Mette A. R. Kuijpers

Three-dimensional imaging methods for quantitative analysis of facial soft tissues and skeletal morphology in patients with orofacial clefts: a systematic review.

Background Current guidelines for evaluating cleft palate treatments are mostly based on two-dimensional (2D) evaluation, but three-dimensional (3D) imaging methods to assess treatment outcome are steadily rising. Objective To identify 3D imaging methods for quantitative assessment of soft tissue and skeletal morphology in patients with cleft lip and palate. Data sources Literature was searched using PubMed (1948–2012), EMBASE (1980–2012), Scopus (2004–2012), Web of Science (1945–2012), and the Cochrane Library. The last search was performed September 30, 2012. Reference lists were hand searched for potentially eligible studies. There was no language restriction. Study selection We included publications using 3D imaging techniques to assess facial soft tissue or skeletal morphology in patients older than 5 years with a cleft lip with/or without cleft palate. We reviewed studies involving the facial region when at least 10 subjects in the sample size had at least one cleft type. Only primary publications were included. Data extraction Independent extraction of data and quality assessments were performed by two observers. Results Five hundred full text publications were retrieved, 144 met the inclusion criteria, with 63 high quality studies. There were differences in study designs, topics studied, patient characteristics, and success measurements; therefore, only a systematic review could be conducted. Main 3D-techniques that are used in cleft lip and palate patients are CT, CBCT, MRI, stereophotogrammetry, and laser surface scanning. These techniques are mainly used for soft tissue analysis, evaluation of bone grafting, and changes in the craniofacial skeleton. Digital dental casts are used to evaluate treatment and changes over time. Conclusion Available evidence implies that 3D imaging methods can be used for documentation of CLP patients. No data are available yet showing that 3D methods are more informative than conventional 2D methods. Further research is warranted to elucidate it. Systematic review registration International Prospective Register of Systematic Reviews, PROSPERO CRD42012002041

Dental Arch Relationships following Palatoplasty for Cleft Lip and Palate Repair

Palatal scarring is assumed to be a primary cause of facial growth derangement in cleft lip and palate. Evidence supporting this hypothesis is confounded by the clinical involvement of various surgeons, and therefore definitive conclusions are not possible. In this study, we investigated the dental arch relationship in two groups, Exposed (47 children; 11.2 yrs) and Unexposed (61 children; 11.2 yrs), with a unilateral cleft lip and palate operated on by the same surgeon. The technique of hard palate repair differed between the two groups. In the Exposed group, palatal bone of the non-cleft side only was left denuded, inducing scar formation. In the Unexposed group, a vomerplasty with tight closure of the soft tissues was applied. Three raters graded the dental arch relationship and palatal morphology using the EUROCRAN Index. The dental arch relationship in the Exposed group was less favorable than in the Unexposed group (p = 0.009). Palatal morphology in both groups was comparable (p = 0.323). This study demonstrates that reduction of denuded bony areas on the palate after palatal repair with a vomer flap had a favorable effect on the dental arch relationship. For palatal morphology, no effect of the type of palatal repair was found.

Rating nasolabial appearance on three-dimensional images in cleft lip and palate: a comparison with standard photographs

BACKGROUND/OBJECTIVE Judgement of nasolabial aesthetics in cleft lip and palate (CLP) is a vital component of assessment of treatment outcome. It is usually performed based on two-dimensional (2D) facial photographs. An increasing use of three-dimensional (3D) imaging warrants an assessment if 3D images can substitute 2D photographs during aesthetic evaluation. The aim of this study was to compare reliability of rating nasolabial appearance on 3D images and standard 2D photographs in prepubertal children. METHODS Forty subjects (age: 8.8-12) with unilateral CLP treated according to a standardized protocol, who had 2D and 3D facial images were selected. Eight lay raters assessed nasal form, nasal deviation, vermilion border, and nasolabial profile on cropped 2D and 3D images using a 100-mm visual analogue scale (VAS). Additionally, raters answer two questions: 1. Do 2D or 3D images provide more information on nasolabial aesthetics? and 2. Is aesthetic evaluation easier on 2D or 3D images? RESULTS Intrarater agreement demonstrated a better reliability of ratings performed on 3D images than 2D images (correlation coefficients for 3D images ranged from 0.733 to 0.857; for 2D images from 0.151 to 0.611). The mean scores showed, however, no difference between 2D and 3D formats (>0.05). 3D images were regarded more informative than 2D images (P = 0.001) but probably more difficult to evaluate (P = 0.06). LIMITATIONS Basal view of the nose was not assessed. CONCLUSIONS 3D images seem better than 2D images for rating nasolabial aesthetics but raters should familiarize themselves with them prior to rating.

