, Iran
| | - Sahar Khademioore
- Department of Health Research Methods, Evidence and Impact, McMaster University, Hamilton, ON, Canada
| | - Yeganeh Khazaei
- Department of Statistics, Statistical Consultation Unit, StaBLab, LMU Munich, Munich, Germany
| | - Amin Vahdati
- Centre for Biostatistics, University of Manchester, Manchester, United Kingdom
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Li S, Zhu H, Yang C, Wang C, Liu J, Jin L, Li Z, Ren A, Wang L. Prenatal co-exposure to phthalate metabolites and bisphenols among non-syndromic cleft lip and/or palate in offspring. ENVIRONMENTAL POLLUTION (BARKING, ESSEX : 1987) 2024; 362:125001. [PMID: 39322108 DOI: 10.1016/j.envpol.2024.125001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 06/13/2024] [Revised: 09/16/2024] [Accepted: 09/19/2024] [Indexed: 09/27/2024]
Abstract
Phthalate metabolites and bisphenols can cause adverse pregnancy outcomes. However, there is no study to evaluate the associations of prenatal exposure to phthalate metabolites and bisphenols with non-syndromic cleft lip and/or palate (NSCL/P) risk in offspring. A population-based case-control study was conducted in a multicenter setting from 2005 to 2021, enrolling 448 pregnant women. Seven phthalate metabolites and six bisphenols were quantified in placenta using liquid chromatography-tandem mass spectrometry. In the logistic regression analysis, high levels of mono-ethyl phthalate, mono-cyclohexyl phthalate, mono-octyl phthalate, bisphenol A, bisphenol AF, bisphenol AP, and fluorene-9-bisphenol were associated with increased NSCL/P risk with odds ratios (95% confidence intervals) of 1.86(1.07,3.25), 6.56(3.47,12.39), 8.49(4.44,16.24), 8.34(4.32,16.08), 3.19(1.81,5.62), 2.78(1.59,4.86), and 5.16(2.82,9.44). The Bayesian kernel machine regression model revealed that co-exposure to phthalate metabolites and bisphenols was associated with increased NSCL/P risk. Similarly, quantile-based g-computation analysis indicated that each quantile increase in mixture concentration was positively related to higher risk for NSCL/P [odds ratio (95% confidence interval) = 2.98(1.97,4.51)]. This study provides novel evidence that prenatal single and co-exposure to phthalate metabolites and bisphenols were associated with increased NSCL/P risk, suggesting that exposure to phthalate metabolites and bisphenols during pregnancy should be minimized to reduce the incidence of NSCL/P in offspring.
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Affiliation(s)
- Sainan Li
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Haiyan Zhu
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Chen Yang
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Chengrong Wang
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Jufen Liu
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Lei Jin
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Zhiwen Li
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Aiguo Ren
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China
| | - Linlin Wang
- Institute of Reproductive and Child Health/National Health Commission Key Laboratory of Reproductive Health, Peking University, Beijing, 100191, China; Department of Epidemiology and Biostatistics, School of Public Health, Peking University, Beijing, 100191, China.
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Sajovic J, Drevenšek G, Plut A, Eberlinc A, Kosmač Vrabec M, Setnikar Lesjak A, Selič Zupančič P, Drevenšek M. Beyond the face: An interdisciplinary evaluation of satisfaction with appearance in young people with orofacial clefts. iScience 2024; 27:110738. [PMID: 39290840 PMCID: PMC11406071 DOI: 10.1016/j.isci.2024.110738] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/12/2024] [Revised: 06/18/2024] [Accepted: 08/12/2024] [Indexed: 09/19/2024] Open
Abstract
Orofacial clefts are the most common congenital anomaly of the face, and they significantly affect appearance. The combined effects of demographics, psychology, neurophysiology, and cleft characteristics to explain satisfaction with appearance in young people with a cleft have not yet been comprehensively studied in an interdisciplinary manner. We found that interpersonal difficulties, age, and conscientiousness were significant explanatory factors for satisfaction with appearance (tinterpersonal difficulties = -3.022, p = 0.006; tage = -3.563, p = 0.016; tconscientiousness = 4.161, p = 0.003); the model explained 50% of variance in satisfaction with appearance (R2 Adjusted = 0.504, Fvs. constant = 4.05, p = 0.00117). Furthermore, frontal alpha asymmetry was complexly intertwined with other variables, affecting the overall accuracy of the model, but explaining only 10.5% of variance in satisfaction with appearance when used as a factor alone. The results show that an interdisciplinary approach can substantially expand our understanding of the factors influencing self-perception in young people with orofacial clefts.
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Affiliation(s)
- Jakob Sajovic
- Faculty of Medicine, University of Ljubljana, 1000 Ljubljana, Slovenia
- Department of Orthodontics, University Medical Centre Ljubljana, 1000 Ljubljana, Slovenia
| | - Gorazd Drevenšek
- Faculty of Medicine, University of Ljubljana, 1000 Ljubljana, Slovenia
| | - Alja Plut
- Department of Orthodontics, University Medical Centre Ljubljana, 1000 Ljubljana, Slovenia
| | - Andreja Eberlinc
- Department of Maxillofacial and Oral Surgery, University Medical Centre Ljubljana, 1000 Ljubljana, Slovenia
| | - Manca Kosmač Vrabec
- Faculty of Mathematics, Natural Sciences and Information Technologies, University of Primorska, 6000 Koper, Slovenia
| | - Anina Setnikar Lesjak
- Department of Orthodontics, University Medical Centre Ljubljana, 1000 Ljubljana, Slovenia
| | - Polona Selič Zupančič
- Faculty of Medicine, University of Ljubljana, 1000 Ljubljana, Slovenia
- Faculty of Medicine, University of Maribor, 2000 Maribor, Slovenia
| | - Martina Drevenšek
- Faculty of Medicine, University of Ljubljana, 1000 Ljubljana, Slovenia
- Department of Orthodontics, University Medical Centre Ljubljana, 1000 Ljubljana, Slovenia
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Yan S, Yu Q, Zhou H, Huang R, Wang Y, Ma C, Guo F, Fu F, Li R, Li F, Jin X, Zhen L, Pan M, Li D, Liao C. Association of prenatal Cleft Lip and Palate ultrasound abnormalities with copy number variants at a single Chinese tertiary center. Ital J Pediatr 2024; 50:152. [PMID: 39169438 PMCID: PMC11337895 DOI: 10.1186/s13052-024-01720-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/07/2024] [Accepted: 08/06/2024] [Indexed: 08/23/2024] Open
Abstract
BACKGROUD A systematic analysis was conducted to investigate the molecular etiology of fetal cleft lip and/or palate (CL/P) and the association between various types of CL/P and copy number variations (CNVs), as well as their impact on birth outcomes. METHODS In this retrospective study conducted between January 2016 and July 2022, a cohort of pregnancies diagnosed with fetal CL/P was enrolled and comprehensive clinical data for all cases were extracted from our medical record database, including demographic data about the pregnancies, ultrasound findings, results of Chromosomal microarray (CMA), as well as relevant pregnant and perinatal outcomes. RESULTS Among the 358 cases, 32 clinically significant variants in 29 (8.1%) fetuses with CL/P were detected by CMA. In 338 singleton pregnancies, the diagnostic yield of CMA in the context of CL/P fetuses was determined to be 7.7% (26/338). CP cases exhibited a relatively higher prevalence of pathogenic/likely pathogenic CNVs at a rate of 25% (3/12), followed by CLP cases at 8.0% (23/288). Notably, the CL group did not demonstrate any pathogenic/likely pathogenic CNV findings among the examined cases (0/38). The diagnostic rate of clinically significant variants was notably higher in the non-isolated CL/P group than in the isolated CL/P group (11/33, 33.3% vs. 15/305, 4.9%, p < 0.001). Within the remaining 20 twin pregnancies, three clinically significant variants (15%) were observed. CONCLUSIONS This study provides powerful evidence supporting the efficacy of CMA as a valuable tool for facilitating the prenatal genetic diagnosis of fetal CL/P. The presence of CP and CLP in fetal cases demonstrated a relatively higher incidence of pathogenic/likely pathogenic CNVs. Moreover, when these cases were accompanied by additional ultrasound abnormalities, the likelihood of identifying diagnostic CNVs significantly increased. Conversely, cases of CL alone might not be associated with positive CNVs. The present data may significantly enhance prenatal diagnosis accuracy and facilitate informed genetic counseling for cases of fetal CL/P.
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Affiliation(s)
- Shujuan Yan
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Qiuxia Yu
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Hang Zhou
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Ruibin Huang
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - You Wang
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Chunling Ma
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Fei Guo
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Fang Fu
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Ru Li
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Fucheng Li
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Xiangyi Jin
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Li Zhen
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Min Pan
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Dongzhi Li
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China
| | - Can Liao
- Prenatal Diagnostic Center, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangzhou, Guangdong, 510620, China.
