Aspects of Hemifacial Microsomia

Hemifaciale microsomia (HFM) is een aangeboren asymmetrische afwijking van het gelaat met grote variatie. Het ontstaan is onduidelijk. Buiten de craniofaciale (aangezichts) afwijkingen, komen ook andere defecten voor. Het klinisch beeld toont een enkelzijdige afwijking van het oor en een onderontwikkeling van het skelet, de weke delen en de aangezichtsspieren aan de aangedane gezichtshelft. Behandeling moet leiden tot een verbeterde functie en optimale gelaatssymmetrie aan het einde van de groei. Het uiteindelijke behandelresultaat wordt beïnvloed door externe factoren, zoals de chirurgische techniek, maar ook door intrinsieke patiënt factoren zoals genetische achtergrond, groei en ontwikkeling. Het eindresultaat is afhankelijk van het psychosociale welzijn van de patiënt en de psychosociale invloed van ouders op de patiënt. Een van de bestudeerde factoren is de aangezichtsgroei in HFM. We vonden dat patiënten meer terug liggende kaken hadden in vergelijking met de normale bevolking. De aangedane zijde vertoonde dit sterker dan de niet aangedane zijde. Voor de onderkaak geldt dat HFM patiënten starten met een kleinere onderkaak en ook eindigen met een kleinere onderkaak. Tijdens de groei blijft de verhouding tussen aangedane en niet aangedane zijde gelijk. Er is geen toenemende gezichtsasymmetrie zichtbaar. Deze kleinere onderkaak suggereert dat er ook een afwijking aan de lokale tandontwikkeling bestaat. We vonden een vertraagde tandontwikkeling in ernstig aangedane jonge HFM patiënten. Dit en de verdeling van aangeboren afwezige gebitselementen gerelateerd aan de ernst van de aandoening doen vermoeden dat er een vroege interactie bestaat tussen tand- en onderkaaksontwikkeling. De psychosociale implicaties van HFM op patiënt en ouders zijn onvoldoende bestudeerd. Voor het aanpassen aan de stressvolle gebeurtenis van het krijgen van een kind met een aangezichtsafwijking, zoals HFM, kan een grote rol zijn weggelegd voor verwerkingsstrategieën door de ouders. We onderzochten ouderlijke stress in relatie tot zowel kenmerken van het kind als de ouderlijke cognitieve verwerkingsprocessen. De manier van verwerking door ouders blijkt een groot deel van de ouderlijke stress te verklaren naast bepaalde kenmerken van hun kind. Vervolg onderzoek zou zich ook op functionele veranderingen moeten richten, zoals beweging van de onderkaak, esthetiek bij het lachen en de effectiviteit van kauwen waarbij rekening wordt gehouden met de drie dimensionale aard van de afwijking. Er kan dieper in gegaan worden op de manier waarop ouders verwerken dat zij een kind hebben met HFM en de strategie die zij daarvoor gebruiken. Dit kan het eindresultaat van de behandeling van hun kind beïnvloeden en daarmee, de kwaliteit van leven van hun kind

Erratum to: Transverse dental arch relationship at 9 and 12 years in children with unilateral cleft lip and palate treated with infant orthopedics: a randomized clinical trial (DUTCHCLEFT)

The original version of the above article contained a mistakedue to incorrect alignment of entries in Tables 11 and 14during production. The entries are presented correctly below

Influence of self-esteem on perceived orthodontic treatment need and oral health-related quality of life

Background: Self-esteem (SE) is suggested to influence the relationship between orthodontic treatment need and oral health related quality of life (OHRQoL), but evidence lacks. The aim of the present study was to investigate SE in the relationship between subjective orthodontic treatment need (SOT) and OHRQoL in children. Methods: This cross-sectional study was embedded in the Generation R Study, a multi-ethnic population-based cohort. In total, 3849 10-year old children participated in the present study. OHRQoL, measured with the Child Oral Health Impact Profile-ortho, and SOT were assessed within parental questionnaires. SE was measured with a modified version of the Harter’s self-perception profile rated by the children. The role of SE in the association between SOT and OHRQoL was evaluated with linear regression mode

