The Severity of Initial Unilateral Cleft Lip and Nasal Deformity Predicts Long-Term Postoperative Esthetic Outcome.

BACKGROUND: Infantile cleft lip and nasal severity influence the final esthetic result of the repair. Although various authors have described methods of cleft lip and nasal repair, there is a paucity of data that correlates cleft severity with esthetic outcomes. The aim of this study was to examine the correlation between presurgical severity of unilateral cleft deformity and long-term postoperative esthetic outcomes. METHODS: This retrospective study, based at a single institution, investigated patients with complete unilateral cleft lip, with or without cleft palate, who underwent repair by a single surgeon, had preoperative infantile facial casts, and had postoperative facial photographs at 6 to 11 years of age (N=31). Preoperative nostril width ratio and columellar angle measurements were taken from facial casts. Postoperative, long-term nasolabial appearance was rated by 5 blinded observers used a modified Kuijpers-Jagtman scale. Linear regression was used to determine the relationship between preoperative cleft severity and postoperative ratings. RESULTS: Preoperative nostril width ratio directly correlated with postoperative nasal form score (r=0.40; P=0.026); likewise, preoperative columellar angle predicted postoperative nasal form score (r=0.37; P=0.040). Preoperative cleft severity was not significantly correlated with vermillion border appearance. Cronbach α values of 0.91 (nasal form) and 0.79 (vermillion border) indicated good inter-rater reliability. Kappa values of 0.87 (nasal form) and 0.70 (vermillion border) indicated good intrarater reliability. CONCLUSIONS: Preoperative unilateral cleft nose severity directly correlates with long-term postoperative nasal appearance in childhood. Outcome studies should present and control for preoperative severity to allow more accurate assessment of repair techniques.

A Longitudinal Investigation of Nasolabial Changes With and Without Revision Surgery in Patients with Non-Syndromic Unilateral Cleft Lip and Palate.

OBJECTIVE: To determine a baseline of anticipated change in nasolabial appearance following primary repair of unilateral cleft lip/palate and evaluate the degree to which revision surgery improves nasolabial appearance. DESIGN: Retrospective chart review. SETTING: Patients treated at the Lancaster Cleft Palate Clinic interdisciplinary clinic. PATIENTS: Twenty-three patients with complete unilateral cleft lip and palate who underwent primary surgical repair and 19 additional patients who underwent subsequent revision surgery were included. INTERVENTIONS: Patients in the non-revision group underwent a Tennison-Randall triangular flap lip repair at 3mo. Patients in the revision group underwent a modification of the Nakajima straight-line repair after primary Tennison-Randall triangular flap lip repair at an average age of 141mo. MAIN OUTCOME MEASURES: A modification of the Asher-McDade Aesthetic Index was utilized to evaluate Nasolabial Frontal (NLF), Nasolabial Profile (NLP), Vermillion Border (VB), and total change in appearance. Scores for patients in the revision group were evaluated before and after revision while appearance for patients without revision was evaluated at 3 distinct ages. Scores were averaged across time-points and inter-rater reliability was assessed. RESULTS: Nasolabial appearance in the non-revision sample did not change significantly over time, except for nasal profile. Scores improved after revision surgery - NLP: 3.48 to 2.97, (p = 0.001); NLF: 3.50 to 2.95 (p = 0.001); and Total Nasolabial Score: 3.29 to 3.01 (p = 0.004), with no significant change in VB. CONCLUSION: Decisions regarding need for nasolabial revision surgery may be made as early as 5yo with successful outcomes following secondary surgery improving appearance except for vermillion border appearance.

Genetic Analyses of Enamel Hypoplasia in Multiethnic Cohorts.