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Alkhathami AM, Alturki AT, Ulhaq MM, Al-Meshal O. Pattern of Cleft Lip and Palate Clefts at a Tertiary Care Hospital in Saudi Arabia. Cleft Palate Craniofac J 2024:10556656241267234. [PMID: 39094378 DOI: 10.1177/10556656241267234] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 08/04/2024] Open
Abstract
BACKGROUND Clefts of the lip and palate (CL/P) and cleft palate (CP) are the most common craniofacial congenital anomalies. Clefts are classified as syndromic and nonsyndromic. Nonsyndromic clefts have no known genetic causes. OBJECTIVES This study combines prospective and retrospective studies to review the patterns of CL/P and CP and associated syndromes and conditions in patients registered for CL/P surgery at a tertiary care pediatric center in our tertiary care hospital in Saudi Arabia. METHODS It included patient data from May 2015 through April 2023. Patient record forms and SPSS (IBM version 20.0) were used to collect and analyze data. A significance level of 5% was used, with p ≤ 0.05 considered statistically significant. RESULTS Of the 319 patients who met our inclusion criteria, 175 were male. Of the total, 99 had a left unilateral isolated cleft lip, 61 had a right unilateral isolated cleft lip, 69 had a bilateral cleft lip, and 90 had an isolated CP. Of the total, 140 had CL/P. Around 242 were nonsyndromic. The Chi-square test revealed a significant association between the prevalence of isolated CP and CLP and gender. The prevalence of left unilateral isolated cleft lip and bilateral and isolated CP was significantly associated with syndromic and nonsyndromic cases. CONCLUSION Males are more likely to be affected by orofacial clefts, which is consistent with the global trend. Isolated CP was the most common orofacial cleft. Within the sample, syndromes' association with orofacial clefts was significantly weaker than that of isolated and bilateral clefts.
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Affiliation(s)
- Ali M Alkhathami
- Department of Plastic Surgery, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Ahmed T Alturki
- Department of Plastic Surgery, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Muhammad M Ulhaq
- Department of Plastic Surgery, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Obaid Al-Meshal
- Department of Plastic Surgery, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
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Chwa ES, Stoehr JR, Gosain AK. Predictors of Adverse Outcomes Following Cleft Palate Repair: An Analysis of Over 2500 Patients Using International Smile Train Data. Cleft Palate Craniofac J 2024; 61:844-853. [PMID: 36594527 DOI: 10.1177/10556656221148901] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/04/2023] Open
Abstract
OBJECTIVE The objective of this study was to use data from Smile Train's global partner hospital network to identify patient characteristics that increase odds of fistula and postoperative speech outcomes. DESIGN Multi-institution, retrospective review of Smile Train Express database. SETTING 1110 Smile Train partner hospitals. PATIENTS/PARTICIPANTS 2560 patients. INTERVENTIONS N/A. MAIN OUTCOME MEASURE(S) Fistula occurrence, nasal emission, audible nasal emission with amplification (through a straw or tube) only, nasal rustle/turbulence, consistent nasal emission, consistent nasal emission due to velopharyngeal dysfunction, rating of resonance, rating of intelligibility, recommendation for further velopharyngeal dysfunction assessment, and follow-up velopharyngeal dysfunction surgery. RESULTS The patients were 46.6% female and 27.5% underweight by WHO standards. Average age at palatoplasty was 24.7 ± 0.5 months and at speech assessment was 6.8 ± 0.1 years. Underweight patients had higher incidence of hypernasality and decreased speech intelligibility. Palatoplasty when under 6 months or over 18 months of age had higher rates of affected nasality, intelligibility, and fistula formation. The same findings were seen in Central/South American and African patients, in addition to increased velopharyngeal dysfunction and fistula surgery compared to Asian patients. Palatoplasty technique primarily involved one-stage midline repair. CONCLUSIONS Age and nutrition status were significant predictors of speech outcomes and fistula occurrence following palatoplasty. Outcomes were also significantly impacted by location, demonstrating the need to cultivate longitudinal initiatives to reduce regional disparities. These results underscore the importance of Smile Train's continual expansion of accessible surgical intervention, nutritional support, and speech-language care.
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Affiliation(s)
- Emily S Chwa
- Northwestern University Feinberg School of Medicine, Chicago, IL, USA
| | - Jenna R Stoehr
- Division of Plastic and Reconstructive Surgery, University of South Florida, Tampa, IL, USA
| | - Arun K Gosain
- Northwestern University Feinberg School of Medicine, Chicago, IL, USA
- Division of Pediatric Plastic and Reconstructive Surgery, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA
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Verdier EF, Saloux AL, Azzis OM, Lebullenger RM, Davit-Béal TA, Brézulier DY. Bioglass 45S5, a relevant alternative to autogenous harvesting for secondary alveolar bone grafts in clefts? Retrospective study of one hundred surgeries. J Craniomaxillofac Surg 2024; 52:85-92. [PMID: 38129189 DOI: 10.1016/j.jcms.2023.12.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/01/2023] [Revised: 09/26/2023] [Accepted: 12/11/2023] [Indexed: 12/23/2023] Open
Abstract
The secondary alveolar bone grafting (SABG) step restores the continuity of the alveolar bone necessary for dentition. Faced with the complications of autografts, synthetic biomaterials such as Bioglass (BG) 45S5 have been proposed. The objective was to evaluate the success rate of SABG with the addition of BG 45S5 and to highlight the prognostic factors. Patients who underwent operation between 2015 and 2021 and had follow-up cone-beam computed tomography (CBCT) were analyzed. Multivariate analysis was performed to determine factors influencing radiographic success. A total of 102 SABG were analyzed. They were unilateral total cleft lip and palate (49, 48.0%). The mean age at surgery was 9.32 ± 3.09 years. Surgeries were performed mainly outside a syndromic context and without a family history after orthodontic preparation. The radiographic success rate at 1 year was 80.4%. Mixed dentition stage (odds ratio [OR] = 7.3, p = 0.024), absence of syndromic context (OR = 20.7, p = 0.024) and female sex (OR = 4.88, p = 0.021) were factors predictive of surgical success. The use of BG 45S5 instead of autograft is relevant for SABG, with a 1-year success rate of over 80%. The stage of mixed dentition, the absence of syndromic context, and female sex were factors for good prognosis.
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Affiliation(s)
| | | | - Olivier M Azzis
- CHU Rennes, Univ Rennes, Service de Chirurgie Pédiatrique, France
| | | | | | - Damien Y Brézulier
- CHU Rennes, Univ Rennes, Pôle Odontologie, France; Univ Rennes, ISCR UMR 6226, France.
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Imani MM, Shalchi M, Ahmadabadi G, Sadeghi M. Evaluation of matrix metalloproteinases (MMPs) and tissue inhibitors of metalloproteinases (TIMPs) in human cases with orofacial clefts: A systematic review. Int Orthod 2023; 21:100781. [PMID: 37301105 DOI: 10.1016/j.ortho.2023.100781] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2023] [Revised: 05/01/2023] [Accepted: 05/02/2023] [Indexed: 06/12/2023]
Abstract
INTRODUCTION The interaction between several cell populations or many genes and the coordination of multiple signal transmission pathways can lead to defects such as orofacial clefts (OFCs). Herein, a systematic review was designed to evaluate a group of important biomarkers (matrix metalloproteinases [MMPs] and tissue inhibitors of metalloproteinases [TIMPs]) in human cases with OFCs. MATERIAL AND METHODS Four databases including PubMed, Scopus, Web of Science, and Cochrane Library databases were searched until March 10, 2023, without any restriction. STRING, the protein-protein interaction (PPI) network software, was applied to investigate the functional interactions among the examined genes. The effect sizes including odds ratio (OR) dealing with a 95% confidence interval (CI), were extracted by the Comprehensive Meta-Analysis version 2.0 (CMA 2.0) software. RESULTS Thirty-one articles were entered into the systematic review that four articles were analyzed in the meta-analysis. Single studies reported that several polymorphisms of MMPs (rs243865, rs9923304, rs17576, rs6094237, rs7119194, and rs7188573); and TIMPs (rs8179096, rs7502916, rs4789936, rs6501266, rs7211674, rs7212662, and rs242082) had an association with OFC risk. There was no significant difference for MMP-3 rs3025058 polymorphism in allelic (OR: 0.832; P=0.490), dominant (OR: 1.177; P=0.873), and recessive (OR: 0.363; P=0.433) models and MMP-9 rs17576 polymorphism in an allelic model (OR: 0.885; P=0.107) between the OFC cases and the controls. Based on immunohistochemistry reports, three MMPs (MMP-2, MMP-8, and MMP-9) and TIMP-2 had significant correlations with several other biomarkers in OFC cases. CONCLUSIONS MMPs and TIMPs can impact the tissue and cells affected by OFCs and the process of apoptosis. The interaction between some biomarkers with MMPs and TIMPs (e.g., TGFb1) in OFCs can be interesting for future research.
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Affiliation(s)
- Mohammad Moslem Imani
- Department of Orthodontics, Kermanshah University of Medical Sciences, Kermanshah, Iran
| | - Majid Shalchi
- Department of Orthodontics, School of Dentistry, Guilan University of Medical Sciences, Rasht, Iran
| | | | - Masoud Sadeghi
- Medical Biology Research Center, Kermanshah University of Medical Sciences, Kermanshah, Iran.
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11
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Wilkes C, Graetz M, Downie L, Bethune M, Chong D. Prenatal diagnosis of cleft lip and/or palate: What do we tell prospective parents? Prenat Diagn 2023; 43:1310-1319. [PMID: 37552068 DOI: 10.1002/pd.6418] [Citation(s) in RCA: 5] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/30/2023] [Revised: 07/22/2023] [Accepted: 07/24/2023] [Indexed: 08/09/2023]
Abstract
Cleft lip and/or palate (CL/CP) is the most common congenital craniofacial anomaly and parents often ask, "how did this happen?" Patients and families may benefit from access to a multidisciplinary team (MDT) from prenatal diagnosis into early adulthood. Multiple factors can contribute to the development of a cleft. We discuss the epidemiology and risk factors that increase the likelihood of having a newborn with a cleft. The purpose of this article is to review the prenatal investigations involved in the diagnosis and workup of these patients in addition to postpartum treatment, prognostic factors, and counseling families regarding future recurrence risk.