Ancestry and dental development: A geographic and genetic perspective

Objective: In this study, we investigated the influence of ancestry on dental development in the Generation R Study. Methods: Information on geographic ancestry was available in 3,600 children (1,810 boys and 1,790 girls, mean age 9.81±0.35 years) and information about genetic ancestry was available in 2,786 children (1,387 boys and 1,399 girls, mean age 9.82±0.34 years). Dental development was assessed in all children using the Demirjian method. The associations of geographic ancestry (Cape Verdean, Moroccan, Turkish, Dutch Antillean, Surinamese Creole and Surinamese Hindustani vs Dutch as the reference group) and genetic content of ancestry (European, African or Asian) with dental development was analyzed using linear regression models. Results: In a geographic perspective of ancestry, Moroccan (β=0.18; 95% CI: 0.07, 0.28), Turkish (β=0.22; 95% CI: 0.12, 0.32), Dutch Antillean (β=0.27; 95% CI: 0.12, 0.41), and Surinamese Creole (β=0.16; 95% CI: 0.03, 0.30) preceded Dutch children in dental development. Moreover, in a genetic perspective of ancestry, a higher proportion of European ancestry was associated with decelerated dental development (β=-0.32; 95% CI: -.44, -.20). In contrast, a higher proportion of African ancestry (β=0.29; 95% CI: 0.16, 0.43) and a higher proportion of Asian ancestry (β=0.28; 95% CI: 0.09, 0.48) were associated with accelerated dental development. When investigating only European children, these effect estimates increased to twice as large in absolute value. Conclusion: Based on a geographic and genetic perspective, differences in dental development exist in a population of heterogeneous ancestry and should be considered when describing the physiological growth in children

The association of maternal folic acid supplementation and prenatal folate and vitamin B12 concentrations with child dental development

Objective: Low folic acid, folate and vitamin B12 might affect tooth formation and mineralization. The conversion of folic acid into folate is catalysed by the methylenetetrahydrofolate (MTHFR) enzyme which is encoded by the MTHFR gene. Among 3728 mothers and their 10-year-old children from the Generation R Study, we investigated associations of maternal folic acid supplementation and prenatal folate and vitamin B12 concentrations with child dental development. Secondly, we checked the modifying effect of MTHFR-C677T polymorphism. Methods: Information on folic acid supplementation was obtained by questionnaires. Concentrations of folate and vitamin B12 were measured from venous samples taken in early pregnancy. Developmental stages of teeth were defined by the Demirjian method at the age-10 assessment. In addition, dental age of the children was calculated using the Dutch standard. GLM and multivariate linear regression models were built to study the associations. Results: Folic acid supplementation started when pregnancy was known (β = −0.09; 95% CI: −0.17, −0.01) and folic acid supplementation started prior to known pregnancy (β = −0.12; 95% CI: −0.20, −0.04) were both associated with decelerated dental development by 1-2 months lower dental age of 10-year-old children. Folate (β = −0.02, 95% CI: −0.05, 0.02) and vitamin B12 (β = 0.03, 95% CI: −0.00, 0.06) were not associated with dental age. MTHFR-C677T did not modify the associations. Conclusions: Maternal folic acid supplementation delays dental development of children by 1-2 months dental age, whereas maternal folate and vitamin B12 concentrations in early pregnancy do not affect the timing of child dental development

Impact of orthodontic treatment need and deviant occlusal traits on oral health–related quality of life in children: A cross-sectional study in the Generation R cohort

Introduction: Many studies have investigated the impact of orthodontic treatment need (OTN) on children's oral health–related quality of life (OHRQOL). However, few studies have explored the impact of deviant occlusal traits on OHRQOL re

Permanent tooth agenesis in non-syndromic Robin sequence and cleft palate: prevalence and patterns

