A systematic review: facial, dental and orthodontic findings and orofacial diagnostics in patients with FASD

BackgroundThe fetal alcohol spectrum disorder is a group of developmental disorders caused by maternal alcohol consumption. Patients with fetal alcohol syndrome show abnormal orofacial features. This review presents an overview over the facial, oral, dental or orthodontic findings and diagnostic tools concerning these features.MethodsFor this systematic review Cochrane, Medline and Embase databases were considered and the review was performed according to the PRISMA checklist. Two independent reviewers evaluated all studies and recorded results in a summary of findings table. Risk of bias was analyzed via Quadas-2 checklist.Results61 studies were eligible for inclusion. All included studies were clinical studies. Methods and results of the studies were not comparable, guidelines or methods for the detection of FASD varied across studies. Facial features most often measured or found as distinguishing parameter were: palpebral fissure length, interpupillary or innercanthal distance, philtrum, upper lip, midfacial hypoplasia or head circumference.ConclusionsThis review shows that to date a multitude of heterogeneous guidelines exists for the diagnosis of FASD. Uniform, objective diagnostic criteria and parameters for the orofacial region in FASD diagnosis are needed. A bio database with values and parameters for different ethnicities and age groups should be made available for diagnosis

Effects of estrogen deficiency during puberty on maxillary and mandibular growth and associated gene expression – an μCT study on rats

Background Estrogen is a well-known and important hormone involved in skeletal homeostasis, which regulates genes involved in bone biology. Some studies support that estrogen is important for craniofacial growth and development. Therefore this in vivo animal study aimed to investigate, whether and in which way low estrogen levels in the prepubertal period affect craniofacial development in the postpubertal stage and to quantify the gene expression of RANK, RANKL and OPG in cranial growth sites in ovariectomized estrogen-deficient rats during puberty. Methods Control (sham-operated, n = 18) and ovariectomy (OVX, n = 18) surgeries were performed on 21-days-old female Wistar rats. Animals euthanized at an age of 45 days (pubertal stage) were used for gene expression analyses (n = 6 per group) and immunohistochemistry of RANK, RANKL and OPG. Animals euthanized at 63 days of age (post-pubertal stage) were used for craniofacial two-dimensional and three-dimensional craniofacial measurements using μCT imaging (n = 12 per group). Results In the μCT analysis of the mandible and maxilla many statistically significant differences between sham-operated and OVX groups were observed, such as increased maxillary and mandibular bone length in OVX animals (p < 0.05). Condylar volume was also significantly different between groups (p < 0.05). The sham-operated group showed a higher level of RANK expression in the midpalatal suture (p = 0.036) and the RANKL:OPG ratio levels were higher in the OVX group (p = 0.015). Conclusions Our results suggest that estrogen deficiency during the prepubertal period is associated with alterations in the maxillary and mandibular bone length and condylar growth

Tooth Malformations, DMFT Index, Speech Impairment and Oral Habits in Patients with Fetal Alcohol Syndrome

Background: Fetal alcohol spectrum disorder (FASD) is a developmental disorder with severe negative lifetime consequences. Although knowledge about the harmfulness of alcohol consumption during pregnancy has spread, the prevalence of fetal alcohol spectrum disorder is very high. Our study aims at identifying fetal alcohol syndrome (FAS)-associated dental anomalies or habits, which need early attention. Methods: Sixty children (30 FAS; 30 controls) were examined prospectively. Swallowing pattern, oral habits, breastfeeding, speech therapy, ergotherapy, physiotherapy, exfoliation of teeth, DMFT (decayed, missing, filled teeth) index, modified DDE (developmental defects of enamel) index and otitis media were recorded. Results: Swallowing pattern, exfoliation of teeth, and otitis media were not significantly different. Significant differences could be found concerning mouthbreathing (p = 0.007), oral habits (p = 0.047), age at termination of habits (p = 0.009), speech treatment (p = 0.002), ergotherapy, physiotherapy, and breastfeeding (p &le; 0.001). DMFT (p &le; 0.001) and modified DDE (p = 0.001) index showed significantly higher values for children with fetal alcohol syndrome. Conclusions: Children with fetal alcohol syndrome have a higher need for early developmental promotion such as speech treatment, ergotherapy, and physiotherapy. Mouthbreathing, habits, and lack of breastfeeding may result in orthodontic treatment needs. High DMFT and modified DDE indexes hint at a higher treatment and prevention need in dentistry