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Affiliation(s)
- Courtney Wilkes
- Department of Plastic and Maxillofacial Surgery, The Royal Children's Hospital, Melbourne, Victoria, Australia
| | - Melissa Graetz
- Department of Genetics, Mercy Hospital for Women, Melbourne, Victoria, Australia
| | - Lilian Downie
- Department of Genetics, Mercy Hospital for Women, Melbourne, Victoria, Australia
| | - Michael Bethune
- Department of Medical Imaging and Perinatal Medicine, The Mercy Hospital for Women, Melbourne, Victoria, Australia
- Specialist Women's Ultrasound, Melbourne, Victoria, Australia
| | - David Chong
- Department of Plastic and Maxillofacial Surgery, The Royal Children's Hospital, Melbourne, Victoria, Australia
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12
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Won HJ, Kim JW, Won HS, Shin JO. Gene Regulatory Networks and Signaling Pathways in Palatogenesis and Cleft Palate: A Comprehensive Review. Cells 2023; 12:1954. [PMID: 37566033 PMCID: PMC10416829 DOI: 10.3390/cells12151954] [Citation(s) in RCA: 9] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/28/2023] [Revised: 07/08/2023] [Accepted: 07/24/2023] [Indexed: 08/12/2023] Open
Abstract
Palatogenesis is a complex and intricate process involving the formation of the palate through various morphogenetic events highly dependent on the surrounding context. These events comprise outgrowth of palatal shelves from embryonic maxillary prominences, their elevation from a vertical to a horizontal position above the tongue, and their subsequent adhesion and fusion at the midline to separate oral and nasal cavities. Disruptions in any of these processes can result in cleft palate, a common congenital abnormality that significantly affects patient's quality of life, despite surgical intervention. Although many genes involved in palatogenesis have been identified through studies on genetically modified mice and human genetics, the precise roles of these genes and their products in signaling networks that regulate palatogenesis remain elusive. Recent investigations have revealed that palatal shelf growth, patterning, adhesion, and fusion are intricately regulated by numerous transcription factors and signaling pathways, including Sonic hedgehog (Shh), bone morphogenetic protein (Bmp), fibroblast growth factor (Fgf), transforming growth factor beta (Tgf-β), Wnt signaling, and others. These studies have also identified a significant number of genes that are essential for palate development. Integrated information from these studies offers novel insights into gene regulatory networks and dynamic cellular processes underlying palatal shelf elevation, contact, and fusion, deepening our understanding of palatogenesis, and facilitating the development of more efficacious treatments for cleft palate.
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Affiliation(s)
- Hyung-Jin Won
- Department of Anatomy, School of Medicine, Kangwon National University, Chuncheon 24341, Republic of Korea
- BIT Medical Convergence Graduate Program, Department of Microbiology and Immunology, School of Medicine, Kangwon National University, Chuncheon 24341, Republic of Korea
| | - Jin-Woo Kim
- Graduate School of Clinical Dentistry, Ewha Womans University, Seoul 03760, Republic of Korea
- Department of Oral and Maxillofacial Surgery, School of Medicine, Ewha Womans University, Seoul 03760, Republic of Korea
| | - Hyung-Sun Won
- Department of Anatomy, Wonkwang University School of Medicine, Iksan 54538, Republic of Korea
- Jesaeng-Euise Clinical Anatomy Center, Wonkwang University School of Medicine, Iksan 54538, Republic of Korea
| | - Jeong-Oh Shin
- Department of Anatomy, College of Medicine, Soonchunhyang University, Cheonan 33151, Republic of Korea
- BK21 FOUR Project, College of Medicine, Soonchunhyang University, Cheonan 33151, Republic of Korea
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Dyląg KA, Anunziata F, Bandoli G, Chambers C. Birth Defects Associated with Prenatal Alcohol Exposure-A Review. CHILDREN (BASEL, SWITZERLAND) 2023; 10:children10050811. [PMID: 37238358 DOI: 10.3390/children10050811] [Citation(s) in RCA: 6] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 04/04/2023] [Revised: 04/20/2023] [Accepted: 04/25/2023] [Indexed: 05/28/2023]
Abstract
Since the recognition of fetal alcohol syndrome, alcohol has been accepted as a human teratogen. However, little is known about the relation between prenatal alcohol exposure and the spectrum of associated major birth defects. The objective of this review was to summarize data on the association of major congenital abnormalities and prenatal alcohol exposure. We included all major birth defects according to ICD-10 classification. We found that the strongest evidence to date lies in the research examining herniation (gastroschisis and omphalocele), oral clefts (cleft lip with or without palate and cleft palate) and cardiac defects. There is less consistent evidence supporting the association between prenatal alcohol exposure and anomalies of gastrointestinal system, diaphragmatic hernia, genitourinary system and neural tube defects. We found no material support for PAE and choanal atresia, biliary atresia or clubfoot.
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Affiliation(s)
- Katarzyna Anna Dyląg
- Department of Pathophysiology, Jagiellonian University Medical College, Czysta 18, 31-121 Krakow, Poland
- St. Louis Children Hospital, ul. Strzelecka 2, 31-503 Krakow, Poland
| | - Florencia Anunziata
- Department of Pediatrics, University of California, San Diego, 9500 Gilman Drive, MC0828, La Jolla, CA 92093-0412, USA
| | - Gretchen Bandoli
- Department of Pediatrics, University of California, San Diego, 9500 Gilman Drive, MC0828, La Jolla, CA 92093-0412, USA
| | - Christina Chambers
- Department of Pediatrics, University of California, San Diego, 9500 Gilman Drive, MC0828, La Jolla, CA 92093-0412, USA
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Sajovic J, Setnikar Lesjak A, Plut A, Eberlinc A, Primožič J, Drevenšek E, Drevenšek M. Maxillary arch dimensions, occlusion assessment and space conditions in patients with cleft palate in the period of deciduous dentition-A retrospective study. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2023; 124:101356. [PMID: 36526111 DOI: 10.1016/j.jormas.2022.101356] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 08/31/2022] [Revised: 11/28/2022] [Accepted: 12/09/2022] [Indexed: 12/15/2022]
Abstract
PURPOSE The aim of this study was to evaluate the maxillary arch dimensions, dentoalveolar relationships and spacing conditions in patients with cleft palate in comparison with the control group. METHODS The subjects consisted of 31 children with cleft palate only (CP) aged 5.5 ± 0.51 (20 with a cleft of hard and soft palate (SHPC group) and 11 with a cleft of soft palate only (SPC group)). In the control group 30 subjects had a normal occlusion at age 5. Maxillary arch dimensions, dentoalveolar relationship according to the Huddard Bodenham index (HBI) and space conditions were compared with the control group. RESULTS In all variables of maxillary arch dimensions except for total arch height the control group exhibited greater values. Most of the significant differences were on account of differences between the control and the SHPC group, with only three comparisons yielding significant results when comparing the two groups of children with clefts (SHPC vs SPC, IV-IV central: pDunnett T3= 0.0002, 95%CIDifference=-9.9-(-3.18); V-V distopalatal cusps: pDunnett T3= 0.0002, 95%CIDifference=-9.97-(-3.17); Total arch length: pDunnett T3= 0.0014, 95%CIDifference=1.74-7.85). The three groups differed significantly in anterior HBI only (HKruskal-Wallis=15.56, p = 0.0067). The spacing conditions in both jaws were also shown to be significantly dependent on the group of subjects studied (Upper jaw: χ2omnibus= 16.79, p = 0.0018; lower jaw: χ2omnibus= 13.75, p = 0.0102). CONCLUSIONS The growth of the upper dental arch at the age of five is impeded in participants with CP in comparison to a control group. It is important to assess the effect of cleft subtypes on growth and development to get a better understanding.
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Affiliation(s)
- Jakob Sajovic
- University Medical Centre Ljubljana, Department of Orthodontics, Hrvatski trg 6, 1000 Ljubljana, Slovenia; University of Ljubljana, Faculty of Medicine, Institute for Pharmacology and Experimental Toxicology, Korytkova ulica 2, 1000 Ljubljana, Slovenia.
| | - Anina Setnikar Lesjak
- University Medical Centre Ljubljana, Department of Orthodontics, Hrvatski trg 6, 1000 Ljubljana, Slovenia
| | - Alja Plut
- University Medical Centre Ljubljana, Department of Orthodontics, Hrvatski trg 6, 1000 Ljubljana, Slovenia
| | - Andreja Eberlinc
- University Medical Centre Ljubljana, Department of Maxillofacial Surgery, Hrvatski trg 6, 1000 Ljubljana, Slovenia
| | - Jasmina Primožič
- University Medical Centre Ljubljana, Department of Orthodontics, Hrvatski trg 6, 1000 Ljubljana, Slovenia; University of Ljubljana, Faculty of Medicine, Department of Jaw and Dental Orthopaedics, Vrazov trg 2, 1000 Ljubljana, Slovenia
| | - Eva Drevenšek
- Hospital for Gynaecology and Obstetrics Kranj, Kidričeva 38a, 4000 Kranj, Slovenia
| | - Martina Drevenšek
- University Medical Centre Ljubljana, Department of Orthodontics, Hrvatski trg 6, 1000 Ljubljana, Slovenia; University of Ljubljana, Faculty of Medicine, Department of Jaw and Dental Orthopaedics, Vrazov trg 2, 1000 Ljubljana, Slovenia
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Albalawi F, Alsaeed S, Alalola B, Alotaib GS, Kalagi S. Prevalence and Patterns of Orofacial Clefts among Children from Different Regions of Saudi Arabia: A Systematic Review. Int J Clin Pediatr Dent 2023; 16:124-130. [PMID: 37020763 PMCID: PMC10068006 DOI: 10.5005/jp-journals-10005-2507] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/07/2023] Open
Abstract
Aim and objective To report on the prevalence and patterns of orofacial clefts (OFC) among Saudi Arabian children by analyzing the results of the studies that have been reported in the literature. Materials and methods A data search was carried out for the articles that had reported on the prevalence of OFC among the Saudi Arabian population in databases like Web of Science, PubMed, Google Scholar, Scopus, and Saudi Digital Library. Articles that were published over the last 15 years were included in this study, following which 13 studies were assessed for qualitative data. Newcastle-Ottawa Quality Assessment Scales for cross-sectional studies were used for analyzing the methodological quality of these studies. Results The prevalence of OFC was within the range of 0.65-1.9/1,000 live births. The highest was witnessed in the Medina region. Parent's consanguinity was the most common risk factor in OFC cases in the included studies. OFC was found to be higher among the male population in comparison with the female. Conclusion The prevalence of OFC in Saudi Arabian children follows the global patterns of OFC. Isolated cleft lip (CL) and cleft palate (CP) are the most common forms of OFC. The prevalence of orofacial anomalies was reported more among children born to parents who had consanguineous marriages. Considering the higher rate of consanguinity among this population, there is an urgent need of developing educational and counseling programs to address the genetic consequences. How to cite this article Albalawi F Alsaeed S, Alalola B, et al. Prevalence and Patterns of Orofacial Clefts among Children from Different Regions of Saudi Arabia: A Systematic Review. Int J Clin Pediatr Dent 2023;16(1):124-130.
