The prevalence of cleft in the Netherlands and Northern Netherlands in 1997-2007:Trend analysis of data from three Dutch registries

Objective. To investigate the prevalence of oral cleft live births in the Netherlands, we analyzed time-trends in the Netherlands and Northern Netherlands (NNL) over the period 1997-2007 and stratified these trends by cleft category (cleft lip/alveolus ± cleft palate: CL±P; cleft palate only: CP). Methods. Patients born alive with oral clefts in the Netherlands during 1997-2007 were included from three Dutch registries on congenital anomalies/oral clefts. The estimated annual percentage change (EAPC) in prevalence was calculated using Poisson regression. Results. The overall prevalence of oral clefts per 10,000 live births was 16.6 in the Netherlands and 21.4 in NNL. During 1997-2007, the live-birth prevalence of oral clefts in the Netherlands decreased significandy (EAPC -2.0%; 95% CI: -3.0% to -0.90%), as did the CL±P prevalence (EAPC -2.1%; 95% CI: -3.4% to -0.80%), while no significant trend in CP was found. The rates of the region NNL showed no significant trends. Discussion. The national decrease in prevalence may be explained by two hypotheses: 1) greater prenatal detection of congenital anomalies including oral clefts has led to more pregnancy terminations; and 2) increased periconceptional folic acid use has reduced the risk of oral clefts. Both hypotheses would mainly apply to the category CL±P, since, unlike CP, this category can be detected prenatally by 2D ultrasound and develops during the period recommended for folic acid use. Conclusion. Because the CL±P prevalence in the Netherlands decreased, that of all oral clefts decreased. Although the rates in the region NNL were higher, they showed no significant trends

Stem cells and extracellular vesicles to improve preclinical orofacial soft tissue healing

Orofacial soft tissue wounds caused by surgery for congenital defects, trauma, or disease frequently occur leading to complications affecting patients' quality of life. Scarring and fibrosis prevent proper skin, mucosa and muscle regeneration during wound repair. This may hamper maxillofacial growth and speech development. To promote the regeneration of injured orofacial soft tissue and attenuate scarring and fibrosis, intraoral and extraoral stem cells have been studied for their properties of facilitating maintenance and repair processes. In addition, the administration of stem cell-derived extracellular vesicles (EVs) may prevent fibrosis and promote the regeneration of orofacial soft tissues. Applying stem cells and EVs to treat orofacial defects forms a challenging but promising strategy to optimize treatment. This review provides an overview of the putative pitfalls, promises and the future of stem cells and EV therapy, focused on orofacial soft tissue regeneration

Aetiological study of the presumed ocular histoplasmosis syndrome in the Netherlands

Aim. To investigate whether presumed ocular histoplasmosis syndrome in the Netherlands is caused by Histoplasma capsulatum and whether other risk factors might play a role in the pathogenesis of this syndrome. Methods. 23 patients were clinically diagnosed as having presumed ocular histoplasmosis syndrome based on the following criteria: peripapillary atrophy, punched out lesions, a macular disciform lesion or scar in one eye without vitritis. As controls, 66 sex and age matched healthy volunteers were used. Serum samples from both patients and controls were tested for the presence of antibodies against H capsulatum, Toxoplasma gondii, Toxocara canis et cati, Ascaris sp, and for the presence of antigens of Cryptococcus neoformans. Serum samples were also tested for the presence of autoantibodies against retinal or choroidal proteins. To investigate other risk factors, patients and controls were asked to fill in a health and travel related questionnaire. Ten patients with ocular toxoplasmosis were used as a disease control group. Results. None of the patients with presumed ocular histoplasmosis syndrome or controls had circulating antibodies directed against H capsulatum. No risk factors could be identified and no indications for autoimmunity and no evidence for the role of the other infectious agents could be demonstrated. Conclusions. In a Dutch group of patients fulfilling the criteria of a disease currently named presumed ocular histoplasmosis syndrome, no risk factors or relation with the fungus H capsulatum could be detected

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

Dental Maturation in Children With the Syndrome of Crouzon and Apert

Purpose: Developing teeth are used to assess maturity and estimate age in a number of disciplines. The purpose of this investigation was to study the dental maturation in children with Crouzon or Apert syndrome compared with nonsyndromic controls. Patients and Methods: Records of 40 children with Crouzon syndrome (18 boys and 22 girls, aged 4.0 to 17.9 years) and 28 children with Apert syndrome (10 boys and 18 girls, aged 3.9 to 15.1 years) were referred to the Department of Orthodontics, Cleft Palate Team and Craniofacial Team, Erasmus MC-Sophia. Data from syndromic children were compared with data from 451 nonsyndromic children (225 boys and 226 girls, aged 2.9 to 16.9 years). From panoramic radiographs, dental maturation was determined for patients with Crouzon and Apert syndromes and compared with data collected from control children. Logistic functions were constructed for dental maturation over time for syndromes and gender. Results: Statistically significant gender differences in dental maturation scores were found for girls with Crouzon (P < .05) and Apert syndrome (P < .05). Patients with Apert syndrome demonstrated a significantly delayed dental maturation (P < .05), while patients with Crouzon syndrome showed a nonsignificant delay. Conclusions: Dental maturation in patients with Apert syndrome was more delayed than in patients with Crouzon syndrome. The delay of tooth formation in patients with Crouzon or Apert syndrome suggests a possible common genetic association

Dental development in hemifacial microsomia.

Item does not contain fulltextHemifacial microsomia (HFM) is a congenital disorder marked by facial asymmetry. Whether facial asymmetry accounts for asymmetrical dental development is unknown. There are few data on dental development relative to mandibular development or severity of HFM, or on development over time. We hypothesized that when mandibular development was severely disturbed, local dental development was also affected. We compared dental development scores between affected and non-affected mandibular sides in patients with HFM (n = 84) and compared these data with those collected from Dutch control children (n = 451). Logistic functions were constructed for dental age over time for all four Pruzansky/Kaban types. The results showed a tendency toward delayed dental development in Pruzansky/Kaban types IIb and III at younger ages. The temporary delay of tooth formation in patients with severe forms of HFM and the distribution of agenic teeth suggest an interaction between mandibular and dental development.1 december 201