3D-Analysis of Mouth, Nose and Eye Parameters in Children with Fetal Alcohol Syndrome (FAS)

Background: Fetal alcohol spectrum disorder (FASD) is a developmental disorder with severe negative lifetime consequences for the affected person. Numerous diagnostic methods for facial assessment in FAS exist, but most of them are based on subjective evaluations. Our aim was therefore to find objective methods for the verification of FAS(D). Methods: 58 children (28 FAS; 30 controls) were examined prospectively. 3D facial scans were performed for each child and facial parameters at the mouth, nose and eye regions were measured and compared between the groups. Results: Significant differences could be found for the distance between right and left sulcus nasi at the transition point to the philtrum (p &lt; 0.001), for the inner canthal distance (p = 0.001) as well as for the right and left palpebral fissure length (p &lt; 0.001). No significant difference between the FAS and control children could be found for the measurements of mouth breadth (p = 0.267) and breadth between the left and right alares nasi (p = 0.260). Conclusions: Measurements of mouth breadth and nose breadth for the alares nasi are not suitable for FAS diagnosis. In contrast, digital contactless measurements of the distance between the right and left sulcus nasi at the transition point to the philtrum, as well as the inner canthal distance and palpebral fissure length of the left and right eyes, showed significant differences when comparing children with FAS to healthy controls. These measurements could thus be additional objective means for the verification of FAS

Oral Conditions and Oral Health-Related Quality of Life of People with Ehlers-Danlos Syndromes (EDS): A Questionnaire-Based Cross-Sectional Study

Background and objective: To date, there have only been a few studies on oral health-related quality of life (OHRQoL) of people with Ehlers&ndash;Danlos syndromes (EDS) and oral conditions. The aim of this study was, therefore, to analyze the OHRQoL of people with EDS from their own point of view as well as obtain information about their age at the time of the diagnosis, the period of time until diagnosis, and the presence of oral conditions (if any) and their association with oral health quality. Methods: The study was designed as an anonymous questionnaire-based cross-sectional study. We conducted a descriptive analysis of the Oral Health Impact Profile-14 (OHIP-14) scores, age of the participants, age at diagnosis, and the time-period between the first signs of the disease and the diagnosis of EDS. To verify the differences in OHIP-14 scores between patients with and without oral conditions, a Mann&ndash;Whitney U test was performed. A multivariate quantile (median) regression analysis was performed to evaluate the effect of different general characteristics (gender, age, and the presence of oral conditions) on the OHIP 14 scores. Furthermore, using a Mann&ndash;Whitney U test, the influence of different oral conditions was verified by testing the differences between patients without any oral conditions and patients with a specific diagnosis. Results: A total of 79 evaluable questionnaires from 66 female (83.5%) and 13 male (16.5%) participants were analyzed. On average, after the first condition, it takes 18.36 years before EDS are correctly diagnosed. Oral conditions were described by 69.6% of the participants. The median (interquartile range) OHIP-14 score was eight (ten) points for patients without oral conditions and 19 (15) for patients with oral conditions. The multivariable quantile regression shows a statistical notable association between OHIP-14 score and oral conditions (p &lt; 0.001). OHIP-14 scores for dysgnathia, periodontitis, TMD (Temporomandibular dysfunction), a high-arched palate, malocclusion, and the anomaly of tooth formation were statistical notably different between the participants with and the participants without oral conditions. Conclusions: Long diagnostic pathways seem to be a typical problem in patients with EDS. Oral conditions associated with the underlying disease occurred regularly and showed a negative correlation with OHRQoL

Malocclusion Can Give Additional Hints for Diagnosis of Fetal Alcohol Spectrum Disorder