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Affiliation(s)
- Farraj Albalawi
- Department of Preventive Dental Science, College of Dentistry, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia; King Abdullah International Medical Research Centre, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Suliman Alsaeed
- Department of Preventive Dental Science, College of Dentistry, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia; King Abdullah International Medical Research Centre, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Bassam Alalola
- Department of Preventive Dental Science, College of Dentistry, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia; King Abdullah International Medical Research Centre, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Ghada Serhan Alotaib
- King Abdullah International Medical Research Centre, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia; Department of Dental Services, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia
| | - Sara Kalagi
- King Abdullah International Medical Research Centre, Ministry of National Guard Health Affairs, Riyadh, Saudi Arabia; Department of Restorative and Prosthetic Dental Sciences, College of Dentistry, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia
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16
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Gulati S, Yadav A, Sharma R. Congenital palatal fistula: Case report and review of literature. JOURNAL OF CLEFT LIP PALATE AND CRANIOFACIAL ANOMALIES 2023. [DOI: 10.4103/jclpca.jclpca_20_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 03/16/2023] Open
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Weill P, Veyssiere A, Rosette J, Vacher C, Benateau H. Embryological approach to philtral hair sparsity in bilateral cleft lip deformities. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2022; 124:101288. [PMID: 36096396 DOI: 10.1016/j.jormas.2022.09.009] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 04/16/2021] [Revised: 06/19/2022] [Accepted: 09/08/2022] [Indexed: 11/16/2022]
Abstract
OBJECTIVE In bilateral cleft lip cases, surgeons have long known that the medial nasal prominence or prolabium is devoid of muscle. Despite cheiloplasty restoring orbicular muscle continuity, moustache hair growth is less developed on the philtral skin. We sought to understand this hair disparity. BASIC PROCEDURES The authors conducted a comprehensive literature search using reference materials on the physiological and pathological development of the face in utero and the PubMed database using the keywords 'embryology', 'bilateral cleft lip' and 'hair'. MAIN RESULTS In bilateral cleft lip, the normal fusion between the maxillary and intermaxillary process does not occur, and the migration of cranial neural crest cells (CNCC) is disturbed. CNCCs from different locations and with different myoblastic differentiation potential are arrested on each side of the cleft. Therefore, a reduced concentration of myoblasts and myocytes as well as hormone receptors in the medial process could hinders the normal physiological development of the different layers of the philtral skin and especially the skin appendages. CONCLUSION Philtral hair sparsity in patients with bilateral cleft lip can be explain by a defect in the migration of CNCCs to the prolabium, whose cells are not able to develop hair follicles, due to a disruption of the exchange of Wnt/β-catenin, EDA, and or Noggin signals and due to the absence of androgen receptor.
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Affiliation(s)
- Pierre Weill
- Department of Maxillo-Facial and Plastic Surgery, Caen University Hospital 14000 Caen France; Medecine Faculty of CAEN, University of Caen Basse-Normandie 14032 Caen Cedex 5, France.
| | - Alexis Veyssiere
- Department of Maxillo-Facial and Plastic Surgery, Caen University Hospital 14000 Caen France; Normandie Univ. Unicaen, Bioconnect, 14000 CAEN, France; Medecine Faculty of CAEN, University of Caen Basse-Normandie 14032 Caen Cedex 5, France
| | - Jeanne Rosette
- Department of Maxillo-Facial and Plastic Surgery, Caen University Hospital 14000 Caen France
| | - Christian Vacher
- Department of Maxillo-Facial surgery, AP-HP, Hospital Beaujon, 100 Boulevard du General Leclerc, Clichy, France
| | - Hervé Benateau
- Department of Maxillo-Facial and Plastic Surgery, Caen University Hospital 14000 Caen France; Normandie Univ. Unicaen, Bioconnect, 14000 CAEN, France; Medecine Faculty of CAEN, University of Caen Basse-Normandie 14032 Caen Cedex 5, France
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18
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Molecular Mechanisms Contributing to the Etiology of Congenital Diaphragmatic Hernia: A Review and Novel Cases. J Pediatr 2022; 246:251-265.e2. [PMID: 35314152 DOI: 10.1016/j.jpeds.2022.03.023] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/04/2021] [Revised: 03/01/2022] [Accepted: 03/15/2022] [Indexed: 12/25/2022]
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Brézulier D, Chaigneau L, Jeanne S, Lebullenger R. The Challenge of 3D Bioprinting of Composite Natural Polymers PLA/Bioglass: Trends and Benefits in Cleft Palate Surgery. Biomedicines 2021; 9:1553. [PMID: 34829782 PMCID: PMC8615666 DOI: 10.3390/biomedicines9111553] [Citation(s) in RCA: 17] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/23/2021] [Revised: 10/15/2021] [Accepted: 10/19/2021] [Indexed: 11/22/2022] Open
Abstract
Cleft lip and palate is the fourth most common congenital malformation. Its prevalence is about 1 in 750 to 1 in 2000 live births. The consequences of this malformation are major: maxillary growth deficit, unaesthetic appearance, phonation disorders, difficulty in eating, and psycho-social disorders. Cleft palate repair establishes the division between the oral and nasal cavities. The alveolar bone graft is a key step. Different sites of autogenous bone harvesting are used, the most common being the iliac crest. Nevertheless, the large number of complications associated with harvesting has led to the use of substitute biomaterials. Bioactive glasses, discovered in 1969, are a group of synthetic silica-based materials with bone-bonding properties. Although 45S5 granular composition is commonly used in bone surgery to repair critical defects, it is only rarely used in the repair of cleft palates because this galenic form is only moderately adapted. However, advances in bone tissue engineering allow the shaping of three-dimensional scaffolds, which support colonization by host cells. Recent advances in computer-aided design/computer-aided manufacturing (CAD/CAM) have even led to the 3D printing of scaffolds combining 45S5 bioglass with a natural and biocompatible poly-lactic acid matrix. The shape of the parts is customized and adapted to the particular shape of the critical bone defects. The objective of this literature review is to highlight the particularities of alveolar defects subsequent to facial clefts, then to detail the characteristics of the materials and technologies used to elaborate 3D matrices by bioprinting. Finally, we will explore research directions regarding their use in reconstructive surgery of cleft palates.
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Affiliation(s)
- Damien Brézulier
- CNRS, University of Rennes, ISCR-UMR 6226, 35000 Rennes, France; (L.C.); (S.J.); (R.L.)
- Pôle Odontologie, CHU Rennes, University of Rennes, 35043 Rennes, France
| | - Louis Chaigneau
- CNRS, University of Rennes, ISCR-UMR 6226, 35000 Rennes, France; (L.C.); (S.J.); (R.L.)
| | - Sylvie Jeanne
- CNRS, University of Rennes, ISCR-UMR 6226, 35000 Rennes, France; (L.C.); (S.J.); (R.L.)
- Pôle Odontologie, CHU Rennes, University of Rennes, 35043 Rennes, France
| | - Ronan Lebullenger
- CNRS, University of Rennes, ISCR-UMR 6226, 35000 Rennes, France; (L.C.); (S.J.); (R.L.)
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Nasreddine G, El Hajj J, Ghassibe-Sabbagh M. Orofacial clefts embryology, classification, epidemiology, and genetics. MUTATION RESEARCH-REVIEWS IN MUTATION RESEARCH 2021; 787:108373. [PMID: 34083042 DOI: 10.1016/j.mrrev.2021.108373] [Citation(s) in RCA: 83] [Impact Index Per Article: 20.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/14/2020] [Revised: 02/21/2021] [Accepted: 02/23/2021] [Indexed: 01/14/2023]
Abstract
Orofacial clefts (OFCs) rank as the second most common congenital birth defect in the United States after Down syndrome and are the most common head and neck congenital malformations. They are classified as cleft lip with or without cleft palate (CL/P) and cleft palate only (CPO). OFCs have significant psychological and socio-economic impact on patients and their families and require a multidisciplinary approach for management and counseling. A complex interaction between genetic and environmental factors contributes to the incidence and clinical presentation of OFCs. In this comprehensive review, the embryology, classification, epidemiology and etiology of clefts are thoroughly discussed and a "state-of-the-art" snapshot of the recent advances in the genetics of OFCs is presented.
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Affiliation(s)
- Ghenwa Nasreddine
- Department of Natural Sciences, School of Arts and Sciences, Lebanese American University, P.O. Box: 13-5053, Chouran, 1102 2801, Beirut, Lebanon.
| | - Joelle El Hajj
- Department of Natural Sciences, School of Arts and Sciences, Lebanese American University, P.O. Box: 13-5053, Chouran, 1102 2801, Beirut, Lebanon.
| | - Michella Ghassibe-Sabbagh
- Department of Natural Sciences, School of Arts and Sciences, Lebanese American University, P.O. Box: 13-5053, Chouran, 1102 2801, Beirut, Lebanon.