Regional facial asymmetries in unilateral orofacial clefts

OBJECTIVES Assess facial asymmetry in subjects with unilateral cleft lip (UCL), unilateral cleft lip and alveolus (UCLA), and unilateral cleft lip, alveolus, and palate (UCLP), and to evaluate which area of the face is most asymmetrical. METHODS Standardized three-dimensional facial images of 58 patients (9 UCL, 21 UCLA, and 28 UCLP; age range: 8.6-12.3 years) and 121 controls (age range 9-12 years) were mirrored and distance maps were created. Absolute mean asymmetry values were calculated for the whole face, cheek, nose, lips, and chin. One-way analysis of variance, Kruskal-Wallis, and t-test were used to assess the differences between clefts and controls for the whole face and separate areas. RESULTS Clefts and controls differ significantly for the whole face as well as in all areas. Asymmetry is distributed differently over the face for all groups. In UCLA, the nose was significantly more asymmetric compared with chin and cheek (P = 0.038 and 0.024, respectively). For UCL, significant differences in asymmetry between nose and chin and chin and cheek were present (P = 0.038 and 0.046, respectively). In the control group, the chin was the most asymmetric area compared to lip and nose (P = 0.002 and P = 0.001, respectively) followed by the nose (P = 0.004). In UCLP, the nose, followed by the lips, was the most asymmetric area compared to chin, cheek (P < 0.001 and P = 0.016, respectively). LIMITATIONS Despite division into regional areas, the method may still exclude or underrate smaller local areas in the face, which are better visualized in a facial colour coded distance map than quantified by distance numbers. The UCL subsample is small. CONCLUSION Each type of cleft has its own distinct asymmetry pattern. Children with unilateral clefts show more facial asymmetry than children without clefts.

Anterior tooth wear and retention type until 5 years after orthodontic treatment

Objectives. To study occlusal wear of anterior teeth in orthodontic patients retained with different retainers until 5 years post-treatment, and to investigate whether type of retention influences occlusal wear. Material and methods. Orthodontic patients (n=222), aged 15 years maximally at the start of treatment, were followed until 5 years post-treatment. In the maxilla, a retainer bonded on all six teeth or a removable retainer was used; in the mandible, a lingual retainer was bonded on all anterior teeth or on canines only. Dental casts were analyzed before treatment (T0), after treatment (T1), and 5 years post-treatment (T5). Incisal and canine wear were scored by applying a grading scale. Intercanine width, overjet, and overbite were measured with an electronic caliper. Statistics used were: Paired samples t-test for differences over time; Pearson correlation coefficients for associations between wear and retention type; and backward linear regression for influence of retention type on wear. Results. There was an increase in wear during all time periods and for all teeth. From T0 to T5 an increase in maxillary intercanine width and maxillary retention had an effect on changes in canine wear. Incisal wear was associated with an increase in upper intercanine width (T1–T5). For both arches, an increase in maxillary intercanine width during treatment was associated with less progression of canine and incisal wear, but the explained variance was low, 13.4% and 19.3%, respectively. Conclusions. Retention type and, occasionally, an increase in intercanine width influence anterior teeth wear post-treatment. However, the clinical significance and impact of the examined retention methods on occlusal wear are small.

Novel BCOR mutations in patients with oculofaciocardiodental (OFCD) syndrome

Fig. 1. Patient 1 at age 22 years. Broad nasal tip, a long philtrum, facial asymmetry and thick, laterally curved eyebrows were noted (a), the orthopantomogram (OPT) shows radiculomegaly of canines (b). Patient 2 at age 20 months. Note prominent forehead, hypertelorism, microphthalmia of both eyes and a broad nasal tip (c). Patient 3 at age 18 years. Left-sided microphthalmia, mid-facial hypoplasia, a high nasal bridge and a broad nasal tip divided by a small cleft and a long philtrum were noted (d), the OPT shows radiculomegaly of the canines and congenitally absent permanent teeth (e). delayed eruption and persistent primary dentition (1, 2). The genetic analysis of OFCD patients revealed that heterozygous mutations in the BCOR (BCL-6 interacting corepressor) gene on chromosome Xp11.4 are responsible for the phenotype. Deletions, amino acid substitutions and splice-site mutations have been described (3, 4). Here, we report on the clinical and molecular data of three new patients with OFCD and

Secular trend of dental development in Dutch children

Many studies have established dental age standards for different populations; however, very few studies have investigated whether dental development is stable over time on a population level. Therefore, the aim of this study was to analyze changes in dental maturity in Dutch children born between 1961 and 2004. We used 2,655 dental panoramic radiographs of 2- to 16-year-old Dutch children from studies performed in three major cities in the Netherlands. Based on a trend in children born between 1961 and 1994, we predicted that a child of a certain age and gender born in 1963 achieved the same dental maturity on average, 1.5 years later than a child of the same age born 40 years later. After adjusting for the birth year of a child in the analysis, the regression coefficient of the city variable was reduced by 56.6% and it remained statistically significant. The observed trend from 1961 to 1994 was extrapolated to 9- to 10-year-old children born in 2002-2004, and validation with the other samples of children with the same characteristics showed that 95.9%-96.8% of the children had dental maturity within the 95% of the predicted range. Dental maturity score was significantly and positively associated with the year of birth, gender, and age in Dutch children, indicating a trend in earlier dental development during the observation period, 1961-2004. These findings highlight the necessity of taking the year of birth into account when assessing dental development within a population with a wider time span.