FASD prevalence is high and diagnosis is very difficult; Malocclusions can give additional hints for FASD diagnosis; Patients with FASD show growth deficits concerning the maxilla; Early and consistent orthodontic supervision and therapy can prevent facial asymmetries in FASD patients. Abstract Aims Fetal alcohol spectrum disorder (FASD) is a developmental disorder caused by maternal alcohol intake (prevalence: 0.77%). Malocclusion has been described in case reports of patients with FASD, but reliable evidence for associations between FASD and malocclusion is not documented. Malocclusion is defined as tooth irregularity (prevalence: 14.6%) or incorrect relationship between the jaws such as lateral crossbites (prevalence: 3.1%). The purpose of this study was to investigate possible associations between malocclusion and FASD. Short summary FASD prevalence is high and diagnosis is very difficult; Malocclusions can give additional hints for FASD diagnosis; Patients with FASD show growth deficits concerning the maxilla; Early and consistent orthodontic supervision and therapy can prevent facial asymmetries in FASD patients. Methods Thirty patients with FASD and 30 patients of a healthy control group were examined. Inclusion criteria were mixed dentition, verified FASD/absence of FASD (control group), exclusion criteria were orthodontic treatment and disorders other than FASD. The extent and type of malocclusion were quantified with the peer assessment rating (PAR) index based on an analysis of orthodontic plaster models. In addition, anthropometric data such as gestational age, body weight and height at birth as well as present body weight, height and head circumference at examination date were assessed. Results The PAR index showed a significant increase in malocclusions in FASD patients compared to the group that were not diagnosed with FASD (P = 0.002). FASD patients showed particular differences in the upper transversal dimension with a higher prevalence of crossbites (P = 0.018) and a lower head circumference (P < 0.001). Body weight (P < 0.001) and height (P < 0.001) were significantly lower for FASD patients at time of birth, but not at the present examination date (weight: P = 0.329; height: P = 0.496). When relating weight and height measures to age using percentile curves of physiological growth, clinically relevant discrepancies could be found for FASD patients. Conclusions Our results show that malocclusion can provide additional evidence for FASD diagnosis. When FASD is diagnosed in a child, early referral to an orthodontist is advisable to stimulate maxillary growth and consequently prevent further malocclusions

Orthodontic treatment of children/adolescents with special health care needs: an analysis of treatment length and clinical outcome

Background: The aim of this retrospective study was to analyse the treatment time and differences between the pre- and post-treatment peer assessment rating (PAR) index and aesthetic component (AC) of the index of orthodontic treatment need (IOTN) scores in children/adolescents with special health care needs (SHCNs), compared to non-special health care needs (NSHCNs) controls. Methods: Based on certain inclusion and exclusion criteria, medical records of SHCNs and randomly selected NSHCNs controls at the Department of Orthodontics, University Hospital Muenster were analysed retrospectively for the treatment time, number of appointments, chair time (“moderate” or “considerable”), PAR scores, and AC scores. Sample size calculation, descriptive statistics, and explorative analyses were performed using the Mann–Whitney U Test. Results: Twenty-nine children with SHCNs (21 boys, 9 girls; median age: 11 years, pre-treatment) and 29 children with NSHCNs (12 boys, 17 girls; median age: 12 years, pre-treatment) were enrolled in this study. The overall treatment time did not differ between the patient groups. However, more “considerable chair time” was needed for the SHCNs group compared to the control group (p Conclusions: While the overall treatment time and number of appointments did not differ, the overall chair time was higher in the SHCNs group. The pre- and post-treatment PAR and AC scores were significantly higher in the SHCNs group.<br