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21
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Galeh SD, Nouri-Vaskeh M, Alipour M, Fakhim SA. Clinical and Demographical Characteristics of Cleft Lip and/or Palate in the Northwest of Iran: An Analysis of 1500 Patients. Cleft Palate Craniofac J 2021; 58:1281-1286. [PMID: 33380223 DOI: 10.1177/1055665620980633] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
OBJECTIVE Orofacial clefts (OFCs) can occur as an isolated defect or as a manifestation of other syndromes. The current study aimed to evaluate demographic characteristics and distribution of different types of accompanying anomalies for OFCs in the northwest of Iran. DESIGN A retrospective cohort study. SETTING Tertiary pediatric hospital. PATIENTS AND PARTICIPANTS This study was conducted on 1500 cleft lip and/or palate patients born between July 2010 and June 2020 in the northwest of Iran. MAIN OUTCOME MEASURES Demographic and clinical characteristics of the children with OFCs including familial history, accompanying anomalies and syndromes, maternal passive smoking, mothers' and fathers' age, consanguineous marriage, and birth order. RESULTS Among 1500 patients, 441 had cleft lip, 615 had cleft palate, and 444 had cleft lip and palate. The positive family history of OFCs was found to be 20.9% to 25.4% depending on the cleft type. Accompanying anomalies were identified in 29.8% of cases. Cardiac, facial, and ear abnormalities were the most common types. Also, 2.9% were identified with syndromes and sequences. These included Pierre Robin Sequence, Velo-cardio-facial syndrome, and Down syndrome most frequently. CONCLUSION These findings may provide references for appropriate resources to establish and direct counseling and primary preventive projects in the northwest of Iran.
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Affiliation(s)
- Sima Dabbaghi Galeh
- Department of Otorhinolaryngology, Tabriz University of Medical Sciences, Tabriz, Iran
| | - Masoud Nouri-Vaskeh
- Immunology Research Center, Tabriz University of Medical Sciences, Tabriz, Iran.,Network of Immunity in Infection, Malignancy and Autoimmunity (NIIMA), Universal Scientific Education and Research Network (USERN), Tehran, Iran
| | - Mahdieh Alipour
- Dental and Periodontal Research Center, Tabriz University of Medical Sciences, Tabriz, Iran
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Chaudhari PK, Kharbanda OP, Chaudhry R, Pandey RM, Chauhan S, Bansal K, Sokhi RK. Factors Affecting High Caries Risk in Children With and Without Cleft Lip and/or Palate: A Cross-Sectional Study. Cleft Palate Craniofac J 2020; 58:1150-1159. [PMID: 33349037 DOI: 10.1177/1055665620980206] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022] Open
Abstract
OBJECTIVE The aim of the study was to analyze the caries protective factors, salivary parameters, and microbial counts in high caries risk children with cleft lip and/or palate (CL/P). DESIGN This was a cross-sectional study. SETTING This study was conducted in a tertiary health care teaching hospital in New Delhi, India. PARTICIPANTS The study was conducted in 40 children, 20 with CL/P and 20 without aged between 5 and 12 years. METHODS Children with 2 or more caries lesions in both groups were included in this study. Demographic details, dental caries of affected teeth (World Health Organization criteria for Decayed Missing Filled Teeth [WHO-DMFT] and International Caries Detection and Assessment System [ICDAS II]), caries protective factors, salivary parameters, and microbial counts were recorded by one calibrated investigator. MAIN OUTCOME MEASURES Caries protective factors, salivary parameters, and microbial profile. RESULTS The Chi-square (χ2) test and Pearson correlation were used for statistical analysis. All the children participating in the study brushed their teeth only once in a day and consumed sweets more than twice a day. None of the children had ever received fluoride varnish. Resting saliva had a low buffering capacity in 80% of children with CL/P and 95% of children without CL/P. Microbial assessment of stimulated saliva showed that with the increases in the numbers (DMFT scores ≥4) and severity (ICDAS codes from 1-2 to 5-6) of caries lesions, both Streptococci and Lactobacilli counts were ≥105 colony-forming units/mL of saliva in the both groups. CONCLUSIONS Children with CL/P showed limited access to caries protective measures and low buffering capacity in resting saliva, along with elevated levels of salivary Streptococci and Lactobacilli in stimulated saliva.
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Affiliation(s)
- Prabhat Kumar Chaudhari
- Division of Orthodontics and Dentofacial Deformities, Centre for Dental Education and Research, All India Institute of Medical Sciences, New Delhi, India
| | - Om P Kharbanda
- Dr CG Pandit National Chair of ICMR, Department of Plastic Reconstructive & Burns Surgery, All India Institute of Medical Sciences, New Delhi, India
| | - Rama Chaudhry
- Department of Microbiology, All India Institute of Medical Sciences, New Delhi, India
| | - Ravindra Mohan Pandey
- Department of Biostatistics, All India Institute of Medical Sciences, New Delhi, India
| | - Shashank Chauhan
- Department of Plastic Reconstructive & Burns Surgery, All India Institute of Medical Sciences, New Delhi, India
| | - Kalpana Bansal
- Division of Pedodontics and Preventive Dentistry, Centre for Dental Education and Research, All India Institute of Medical Sciences, New Delhi, India
| | - Ramandeep Kaur Sokhi
- Division of Orthodontics and Dentofacial Deformities, Centre for Dental Education and Research, All India Institute of Medical Sciences, New Delhi, India
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Martinelli M, Palmieri A, Carinci F, Scapoli L. Non-syndromic Cleft Palate: An Overview on Human Genetic and Environmental Risk Factors. Front Cell Dev Biol 2020; 8:592271. [PMID: 33195260 PMCID: PMC7606870 DOI: 10.3389/fcell.2020.592271] [Citation(s) in RCA: 47] [Impact Index Per Article: 9.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/06/2020] [Accepted: 09/28/2020] [Indexed: 12/27/2022] Open
Abstract
The epithelial and mesenchymal cells involved in early embryonic facial development are guided by complex regulatory mechanisms. Any factor perturbing the growth, approach and fusion of the frontonasal and maxillary processes could result in orofacial clefts that represent the most common craniofacial malformations in humans. The rarest and, probably for this reason, the least studied form of cleft involves only the secondary palate, which is posterior to the incisive foramen. The etiology of cleft palate only is multifactorial and involves both genetic and environmental risk factors. The intention of this review is to give the reader an overview of the efforts made by researchers to shed light on the underlying causes of this birth defect. Most of the scientific papers suggesting potential environmental and genetic causes of non-syndromic cleft palate are summarized in this review, including genome-wide association and gene–environment interaction studies.
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Affiliation(s)
- Marcella Martinelli
- Department of Experimental, Diagnostic and Specialty Medicine, Alma Mater Studiorum - University of Bologna, Bologna, Italy
| | - Annalisa Palmieri
- Department of Experimental, Diagnostic and Specialty Medicine, Alma Mater Studiorum - University of Bologna, Bologna, Italy
| | - Francesco Carinci
- Department of Morphology, Surgery and Experimental Medicine, University of Ferrara, Ferrara, Italy
| | - Luca Scapoli
- Department of Experimental, Diagnostic and Specialty Medicine, Alma Mater Studiorum - University of Bologna, Bologna, Italy
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Craniofacial malformations and their association with brain development: the importance of a multidisciplinary approach for treatment. Odontology 2019; 108:1-15. [PMID: 31172336 DOI: 10.1007/s10266-019-00433-7] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/30/2018] [Accepted: 05/22/2019] [Indexed: 02/08/2023]
Abstract
The craniofacial complex develops mainly in the first trimester of pregnancy, but its final shaping and the development of the teeth extend into the second and third trimesters. It is intimately connected with the development of the brain because of the crucial role the cranial neural crest cells play and the fact that many signals which control craniofacial development originate in the brain and vice versa. As a result, malformations of one organ may affect the development of the other. Similarly, there are developmental connections between the craniofacial complex and the teeth. Craniofacial anomalies are either isolated, resulting from abnormal development of the first two embryonic pharyngeal arches, or part of multiple malformation syndromes affecting many other organs. They may stem from gene mutations, chromosomal aberrations or from environmental causes induced by teratogens. The craniofacial morphologic changes are generally cosmetic, but they often interfere with important functions such as chewing, swallowing and respiration. In addition, they may cause hearing or visual impairment. In this review we discussed only a small number of craniofacial malformations and barely touched upon related anomalies of dentition. Following a brief description of the craniofacial development, we discussed oral clefts, craniofacial microsomia, teratogens that may interfere with craniofacial development resulting in different malformations, the genetically determined craniosynostoses syndromes and few other relatively common syndromes that, in addition to the craniofacial complex, also affect other organs. The understanding of these malformations is important in dentistry as dentists play an integral role in their diagnosis and multidisciplinary treatment.