Myogenic capacity of muscle progenitor cells from head and limb muscles.

The restoration of muscles in the soft palate of patients with cleft lip and/or palate is accompanied by fibrosis, which leads to speech and feeding problems. Treatment strategies that improve muscle regeneration have only been tested in limb muscles. Therefore, in the present study the myogenic potential of muscle progenitor cells (MPCs) isolated from head muscles was compared with that of limb muscles. Muscle progenitor cells were isolated from the head muscles and limb muscles of rats and cultured. The proliferation of MPCs was analysed by DNA quantification. The differentiation capacity was analysed by quantifying the numbers of fused cells, and by measuring the mRNA levels of differentiation markers. Muscle progenitor cells were stained to quantify the expression of paired box protein Pax 7 (Pax-7), myoblast determination protein 1 (MyoD), and myogenin. Proliferation was similar in the head MPCs and the limb MPCs. Differentiating head and limb MPCs showed a comparable number of fused cells and mRNA expression levels of myosin-1 (Myh1), myosin-3 (Myh3), and myosin-4 (Myh4). During proliferation and differentiation, the number of Pax-7(+), MyoD(+), and myogenin(+) cells in head and limb MPCs was equal. It was concluded that head and limb MPCs show similar myogenic capacities in vitro. Therefore, in vivo myogenic differences between those muscles might rely on the local microenvironment. Thus, regenerative strategies for limb muscles might also be used for head muscles.

Maxillary arch width in unoperated adult bilateral cleft lip and alveolus and complete bilateral cleft lip and palate

OBJECTIVES To study maxillary arch width in adult patients with bilateral cleft lip and alveolus (BCLA) or with complete bilateral cleft lip and palate (BCLP), who have not had any surgery. SETTING AND SAMPLING POPULATION: Eighteen patients with BCLA, 13 patients with BCLP, and 24 controls from remote areas of Indonesia collected over 10 years. MATERIALS AND METHODS Dental casts were digitized three-dimensionally using an industrial coordinate measuring machine (CCM) (Zeiss Numerex; Carl Zeiss, Stuttgart, Germany). Transversal distance between molars was measured on the tip of the distobuccal cusp and the tip of the mesiobuccal cusp, and for premolars and canines, the tip of the buccal cusps was recorded. Means and standard deviations were calculated for all variables. t-Test was used to determine whether the mean values of the cleft groups showed significant differences from each other and from the controls. Level of significance was set at p < 0.05. RESULTS Transversal arch dimensions in the BCLA group were comparable to the controls except at the canine level. Intercanine distance, which is close to the alveolar cleft, was 4.3 mm (SE 1.4) smaller in the BCLA group (p = 0.002). In the BCLP group, a comparable pattern was found. At the canine level, mean transversal width was 7.2 mm (SE 1.9) smaller compared to the control group, but no significant differences were found in the other transversal dimensions. CONCLUSIONS Small differences are found in transversal dimensions in patients with BCLA and BCLP compared to a control group. Differences are most outspoken in the area near the cleft.

Width and elevation of the palatal shelves in unoperated unilateral and bilateral cleft lip and palate patients in the permanent dentition.

Patients with cleft left lip and palate (CLP) normally require extensive surgery from an early age up to the end of adolescence. These surgeries affect the growth of the maxillofacial complex. The degree to which the cleft itself affects growth of the maxillofacial complex remains poorly understood. By analysing the width and elevation of the palatal shelves in unoperated adolescents and adults with unilateral and bilateral cleft lip and palate (UCLP and BCLP, respectively) and a non‐cleft control group, it is possible to gain more insight into the real intrinsic growth potential of the maxillary structures. In this study, dental casts of the full permanent dentition of individuals with unrepaired UCLP (n = 68) and BCLP (n = 13) and non‐cleft controls (n = 24) from the same area of Indonesia were digitized three‐dimensionally. Maxillary arch width in the canine, premolar and molar regions, and the width and elevation of the palatal shelves were measured. Results showed that in patients with UCLP, the width of the palatal shelves on the cleft side in all regions, and on the non‐cleft side in the canine/first premolar region, was significantly smaller compared with the control group. BCLP subjects showed similar deviations. In the UCLP group, the palatal shelves were rotated cranially and positioned more vertically. In the BCLP group, the palatal shelves were inclined by almost 10 ° more than the control group. The width of the palatal shelf and width of the maxillary arch positively correlated in the canine and first premolar regions for both the cleft and non‐cleft side in patients with UCLP, and in the canine region for patients with BCLP. This means that the wider the palatal shelf, the wider the maxillary arch. The elevation of palatal shelves correlated with the maxillary arch width in all regions in patients with UCLP, and only in the premolar region in the control group. Thus, the wider the arch width, the smaller the elevation angle (the maxillary shelves are less vertical). No correlations between palatal shelf elevation and maxillary arch width were found in the BCLP group. This shows that the intrinsic growth potential in patients with UCLP and BCLP is affected by a smaller palatal shelf width and larger elevation of the shelves. These deviations may result in a wider cleft.