3D Analysis of Philtrum Depth in Children with Fetal Alcohol Syndrome

Aims Diagnosis of fetal alcohol spectrum disorder (FASD) is complex and difficult. The estimated number of unreported FASD is thus assumed to be substantial. In our cross-sectional study, we aimed to identify possible metric differences in philtrum depth in children with fetal alcohol syndrome (FAS) compared to healthy controls based on non-invasive 3D facial scanning in order to provide an objective, metrical tool improving FASD diagnosis. Methods Twenty-five children with confirmed FAS and 30 healthy school children without FAS, both in the mixed dentition, were prospectively recruited and 3D facial scans were performed after recording body length, weight and head circumference. Philtrum surface data were extracted and metric philtrum depth was determined at four geometrically defined measuring points (P1-P4) along the vertical length of the philtrum. Results Philtrum depths at P1 (P = 0.025), P2 (P = 0.001), P3 (P < 0.001) and P4 (P = 0.001) as well as mean philtrum depth P1-P4 (P < 0.001) differed significantly between patients with and without FAS. Compared to controls, the philtrum was shallower in patients with FAS by on average 0.4 mm at each of the respective points. Whereas no differences could be determined for body height and weight, head circumference was significantly smaller in patients with FAS (P = 0.001), particularly in girls (P = 0.008). Conclusions Apart from head circumference, philtrum depth is significantly reduced in children with FAS and can thus be used as diagnostic indicator to aid and confirm FAS diagnosis. In contrast to visual assessments, 3D face scan methods allow a more objective quantification and can thus provide additional evidence in FAS diagnosis

Asymmetry-index and orthodontic facial analysis of children with foetal alcohol syndrome using 3D-facial scans

Background The foetal alcohol spectrum disorder (FASD) is a complex and heterogenic disorder, caused by gestational exposure to alcohol. Patients with foetal alcohol syndrome (FAS-most severe form) show abnormal facial features. Our study aims at finding additional reliable and objective parameters for FAS diagnosis. Methods Facial three-dimensional scans of 30 children with FAS and 30 controls were analysed. Orthodontic profile analysis (concerning position of upper and lower jaw) was performed. Vertical facial proportions were taken and facial asymmetry index (right to left side) was calculated. Results Profile type was significantly different for children with FAS (p = 0.001) with lower jaws more frequently in a retral position. Profile angle was significantly larger in the group with FAS (p = 0.009). Children with FAS had shorter middle thirds and longer lower thirds of the face (p < 0.001). Stomion (point between upper and lower lip) was located significantly more caudally in the FAS group (p < 0.001). Facial asymmetry index was not significantly different. Conclusions Children with FAS differ significantly from controls in vertical and sagittal facial measurements. Profile analysis and measurement of vertical proportions are easy to apply standard procedures in everyday orthodontic practice and could be time-saving and objective means for additional verification of FAS

Impact of NSAID etoricoxib on side effects of orthodontic tooth movement

Objectives: The non-steroidal anti-inflammatory drug etoricoxib is the most highly selective inhibitor of cyclooxygenase-2 available (344:1) and has been approved for postoperative pain therapy following dental interventions in Europe. At clinically relevant doses it has been reported to only have marginal effects on the velocity of orthodontic tooth movement (OTM). Its effects on associated dental root resorptions, osteoclastogenesis, trabecular number in the alveolar bone and periodontal bone loss during OTM, however, have not yet been investigated. Material and methods: 40 male Fischer344 rats were divided into four groups: 1.5 ml tap water/day p.o. (control, 1), additional 7.8 mg/kg/day etoricoxib (normal dose) for three (2) or seven (3) days/week and 13.1 mg/kg/day (high dose) for seven days/week, respectively (4). After a week of premedication, OTM in anterior direction of the first left upper molar was performed for 28 days by means of a nickel-titanium coil spring (0.25 N). We quantified OTM-associated dental root resorptions, osteoclastogenesis, trabecular number and periodontal bone loss by histomorphometrical, histochemical and mu CT analyses of the disected tooth-bearing upper jaw sections. Results: After 28 days of OTM, associated reduction of trabecular number seemed to be slightly alleviated by high doses of etoricoxib, whereas no significant other etoricoxib effects in the doses administered could be detected regarding OTM-induced or -associated dental root resorptions, osteoclastogenesis or periodontal bone loss. Conclusions: Dental root resorptions, osteoclastogenesis and periodontal bone loss during OTM in rats were not significantly affected by etoricoxib in the clinically relevant dosages investigated with only a slight inhibitory effect on bone remodelling to be expected at high dosages. Etoricoxib is therefore not suitable for the prevention of these detrimental effects, but could be a suitable analgesic during OTM, as it has been reported not to affect tooth movement. (C) 2020 Elsevier GmbH. All rights reserved