Enamel hypoplasia causes reduction in the thickness of affected enamel and is one of the most common dental anomalies. This defect is caused by environmental and/or genetic factors that interfere with tooth formation, emphasizing the importance of investigating enamel hypoplasia on an epidemiological and genetic level. A genome-wide association of enamel hypoplasia was performed in multiple cohorts, overall comprising 7,159 individuals ranging in age from 7-82 years. Mixed-models were used to test for genetic association while simultaneously accounting for relatedness and genetic population structure. Meta-analysis was then performed. More than 5 million single-nucleotide polymorphisms were tested in individual cohorts. Analyses of the individual cohorts and meta-analysis identified association signals close to genome-wide significance (P < 510-8), and many suggestive association signals (510-8 < P < 510-6) near genes with plausible roles in tooth/enamel development. The strongest association signal (P = 1.5710-9) was observed near BMP2K in one of the individual cohorts. Additional suggestive signals were observed near genes with plausible roles in tooth development in the meta-analysis, such as SLC4A4 which can influence enamel hypoplasia. Additional human genetic studies are needed to replicate these results and functional studies in model systems are needed to validate our findings.

Early Secondary Alveolar Bone Grafting and Facial Growth of Patients with Complete Unilateral Cleft Lip and Palate.

OBJECTIVE: To investigate the craniofacial growth outcomes of early secondary alveolar bone grafting(ABG) around 6 years of age. DESIGN: Retrospective cohort study. SETTING: 1 North-American and 5 Northern-European cleft centers. SUBJECTS: 33 subjects with CUCLP consecutively treated with secondary ABG around 6 years of age were compared to 105 subjects from 4 centers treated with late secondary ABG and 19 subjects from 1 center with primary ABG. METHODS: Preorthodontic standardized lateral cephalometric radiographs taken after 12 years of age were traced and analyzed according to the Eurocleft Study protocol. Fourteen angular and two proportional measurements were performed. Measurement means from the Study Center(SC) were compared to 5 Northern-European centers using analysis of variance and Welch's modified t-tests, and P < .05 was considered statistically significant. RESULTS: For the SC, the mean age ± SD at the time of bone graft was 5.85 ± 0.71 years and the mean age at the time of the lateral cephalogram was 13.4 ± 1.8 years. The sagittal maxillary prominence of the SC was favorably comparable to the 5 Northern-European centers. The mean SNA (78.1 ± 4.3) for the SC was significantly higher compared to 4 of the 5 Northern-European centers(all P < .05), and the mean ANB angle was comparable to 4 of the 5 centers. Similarly, the mean soft tissue ANB angle was not significantly different to the 5 centers. The soft tissue vertical proportions compared favorably to all 5 Northern-European centers(all P < .01). CONCLUSIONS: Craniofacial growth outcomes of early secondary ABG around 6 years compare favorably to the outcomes of late secondary ABG.

Evaluating SWAG and Its Validity When Compared to 3D Imagery of Secondarily Grafted Cleft Sites.

OBJECTIVE: To test validity of 2D Standardized Way to Assess Grafts (SWAG) ratings to assess 3D outcomes of bone grafting (ABG). PATIENTS: 43 patients (34 UCLP, 9 BCLP) with non-syndromic complete clefts, bone-grafted at mean age 9yrs/3mos, with available post-graft occlusal radiographs and cone beam computed tomography (CBCT) (taken mean 4yrs/9mos post-ABG). MAIN OUTCOME MEASURES: 2D occlusal radiographs rated twice using SWAG by 6 calibrated raters. 12 scores were averaged and converted to a percentage reflecting bone-fill. Weighted Kappas were assessed for SWAG reliability. 3D cleft-site bone volume was calculated by 1 rater using ITK-SNAP. 13 cleft sites were re-measured by the 'one rater' for 3D reliability using Intraclass Correlation Coefficient (ICC). 2D versus 3D ratings were compared using paired t-test, independent samples t-test, Bland-Altman and Linear Regression. Significance level was P = .5. RESULTS: 2D reliability was 0.724 (intra-rater) and 0.546 (inter-rater). 3D reliability was 0.986. Bland-Altman plot comparing 2D vs 3D showed for 45 of 47 graft-sites were within 2 SD's. Mean % bone-fill was 64.11% with 2D and 69.06% with 3D (mean difference = 4.95%) that was a non-significant difference in both t-tests. Regression showed a statistically significant relation between the two methods (r2 = 0.46; P = .0001). CONCLUSION: 2D SWAG systematically and non-significantly underestimated bone-fill. There was a significant correlation between 2D/3D methods. Bland-Altman analysis illustrated the similarity of the two methods. For comparisons of group (cleft treatment Centers') bone grafting outcomes, the 2D method may suffice as a proxy for the 3D method. However, with individual variation up to 40% in 2D estimates of actual 3D volume, 2D SWAG method cannot be used in place of 3D images.