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Mandal E, Filip C, Andersson MEM, Øgaard B. Eighteen-Year Follow-Up of 160 Consecutive Individuals Born With Unilateral Cleft Lip or Cleft Lip and Alveolus Treated by the Oslo Cleft Lip and Palate Team. Cleft Palate Craniofac J 2019; 56:853-859. [PMID: 30686058 DOI: 10.1177/1055665618820753] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
OBJECTIVE Describe patients born with unilateral cleft lip with or without cleft alveolus (CL±A) in relation to cleft severity and laterality, gender, associated anomalies and syndromes, number and type of lip- and nose operations, and time of alveolar bone graft (ABG) treatment in relation to dental status in cleft area. MATERIALS AND METHODS Patients included 220 children born with unilateral CL±A, born between 1988 and 1997 referred to the Oslo Cleft Lip and Palate Team. The data were collected retrospectively. All patients were followed up until 18 years of age. RESULTS Among all CL±A, 3.6% had recognized syndromes, 6.8% had associated anomalies, and in 89.6% CL±A was the only malformation. CL±A was more common, but not more severe, on the left side. Among the 160 individuals with CL±A without syndromes and associated anomalies, 66.9% had an isolated soft tissue CL, and 33.1% were diagnosed with a CL alveolus (CL+A). Male predominance was observed. Children with CL+A had more severe soft tissue clefts of the lip and underwent more lip and nose surgeries than children born with CL. The time of ABG was found to be at a younger age when the patient had a lateral incisor in the cleft area than when this tooth was missing. CONCLUSION Findings provide a reference for morphologic variations in CL±A, and insight into the surgical burden of care until the age of 18 years.
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Affiliation(s)
- Emeline Mandal
- 1 Department of Orthodontics, Faculty of Dentistry, University of Oslo, Norway
| | - Charles Filip
- 2 Department of Plastic and Reconstructive Surgery, Oslo University Hospital, Rikshospitalet, Oslo, Norway
| | | | - Bjørn Øgaard
- 1 Department of Orthodontics, Faculty of Dentistry, University of Oslo, Norway
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Smarius B, Loozen C, Manten W, Bekker M, Pistorius L, Breugem C. Accurate diagnosis of prenatal cleft lip/palate by understanding the embryology. World J Methodol 2017; 7:93-100. [PMID: 29026689 PMCID: PMC5618146 DOI: 10.5662/wjm.v7.i3.93] [Citation(s) in RCA: 42] [Impact Index Per Article: 5.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/15/2016] [Revised: 05/09/2017] [Accepted: 05/31/2017] [Indexed: 02/06/2023] Open
Abstract
Cleft lip with or without cleft palate (CP) is one of the most common congenital malformations. Ultrasonographers involved in the routine 20-wk ultrasound screening could encounter these malformations. The face and palate develop in a very characteristic way. For ultrasonographers involved in screening these patients it is crucial to have a thorough understanding of the embryology of the face. This could help them to make a more accurate diagnosis and save time during the ultrasound. Subsequently, the current postnatal classification will be discussed to facilitate the communication with the CP teams.
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Affiliation(s)
- Bram Smarius
- Division of Pediatric Plastic Surgery, Cleft Palate Team, Wilhelmina Children’s Hospital, 3584 EA Utrecht, The Netherlands
| | - Charlotte Loozen
- Division of Pediatric Plastic Surgery, Cleft Palate Team, Wilhelmina Children’s Hospital, 3584 EA Utrecht, The Netherlands
| | - Wendy Manten
- Division of Gynecology and Obstetrics, Wilhelmina Children’s Hospital, 3584 EA Utrecht, The Netherlands
| | - Mireille Bekker
- Division of Gynecology and Obstetrics, Wilhelmina Children’s Hospital, 3584 EA Utrecht, The Netherlands
| | - Lou Pistorius
- Division of Gynecology and Obstetrics, Stellenbosch University and Tygerberg Hospital, Cape Town 7500, South Africa
| | - Corstiaan Breugem
- Division of Pediatric Plastic Surgery, Cleft Palate Team, Wilhelmina Children’s Hospital, 3584 EA Utrecht, The Netherlands
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Zhou Y, Gilboa SM, Herdt ML, Lupo PJ, Flanders WD, Liu Y, Shin M, Canfield MA, Kirby RS. Maternal exposure to ozone and PM 2.5 and the prevalence of orofacial clefts in four U.S. states. ENVIRONMENTAL RESEARCH 2017; 153:35-40. [PMID: 27888746 PMCID: PMC5612445 DOI: 10.1016/j.envres.2016.11.007] [Citation(s) in RCA: 41] [Impact Index Per Article: 5.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/21/2016] [Revised: 11/09/2016] [Accepted: 11/13/2016] [Indexed: 05/05/2023]
Abstract
BACKGROUND While there is some evidence that maternal exposure to ambient air pollution is associated with orofacial clefts in offspring, the epidemiologic studies have been largely equivocal. We evaluated whether maternal exposure to elevated county-level ambient fine particulate matter with aerodynamic diameter ≤2.5µm (PM2.5) and ozone during early gestation was associated with a higher prevalence of orofacial clefts. METHODS Birth data consisting of 4.7 million births from 2001 to 2007 were obtained from National Birth Defects Prevention Network for four states - Arizona, Florida, New York (excluding New York City), and Texas. The air pollution exposure assessment for gestational weeks 5-10 was based on county-level average concentrations of PM2.5 and ozone data generated using a Bayesian fusion model available through CDC's Environmental Public Health Tracking Network. Two outcomes were analyzed separately: cleft lip with or without cleft palate, cleft palate alone. In logistic regression analyses, we adjusted for factors that were suspected confounders or modifiers of the association between the prevalence of orofacial clefts and air pollution, i.e., infant sex, race-ethnicity, maternal education, smoking status during pregnancy, whether this was mother's first baby, maternal age. RESULTS Each 10µg/m3 increase in PM2.5 concentration was significantly associated with cleft palate alone (OR =1.43, 95% CI: 1.11-1.86). There was no significant association between PM2.5 concentration and cleft lip with or without cleft palate. No associations were observed between ozone exposure and the two outcomes of orofacial clefts. CONCLUSIONS Our study suggests that PM2.5 significantly increased the risk of cleft palate alone, but did not change the incidence of cleft lip with or without palate. Ozone levels did not correlate with incidence of orofacial clefts.
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Affiliation(s)
- Ying Zhou
- Environmental Health Tracking Branch, Division of Environmental Hazards and Health Effects, National Center for Environmental Health, Centers for Disease Control and Prevention, Atlanta, GA, USA.
| | - Suzanne M Gilboa
- Birth Defects Branch, Division of Congenital and Developmental Disorders, National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, GA, USA
| | - Michele L Herdt
- New York State Department of Health, Center for Environmental Health, Albany, NY, USA; State University of New York at Albany, Department of Epidemiology and Biostatistics, Rensselaer, NY, USA
| | - Philip J Lupo
- Baylor College of Medicine, Department of Pediatrics, Section of Hematology-Oncology, Houston, TX, USA
| | - W Dana Flanders
- Department of Epidemiology, Rollins School of Public Health, Emory University, Atlanta, GA, USA
| | - Yang Liu
- Department of Environmental Health, Rollins School of Public Health, Emory University, Atlanta, GA, USA
| | - Mikyong Shin
- Environmental Health Tracking Branch, Division of Environmental Hazards and Health Effects, National Center for Environmental Health, Centers for Disease Control and Prevention, Atlanta, GA, USA
| | - Mark A Canfield
- Birth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, TX, USA
| | - Russell S Kirby
- Department of Community and Family Health, College of Public Health, University of South Florida, Tampa, FL, USA
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Tanner JP, Salemi JL, Stuart AL, Yu H, Jordan MM, DuClos C, Cavicchia P, Correia JA, Watkins SM, Kirby RS. Uncertainty in maternal exposures to ambient PM2.5 and benzene during pregnancy: Sensitivity to exposure estimation decisions. Spat Spatiotemporal Epidemiol 2016; 17:117-29. [PMID: 27246278 DOI: 10.1016/j.sste.2016.04.009] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/15/2015] [Revised: 04/12/2016] [Accepted: 04/27/2016] [Indexed: 01/17/2023]
Abstract
We investigate uncertainty in estimates of pregnant women's exposure to ambient PM2.5 and benzene derived from central-site monitoring data. Through a study of live births in Florida during 2000-2009, we discuss the selection of spatial and temporal scales of analysis, limiting distances, and aggregation method. We estimate exposure concentrations and classify exposure for a range of alternatives, and compare impacts. Estimated exposure concentrations were most sensitive to the temporal scale of analysis for PM2.5, with similar sensitivity to spatial scale for benzene. Using 1-12 versus 3-8 weeks of gestational age as the exposure window resulted in reclassification of exposure by at least one quartile for up to 37% of mothers for PM2.5 and 27% for benzene. The largest mean absolute differences in concentration resulting from any decision were 0.78 µg/m(3) and 0.44 ppbC, respectively. No bias toward systematically higher or lower estimates was found between choices for any decision.
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Affiliation(s)
- Jean Paul Tanner
- Birth Defects Surveillance Program, Department of Community and Family Health, University of South Florida, 13201 Bruce B. Downs Blvd. MDC 56, Tampa, FL 33612, USA.
| | - Jason L Salemi
- Birth Defects Surveillance Program, Department of Community and Family Health, University of South Florida, 13201 Bruce B. Downs Blvd. MDC 56, Tampa, FL 33612, USA; Department of Family and Community Medicine, Baylor College of Medicine, 3701 Kirby Dr. Suite 600, Houston, TX 77098, USA.
| | - Amy L Stuart
- Department of Environmental and Occupational Health, University of South Florida, 13201 Bruce B. Downs Blvd. MDC 56, Tampa, FL 33612, USA; Department of Civil and Environmental Engineering, University of South Florida, 4202 E. Fowler Avenue, Tampa, FL 33620, USA.
| | - Haofei Yu
- Department of Environmental and Occupational Health, University of South Florida, 13201 Bruce B. Downs Blvd. MDC 56, Tampa, FL 33612, USA.
| | - Melissa M Jordan
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, 4052 Bald Cypress Way, Tallahassee, FL 32399 USA.
| | - Chris DuClos
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, 4052 Bald Cypress Way, Tallahassee, FL 32399 USA.
| | - Philip Cavicchia
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, 4052 Bald Cypress Way, Tallahassee, FL 32399 USA.
| | - Jane A Correia
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, 4052 Bald Cypress Way, Tallahassee, FL 32399 USA.
| | - Sharon M Watkins
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, 4052 Bald Cypress Way, Tallahassee, FL 32399 USA.
| | - Russell S Kirby
- Birth Defects Surveillance Program, Department of Community and Family Health, University of South Florida, 13201 Bruce B. Downs Blvd. MDC 56, Tampa, FL 33612, USA.
