Background Children missing 6 or more permanent teeth often present with complex dental care needs and significant impacts on their oral health related quality of life (OHRQoL). The most important facet in the overall care for these children is the child’s own experience, but parents primarily make the decisions regarding their child’s dental management. Understanding the parental perspective could have a positive impact on planning and provision of care for these patient groups in the future. The study compared the parental perspectives on OHRQoL impact and dental experience for children with ectodermal dysplasia (ED), severe isolated hypodontia (IH), and matched controls following assessment of their dental features. Design A cross-sectional study of 172 children (mean age: 12.4 years old) was conducted; 86 with severe hypodontia (≥ 6 missing teeth; ED: 29; IH: 57) and 86 age and gender matched controls. The Parental-Caregiver Perceptions Questionnaire (P-CPQ), Family Impact Scale (FIS) and a supplemental questionnaire were used to gather information on parental perceptions of OHRQoL and dental experiences, respectively. Clinical examinations were used to assess and compare the dental features between children with ED, IH and their respective controls. Results Higher scores (p < 0.05) were found in P-CPQ and FIS scores between the children with ED, IH and their respective controls. P-CPQ scores for males with ED had a moderate correlation with functional limitations (Rs = 0.576; p = 0.001*), oral symptoms (Rs = 0.444; p = 0.016*) and overall QoL (Rs = 0.499; p = 0.006*). The ED group reported earlier awareness of issues, the youngest attendance (3.24 years) and highest perceived number of appointments (“20 or more”; 58.6%). The mean number of missing teeth in the ED group was almost twice that of the IH group (ED: 20.17; IH: 10.68) and the median number of missing teeth (Radiographically: ED = 21; IH = 9; Clinically: ED = 11; IH = 6), was significantly greater in the ED group when compared to the IH group (p < 0.001*). Conclusion Parents of children with ED and IH perceive a greater impact on QoL, for both the child and their family. Children with ED need earlier intervention and more extensive treatment than children with IH and their controls.

Data sourcesCochrane, PubMed and Ovid Medline. A search timeline of January 1966 to May 2015 was used and language was restricted to English.Study selectionCase-control and cohort studies relating to oral clefts, with information on maternal active smoking, before and after conception, excluding paternal smoking and exposure to environmental tobacco smoke.Data extraction and synthesisTwo authors extracted data independently. Both Begg's and Egger's funnel plots were used to assess publication bias. Sensitivity analysis was performed to assess impact of the different study types and chi-squared and P values were determined by SPSS software. Studies with missing data were excluded from the meta-analysis.ResultsA statistically significant association was found between maternal active smoking and CL +/- P (odds ratio [OR] 1.368; 95% confidence interval [CI] 1.259-1.486) as well as CP (OR 1.241; 95% CI 1.117-1.378). Half the studies showed positive dose-response effect for each subgroup (test for linear trend, P < .05).ConclusionsWomen who smoke during pregnancy have a moderate risk of having a child with a CL +/- P or CP. A dose-response effect between maternal smoking and clefts was not established.