Parents of Children With Nonsyndromic Orofacial Clefting Show Altered Palate Shape.

OBJECTIVE: The unaffected relatives of individuals with nonsyndromic orofacial clefts have been shown to exhibit subtle craniofacial differences compared with the general population. Here, we investigate whether these morphological differences extend to the shape of the palate. DESIGN: We conducted a geometric morphometric analysis to compare palate shape in the clinically unaffected parents of children with nonsyndromic cleft lip with or without cleft palate and adult controls of European, Asian, and African ancestry. We conducted pairwise group comparisons using canonical variates analysis, and then confirmed and characterized findings of shape differences using Euclidean distance matrix analysis. RESULTS: Significant differences in palate shape were detected in unaffected mothers (but not fathers) compared to demographically matched controls. The differences in shape were ancestry-specific; mothers of Asian-derived and African-derived ancestry showed wider and shorter palates with higher posterior palatal vaults, while mothers of European-derived ancestry showed narrower palates with higher anterior palatal vaults. CONCLUSIONS: Our findings suggest that altered palate shape is a subclinical phenotypic feature, which may be indicative of elevated orofacial cleft risk. The risk phenotype varied by sex and ancestry, suggesting possible etiologic heterogeneity among demographic groups. Understanding the genetic basis of these informative palate shape traits may reveal new genes and pathways relevant to nonsyndromic orofacial clefting.

The Effect of Earlier Bone Grafting, Prior to Orthodontic Treatment, on SWAG Ratings of Graft Outcomes.

OBJECTIVE: To compare the outcomes between 2 groups of patients with complete clefts treated with early secondary alveolar bone grafting (ABG) at 2 centers (5-7 years, before orthodontic intervention) and to a third group of patients treated at one of those centers (center 1) who had received later secondary ABG (8-10 years, after orthodontic intervention). DESIGN: Blind retrospective analysis of cleft site radiographs using Americleft Standardized Way to Assess Grafts (SWAG) scale. PATIENTS: A total of 99 patients with complete clefts from 2 North American cleft/craniofacial centers. INTERVENTIONS: Secondary ABG representing 2 protocols: early grafting at a mean age of 6.6 years prior to any orthodontic intervention, and later grafting at a mean age of 10.2 years following pregrafting orthodontic intervention. MAIN OUTCOME MEASURES: Using occlusal radiographs, the SWAG scale from 0 (failed graft) to 6 (ideal) was used. Six trained, calibrated raters scored each radiograph twice, with the average of the 2 ratings used as the final score. Reliability was assessed using the weighted κ statistic. The significance of differences between groups was determined using the Kruskal-Wallis test and Dunn test for pairwise comparisons. RESULTS: Inter-rater reliability of SWAG method was good (0.631). Intra-rater reliability was excellent (0.817). There was a tendency for improved total graft outcome in the early grafted group from center 1 compared to the later grafted group with improvement being significantly different in only the coronal third of the early, preorthodontic grafted group. However, the difference was not statistically significant for the graft overall.

Genome-Wide Association Study (GWAS) of dental caries in diverse populations.