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Abramson ZR, Peacock ZS, Cohen HL, Choudhri AF. Radiology of Cleft Lip and Palate: Imaging for the Prenatal Period and throughout Life. Radiographics 2015; 35:2053-63. [DOI: 10.1148/rg.2015150050] [Citation(s) in RCA: 18] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/31/2022]
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Tanner JP, Salemi JL, Stuart AL, Yu H, Jordan MM, DuClos C, Cavicchia P, Correia JA, Watkins SM, Kirby RS. Associations between exposure to ambient benzene and PM(2.5) during pregnancy and the risk of selected birth defects in offspring. ENVIRONMENTAL RESEARCH 2015; 142:345-353. [PMID: 26196779 DOI: 10.1016/j.envres.2015.07.006] [Citation(s) in RCA: 72] [Impact Index Per Article: 7.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 02/04/2015] [Revised: 07/06/2015] [Accepted: 07/07/2015] [Indexed: 05/21/2023]
Abstract
OBJECTIVE A growing number of studies have investigated the association between air pollution and the risk of birth defects, but results are inconsistent. The objective of this study was to examine whether maternal exposure to ambient PM2.5 or benzene increases the risk of selected birth defects in Florida. METHODS We conducted a retrospective cohort study of singleton infants born in Florida from 2000 to 2009. Isolated and non-isolated birth defect cases of critical congenital heart defects, orofacial clefts, and spina bifida were identified from the Florida Birth Defects Registry. Estimates of maternal exposures to PM2.5 and benzene for all case and non-case pregnancies were derived by aggregation of ambient measurement data, obtained from the US Environmental Protection Agency Air Quality System, during etiologically relevant time windows. Multivariable Poisson regression was used to estimate adjusted prevalence ratios (aPRs) and 95% confidence intervals (CIs) for each quartile of air pollutant exposure. RESULTS Compared to the first quartile of PM2.5 exposure, higher levels of exposure were associated with an increased risk of non-isolated truncus arteriosus (aPR4th Quartile, 8.80; 95% CI, 1.11-69.50), total anomalous pulmonary venous return (aPR2nd Quartile, 5.00; 95% CI, 1.10-22.84), coarctation of the aorta (aPR4th Quartile, 1.72; 95% CI, 1.15-2.57; aPR3rd Quartile, 1.60; 95% CI, 1.07-2.41), interrupted aortic arch (aPR4th Quartile, 5.50; 95% CI, 1.22-24.82), and isolated and non-isolated any critical congenital heart defect (aPR3rd Quartile, 1.13; 95% CI, 1.02-1.25; aPR4th Quartile, 1.33; 95% CI, 1.07-1.65). Mothers with the highest level of exposure to benzene were more likely to deliver an infant with an isolated cleft palate (aPR4th Quartile, 1.52; 95% CI, 1.13-2.04) or any orofacial cleft (aPR4th Quartile, 1.29; 95% CI, 1.08-1.56). An inverse association was observed between exposure to benzene and non-isolated pulmonary atresia (aPR4th Quartile, 0.19; 95% CI, 0.04-0.84). CONCLUSION Our results suggest a few associations between exposure to ambient PM2.5 or benzene and specific birth defects in Florida. However, many related comparisons showed no association. Hence, it remains unclear whether associations are clinically significant or can be causally related to air pollution exposures.
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Affiliation(s)
- Jean Paul Tanner
- Birth Defects Surveillance Program, Department of Community and Family Health, College of Public Health, University of South Florida, Tampa, FL, USA.
| | - Jason L Salemi
- Birth Defects Surveillance Program, Department of Community and Family Health, College of Public Health, University of South Florida, Tampa, FL, USA; Department of Family and Community Medicine, Baylor College of Medicine, Houston, TX, USA.
| | - Amy L Stuart
- Department of Environmental and Occupational Health, College of Public Health, University of South Florida, Tampa, FL, USA; Department of Civil and Environmental Engineering, College of Engineering, University of South Florida, Tampa, FL, USA.
| | - Haofei Yu
- Department of Environmental and Occupational Health, College of Public Health, University of South Florida, Tampa, FL, USA.
| | - Melissa M Jordan
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, Tallahassee, FL, USA.
| | - Chris DuClos
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, Tallahassee, FL, USA.
| | - Philip Cavicchia
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, Tallahassee, FL, USA.
| | - Jane A Correia
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, Tallahassee, FL, USA.
| | - Sharon M Watkins
- Bureau of Epidemiology, Division of Disease Control and Health Protection, Florida Department of Health, Tallahassee, FL, USA.
| | - Russell S Kirby
- Birth Defects Surveillance Program, Department of Community and Family Health, College of Public Health, University of South Florida, Tampa, FL, USA.
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Épidémiologie des malformations associées aux fentes labiales et palatines à propos d’une étude rétrospective de 324 cas. Arch Pediatr 2015; 22:816-21. [DOI: 10.1016/j.arcped.2015.05.005] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2014] [Revised: 04/05/2015] [Accepted: 05/20/2015] [Indexed: 11/21/2022]
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Shkoukani MA, Lawrence LA, Liebertz DJ, Svider PF. Cleft palate: A clinical review. ACTA ACUST UNITED AC 2014; 102:333-42. [DOI: 10.1002/bdrc.21083] [Citation(s) in RCA: 27] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/17/2014] [Accepted: 10/27/2014] [Indexed: 01/30/2023]
Affiliation(s)
- Mahdi A. Shkoukani
- Department of Otolaryngology-Head and Neck Surgery; Wayne State University School of Medicine; Detroit Michigan
- Department of Otolaryngology-Head and Neck Surgery; Division of Craniofacial Surgery, Wayne State University School of Medicine; Detroit Michigan
- Division of Facial Plastic and Reconstructive Surgery; Wayne State University School of Medicine; Detroit Michigan
| | - Lauren A. Lawrence
- Department of Otolaryngology-Head and Neck Surgery; Wayne State University School of Medicine; Detroit Michigan
| | - Daniel J. Liebertz
- Department of Otolaryngology-Head and Neck Surgery; Wayne State University School of Medicine; Detroit Michigan
| | - Peter F. Svider
- Department of Otolaryngology-Head and Neck Surgery; Wayne State University School of Medicine; Detroit Michigan
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Lei RL, Chen HS, Huang BY, Chen YC, Chen PKT, Lee HY, Chang CW, Wu CL. Population-based study of birth prevalence and factors associated with cleft lip and/or palate in Taiwan 2002-2009. PLoS One 2013; 8:e58690. [PMID: 23555592 PMCID: PMC3608649 DOI: 10.1371/journal.pone.0058690] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/24/2012] [Accepted: 02/06/2013] [Indexed: 12/04/2022] Open
Abstract
Background Facial cleft deformities, including cleft lip with or without cleft palate (CL/P) and cleft palate (CP), are common congenital birth anomalies, especially in Asia. This study aimed to analyze the prevalence of CL/P and CP and to identify associated factors in Taiwan. Methods This population-based epidemiological study retrospectively analyzed birth data obtained from the Department of Health in Taiwan for years 2002–2009. Frequency distribution, percentages and related predictors were investigated, and findings were presented by types of cleft deformities. Logistic regression analysis was performed to identify factors associated with cleft deformities. Results Overall prevalence of cleft deformities among 1,705,192 births was 0.1% for CL/P and 0.04% for CP over the 8-year study period. Higher prevalence of CL/P or CP was observed with multiple pregnancies, being male for CL/P, being female for CP, gestational age ≤37 weeks and lower birth weight (<1.5 kg). Both CL/P and CP were significantly associated with gestational age <37 weeks and birth weight<1.5 kg (all P <0.0001). CL/P was significantly associated with multiple parities (P = 0.0004–0.002). Male newborns and female newborns were significantly associated with CL/P and CP, respectively (both P<0.0001). Conclusions Overall prevalence for congenital cleft deformities in study subjects was 0.1%, in keeping with high rates in Asia. Results suggest the need for awareness and early identification of those at high risk for cleft deformities, including newborns with gestational age <37 weeks, weighing <1.5 kg at birth and women with multiple parities, as a potential strategy to counter long-term adverse effects on speech and language in this population.
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Affiliation(s)
- Ruoh-Lih Lei
- Department of Nursing, College of Medicine and Nursing, HungKuang University, Taiwan, Republic of China.
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Billington CJ, Schmidt B, Zhang L, Hodges JS, Georgieff MK, Schotta G, Gopalakrishnan R, Petryk A. Maternal diet supplementation with methyl donors and increased parity affect the incidence of craniofacial defects in the offspring of twisted gastrulation mutant mice. J Nutr 2013; 143:332-9. [PMID: 23343680 PMCID: PMC3713022 DOI: 10.3945/jn.112.168906] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022] Open
Abstract
Diets rich in methyl-donating compounds, including folate, can provide protection against neural tube defects, but their role in preventing craniofacial defects is less clear. Mice deficient in Twisted gastrulation (TWSG1), an extracellular modulator of bone morphogenetic protein signaling, manifest both midline facial defects and jaw defects, allowing study of the effects of methyl donors on various craniofacial defects in an experimentally tractable animal model. The goal of this study was to examine the effects of maternal dietary supplementation with methyl donors on the incidence and type of craniofacial defects among Twsg1(-/-) offspring. Nulliparous and primiparous female mice were fed an NIH31 standard diet (control) or a methyl donor supplemented (MDS) diet (folate, vitamin B-12, betaine, and choline). Observed defects in the pups were divided into those derived mostly from the first branchial arch (BA1) (micrognathia, agnathia, cleft palate) and midline facial defects in the holoprosencephaly spectrum (cyclopia, proboscis, and anterior truncation). In the first pregnancy, offspring of mice fed the MDS diet had lower incidence of BA1-derived defects (12.8% in MDS vs. 32.5% in control; P = 0.02) but similar incidence of midline facial defects (6.4% in MDS vs. 5.2% in control; P = 1.0). Increased maternal parity was independently associated with increased incidence of craniofacial defects after adjusting for diet (from 37.7 to 59.5% in control, P = 0.04 and from 19.1 to 45.3% in MDS, P = 0.045). In conclusion, methyl donor supplementation shows protective effects against jaw defects, but not midline facial defects, and increased parity can be a risk factor for some craniofacial defects.