Objectives: Partial tooth agenesis is frequently observed in Robin sequence. Tooth anomalies are increasingly considered as an extended phenotype of the cleft palate population. The study objective was to compare the prevalence and patterns of tooth agenesis in a group of patients with non-syndromic Robin sequence (ns-RS) and a group with non-syndromic cleft palate (ns-CP). Materials and methods: The panoramic radiographs of 115 ns-RS and 191 ns-CP patients were assessed for agenesis of the permanent dentition (excluding third molars) and the patterns recorded using the Tooth Agenesis Code. Results: Partial tooth agenesis was observed in 47.8% of ns-RS and 29.8% of ns-CP patients with a greater prevalence in the mandibula than in the maxilla, particularly in ns-RS. The teeth most frequently absent in both groups were the mandibular second premolars and maxillary lateral incisors. Tooth agenesis was bilateral in two-thirds of affected ns-RS patients and one-half of ns-CP patients. In ns-RS, bilateral agenesis of the mandibular second premolars was more frequently observed in female than that in male patients. Completely symmetrical patterns of hypodontia were found in around 45% of ns-RS patients with tooth agenesis compared to 35% in ns-CP. No association was found between the extent of the palatal cleft and the severity of hypodontia. Conclusion: Tooth agenesis is more prevalent in ns-RS than that in ns-CP, demonstrates a much greater predilection for the mandible in ns-RS, and bears no relation to the extent of the palatal cleft. Clinical relevance: When compared to ns-CP, additional developmental disturbances are likely involved in the etiology of tooth agenesis in ns-RS. Future research could help identify the underlying genetic traits and aid in classifying patients in those with and without expected tooth agenesis in order to facilitate orthodontic management strategies

Foetal, neonatal and child vitamin D status and enamel hypomineralization

Objectives: Recent literature suggested that higher vitamin D concentrations in childhood are associated with a lower prevalence of molar incisor hypomineralization (MIH). As tooth development already starts in utero, we aimed to study whether vitamin D status during foetal, postnatal and childhood periods is associated with the presence of hypomineralized second primary molars (HSPMs) and/or MIH at the age of six. Methods: Our study was embedded in the Generation R Study, a population-based, prospective cohort from foetal life onwards in Rotterdam, the Netherlands. HSPMs and MIH were scored from intraoral photographs of the children at their age of six. Serum 25(OH)D concentrations were measured at three points in time, which resulted in three different samples; mid-gestational in mothers' blood (n = 4750), in umbilical cord blood (n = 3406) and in children's blood at the age of 6 years (n = 3983). Results: The children had a mean (±SD) age of 6.2 (±0.5) years at the moment of taking the intraoral photographs. After adjustment for confounders, no association was found between foetal 25(OH)D concentrations and the presence of HSPMs (OR 1.02 per 10 nmol/L higher 25(OH)D, 95% CI: 0.98-1.07) or MIH (OR 1.05 per 10 nmol/L increase, 95% CI: 0.98-1.12) in 6-year-olds. A higher 25(OH)D concentration in umbilical cord blood resulted in neither lower odds of having HSPM (OR 1.05, 95% CI: 0.98-1.13) nor lower odds of having MIH (OR 0.95, 95% CI: 0.84-1.07) by the age of six. Finally, we did not find higher 25(OH)D concentrations at the age of six to be associated with a significant change in the odds of having HSPM (OR 0.97, 95% CI: 0.92-1.02) or MIH (OR 1.07, 95% CI: 0.98-1.16). Conclusions: 25(OH)D concentrations in prenatal, early postnatal and later postnatal life are not associated with the presence of HPSMs or wi

Rare and Common Variants Conferring Risk of Tooth Agenesis

We present association results from a large genome-wide association study of tooth agenesis (TA) as well as selective TA, including 1,944 subjects with congenitally missing teeth, excluding third molars, and 338,554 controls, all of European ancestry. We also tested the association of previously identified risk variants, for timing of tooth eruption and orofacial clefts, with TA. We report associations between TA and 9 novel risk variants. Five of these variants associate with selective TA, including a variant conferring risk of orofacial clefts. These results contribute to a deeper understanding of the genetic architecture of tooth development and disease. The few variants previously associated with TA were uncovered through candidate gene studies guided by mouse knockouts. Knowing the etiology and clinical features of TA is important for planning oral rehabilitation that often involves an interdisciplinary approach