BACKGROUND: Dental caries is one of the most common chronic diseases and is influenced by a complex interplay of genetic and environmental factors. Most previous genetic studies of caries have focused on identifying genes that contribute to dental caries in specific ethnic groups, usually of European descent. METHODS: The aim of this study is to conduct a genome-wide association study (GWAS) to identify associations affecting susceptibility to caries in a large multiethnic population from Argentina, the Philippines, Guatemala, Hungary, and the USA, originally recruited for studies of orofacial clefts (POFC, N = 3686). Ages of the participants ranged from 2 to 12 years for analysis of the primary dentition, and 18-60 years for analysis of the permanent dentition. For each participant, dental caries was assessed by counts of decayed and filled teeth (dft/DFT) and genetic variants (single nucleotide polymorphisms, SNPs) were genotyped or imputed across the entire genome. Caries was analyzed separately for the primary and permanent dentitions, with age, gender, and presence/absence of any type of OFC treated as covariates. Efficient Mixed-Model Association eXpedited (EMMAX) was used to test genetic association, while simultaneously accounting for relatedness and stratification. RESULTS: We identified several suggestive loci (5 × 10-8 < P < 5 × 10-6) within or near genes with plausible biological roles for dental caries, including a cluster of taste receptor genes (TAS2R38, TAS2R3, TAS2R4, TASR25) on chromosome 7 for the permanent dentition analysis, and DLX3 and DLX4 on chromosome 17 for the primary dentition analysis. Genome-wide significant results were seen with SNPs in the primary dentition only; however, none of the identified genes near these variants have known roles in cariogenesis. CONCLUSION: The results of this study warrant further investigation and may lead to a better understanding of cariogenesis in diverse populations, and help to improve dental caries prediction, prevention, and/or treatment in future.

Patient-Centered Satisfaction After Secondary Correction of the Cleft Lip and Nasal Defect.

OBJECTIVE: To evaluate patient-reported aesthetic and psychosocial outcomes of secondary cleft lip and nose revision procedures. DESIGN: Single-center cohort study. SETTING: Tertiary care pediatric hospital. PATIENTS/PARTICIPANTS: Patients who underwent secondary revision procedures for cleft lip and nasal defect (N = 42). INTERVENTIONS: Patients were administered a survey during a routine postoperative clinic visit. MAIN OUTCOME MEASURE(S): Outcome measures were planned prior to data collection and included improvement in appearance seen in lip and nose, satisfaction with revision procedure, changes in self-confidence, likelihood to participate in social activities, and effect on teasing by peers. RESULTS: Patients agreed that an improvement was seen in the appearance of their lip (1.93) and nose (1.98) following surgery. Overall, patients felt satisfied with the results of their revision procedure (1.76). An improvement in confidence and decrease in feelings of self-consciousness was reported. Patients were teased less by their peers and more likely to participate in social activities. CONCLUSIONS: Secondary revision procedures of the cleft lip and nasal defect provide a patient-reported improvement in appearance and a positive psychosocial impact on patient's lives.

Facial Growth of Patients With Complete Unilateral Cleft Lip and Palate Treated With Alveolar Bone Grafting at 6 Years.

OBJECTIVE: To investigate the effect that alveolar bone grafting (ABG) around 6 years of age has on facial growth by assessing craniofacial growth outcomes. DESIGN: Retrospective cohort study. SETTING: North American cleft centers. PARTICIPANTS: A total of 33 children with complete unilateral cleft lip and palate who were consecutively treated with secondary ABG around 6 years of age were compared to 148 participants from 4 centers with late secondary ABG. METHODS: Preorthodontic standardized lateral cephalometric radiographs were analyzed and traced according to the Americleft Study protocol. Sixteen angular and 2 proportional measurements were performed. The outcomes of all ABG were assessed using the Standardized Way to Assess Graft scale. Measurement means from the study center (SC) were compared to 4 North American centers using analysis of variance and Welch modified t tests, and P < .05 was considered statistically significant. RESULTS: For the SC, the mean age (SD) at the time of bone graft was 5.85 (0.71) years and the mean age at the time of the lateral cephalogram was 13.4 (1.8) years. The sagittal maxillary prominence of the SC was comparable to the 4 other centers. The mean SNA (78.1 [4.3]) for the SC was significantly higher compared to one center that used primary bone grafting ( P = .03). The soft tissue mean ANB (3.52 [4.09]) for the SC was significantly lower compared to 3 of the centers. CONCLUSIONS: Early secondary ABG around 6 years of age did not result in reduced midface projection as assessed by SNA and thus did not compromise anterior maxillary growth.

The Americleft Project: Comparison of Ratings Using Two-Dimensional Versus Three-Dimensional Images for Evaluation of Nasolabial Appearance in Patients With Unilateral Cleft Lip and Palate.