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Affiliation(s)
| | | | - Lei Zhang
- Biostatistical Design and Analysis Center
| | | | | | - Gunnar Schotta
- Adolf-Butenandt-Institute, Ludwig-Maximilian-University, Munich, Germany; and
| | - Rajaram Gopalakrishnan
- Diagnostic/Biological Sciences, School of Dentistry, University of Minnesota, Minneapolis, MN
| | - Anna Petryk
- Department of Pediatrics,,Department of Genetics, Cell Biology and Development,,To whom correspondence should be addressed. E-mail:
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Abstract
Abnormalities of the fetal head and neck may be seen in isolation or in association with central nervous system abnormalities, chromosomal abnormalities, and syndromes. Magnetic resonance imaging (MRI) plays an important role in detecting associated abnormalities of the brain as well as in evaluating for airway obstruction that may impact prenatal management and delivery planning. This article provides an overview of the common indications for MRI of the fetal head and neck, including abnormalities of the fetal skull and face, masses of the face and neck, and fetal goiter.
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Affiliation(s)
- David M Mirsky
- Division of Neuroradiology, Department of Radiology, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, 324 South 34th Street, Philadelphia, PA 19104, USA.
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37
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Goenjian HA, Chiu ES, Alexander ME, Hilaire HS, Moses M. Incidence of Cleft Pathology in Greater New Orleans before and after Hurricane Katrina. Cleft Palate Craniofac J 2011; 48:757-61. [DOI: 10.1597/09-246] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/22/2022] Open
Abstract
Background Reports after the 2005 Hurricane Katrina have documented an increase in stress reactions and environmental teratogens (arsenic, mold, alcohol). Objective To assess the incidence of cleft pathology before and after the hurricane, and the distribution of cleft cases by gender and race. Methods Retrospective chart review of cleft lip with or without cleft palate (CUP) and cleft palate (CP) cases registered with the Cleft and Craniofacial Team at Children's Hospital of New Orleans, the surgical center that treated cleft cases in Greater New Orleans between 2004 and 2007. Live birth data were obtained from the Louisiana State Center for Health Statistics. Results The incidence of cleft cases, beginning 9 months after the hurricane (i.e., June 1, 2006) was significantly higher compared with the period before the hurricane (0.80 versus 1.42; p = .008). Within racial group comparisons showed a higher incidence among African Americans versus whites (0.42 versus 1.22; p = .01). The distribution of CUP and CP cases by gender was significant ( p = .05). Conclusion The increase in the incidence of cleft cases after the hurricane may be attributable to increased stress and teratogenic factors associated with the hurricane. The increase among African Americans may have been due to comparatively higher exposure to environmental risk factors. These findings warrant further investigation to replicate the results elsewhere in the Gulf to determine whether there is a causal relationship between environmental risk factors and increased cleft pathology.
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Affiliation(s)
- Haig A. Goenjian
- Division of Plastic Surgery Research Team, Tulane University Health Sciences Center, New Orleans, Louisiana
| | - Ernest S. Chiu
- Plastic and Reconstructive Research, Tulane University Health Sciences Center, New Orleans, Louisiana
| | | | - Hugo St. Hilaire
- Children's Hospital of New Orleans, and Louisiana State University Health Sciences Center, New Orleans, Louisiana
| | - Michael Moses
- Children's Hospital of New Orleans, New Orleans, Louisiana
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Pilon AF. Midline orofacial cleft defects in association with type 1 Duane's retraction syndrome. Clin Exp Optom 2008; 92:133-6. [PMID: 18691219 DOI: 10.1111/j.1444-0938.2008.00311.x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022] Open
Abstract
Anomalies of ocular motility associated with Duane's retraction syndrome (DRS) have often been perceived as isolated phenomena arising as a result of congenital ocular miswiring between the lateral and medial recti muscles. Interestingly, the reporting of concomitant ocular and systemic anomalies arising in the setting of DRS discounts this narrow perception and highlights the importance of a thorough investigation for non-ocular comorbidities. A 37-year-old Caucasian male presented for a routine ocular examination with complaints of gradual reduction in uncorrected near vision. Our testing confirmed the characteristic deficit of abduction, palpebral fissure narrowing and globe retraction consistent with a type 1 Duane's defect of the right eye. A midline cleft lip and palate were also noted in the absence of signs of nuchal rigidity/reduced range of motion in the cervical spine, spinal misalignment, deafness, optic nerve anomalies, hypertelorism or altered mental state. Awareness of key comorbidities and the importance of thorough clinical evaluation with appropriate supplemental testing and consultation with a paediatric ophthalmologist are warranted in non-isolated presentations of DRS.
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Affiliation(s)
- Andrew F Pilon
- Southern California College of Optometry, Fullerton, California 92831, USA.
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39
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Abstract
PURPOSE OF REVIEW Management of bilateral cleft lip and nasal deformity can be a challenging task. This paper provides an overview of bilateral cleft lip and nasal deformity with an updated review of current management issues in the literature. RECENT FINDINGS The Centers for Disease Control and Prevention recently reported that orofacial clefts are now the most common birth defect. While this statistic may be disheartening, the increased prevalence brings the problem to light at the forefront of the medical community, thus gaining more support and resources. Many techniques have been described for repair of bilateral cleft lip and nasal deformity. A recent advancement in presurgical orthopedics is the use of nasoalveolar molding to narrow wide clefts. SUMMARY Surgical management of bilateral cleft lip and nasal deformity poses a challenge to the skill and judgment of the cleft surgeon. Although techniques continue to evolve over the decades, the basic principles of cleft surgery remain the same. The main principles are to achieve an appropriate philtral size and shape, to position the cartilages in a more optimal position, and to attain muscular continuity and symmetry for optimal appearance and function. Thus, while keeping the basic principles in mind, management of bilateral cleft lip and nasal deformity becomes a valuable and rewarding experience for the surgeon, patient and caregiver.
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Affiliation(s)
- Annette M Pham
- Department of Otolaryngology, University of California, Davis School of Medicine, Sacramento, 95817, USA
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Dudas M, Li WY, Kim J, Yang A, Kaartinen V. Palatal fusion - where do the midline cells go? A review on cleft palate, a major human birth defect. Acta Histochem 2007; 109:1-14. [PMID: 16962647 DOI: 10.1016/j.acthis.2006.05.009] [Citation(s) in RCA: 73] [Impact Index Per Article: 4.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2006] [Revised: 05/26/2006] [Accepted: 05/31/2006] [Indexed: 01/14/2023]
Abstract
Formation of the palate, the organ that separates the oral cavity from the nasal cavity, is a developmental process characteristic to embryos of higher vertebrates. Failure in this process results in palatal cleft. During the final steps of palatogenesis, two palatal shelves outgrowing from the sides of the embryonic oronasal cavity elevate above the tongue, meet in the midline, and rapidly fuse together. Over the decades, multiple mechanisms have been proposed to explain how the superficial mucous membranes disappear from the contact line, thus allowing for normal midline mesenchymal confluence. A substantial body of experimental evidence exists for cell death, cell migration, epithelial-to-mesenchymal transdifferentiation (EMT), replacement through new tissue intercalation, and other mechanisms. However, the most recent use of gene recombination techniques in cell fate tracking disfavors the EMT concept, and suggests that apoptosis is the major fate of the midline cells during physiological palatal fusion. This article summarizes the benefits and drawbacks of histochemical and molecular tools used to determine the fates of cells within the palatal midline. Mechanisms of normal disintegration of the midline epithelial seam are reviewed together with pathologic processes that prevent this disintegration, thus causing cleft palate.
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Affiliation(s)
- Marek Dudas
- Developmental Biology Program, The Saban Research Institute of Childrens Hospital Los Angeles, Mail Stop 35, 4650 Sunset Blvd., Los Angeles, CA 90027, USA.
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41
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Abstract
Cleft lip and cleft palate (CL/CP) are the most commonly occurring craniofacial birth defects. Although some CL/CPs are detected on prenatal ultrasound, the majority are immediately recognized in the delivery room. Part 1 of this 2-part article, "Understanding the Embryology and Genetics of Cleft Lip and Palate," presented the embryology of the face, lip, and palate to help the clinician understand the timing, complexity, and factors that may influence the development of these defects. Part 2 provides clinicians with the tools needed to obtain a detailed family and pregnancy history to evaluate for known associated risk factors. It provides a guide for a systematic physical assessment of the infant with CL/CP along with key areas of assessment for other midline defects or physical findings consistent with associated syndromes. Pictures of a variety of types of CL/CP are included to enhance understanding of these defects. Treatment and long-term complications of CL/CP are reviewed with an emphasis on family support, identifying educational resources, and counseling.
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Affiliation(s)
- Linda Merritt
- Neonatal Intensive Care Unit, Medical City Children's Hospital, Dallas, TX 75220, USA
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