This study was conducted to determine if nasolabial appearance is rated with comparable results and reliability on 3-dimensional stereophotogrammetric facial images versus standard clinical photographs (2-dimensional). Twenty-seven consecutively treated patients with repaired complete unilateral cleft lip and palate were selected. Six trained and calibrated raters assessed cropped 2- and 3-dimensional facial images. Nasolabial profile, nasolabial frontal, and vermillion border esthetics were rated with the 5-point scale described by Asher-McDade using the modified Q-sort method. Cropped 3-dimensional images were available for viewing by each rater, allowing for complete rotational control for viewing the images from all aspects. Two- and three-dimensional ratings were done separately and repeated the next day.Interrater reliability scores were good for 2-dimensional (κ = 0.607-0.710) and fair to good for 3-dimensional imaging (κ = 0.374-0.769). Intrarater reliability was good to very good for 2-dimensional (κ = 0.749-0.836) and moderate to good for 3-dimensional imaging (κ = 0.554-0.855). Bland-Altman analysis showed satisfactory agreement of 2- and 3-dimensional scores for nasolabial profile and nasolabial frontal, but more systematic error occurred in the assessment of vermillion border.Although 3-dimensional images may be perceived as more representative of a direct clinical facial evaluation, their use for subjective rating of nasolabial aesthetics was not more reliable than 2-dimensional images in this study. Conventional 2-dimensional images provide acceptable reliability while being readily accessible for most cleft palate centers.

An Intercenter Comparison of Dental Arch Relationships and Craniofacial Form Including a Center Using Nasoalveolar Molding.

OBJECTIVE: To compare dental arch relationship and craniofacial morphology of patients with CUCLP in pre-adolescence from five cleft centers including a center using NAM. DESIGN: Retrospective cohort study. SETTING: Five cleft centers in North America. PATIENTS: One hundred eighty-two subjects with repaired CUCLP from the five cleft centers participated in the craniofacial form study. One hundred forty-eight subjects from four of the five centers participated in the dental arch relationship study. METHODS: Digital dental models were assessed using the GOSLON Yardstick. Eighteen cephalometric measurements were performed. Measurement means, by center, were compared. Analysis of variance and Tukey-Kramer analysis were used to compare GOSLON scores and cephalometric measurements. RESULTS: The center that performed neither PSOT (including NAM) nor primary bone grafting exhibited the most favorable mean GOSLON score. The same center also showed the highest mean SNA, ANB, and ANS-N-Pg angles. However, the mean ANB and ANS-N-Pg angles were not significantly different from those of the center using NAM. No statistically significant differences were seen for mandibular prominence, vertical dimensions, or dental inclinations. The center with NAM also showed a significantly smaller nasoform angle than two of the four other centers. CONCLUSION: The centers that used NAM and other forms of PSOT did not have better dental arch relationships or craniofacial morphology compared with the centers that performed only primary lip repair. However, this study was not designed to investigate the cause-and-effect relationship between specific outcomes and particular features of those protocols.

An Intercenter Comparison of Nasolabial Appearance Including a Center Using Nasoalveolar Molding.

OBJECTIVE: To compare nasolabial appearance outcomes of patients with complete unilateral cleft lip and palate (CUCLP) in preadolescence from 4 cleft centers including a center using nasoalveolar molding (NAM) and primary nasal reconstruction. DESIGN: Retrospective cohort study. SETTING: Four cleft centers in North America. PATIENTS: 135 subjects with repaired CUCLP. METHODS: Frontal and profile facial pictures were assessed using the Asher-McDade rating scale. Intra- and interrater reliability were tested using weighted Kappa statistics. Median scores by center were compared with Kruskal-Wallis statistics. RESULTS: Intrarater reliability scores were moderate to good. Interrater reliability scores were moderate. Significant differences ( P < .05) among centers were found. For nasal form, center G (median = 2.83) had better scores than centers C and D (C median = 3.33, D median = 3.17). For nose symmetry, center G had better scores (median = 2.33) than all other centers (B median = 2.67, C median = 2.83, D median = 2.83). For vermillion border, center G had better scores (median = 2.58) than centers B and C (B median = 3.17, C median = 3.17). For nasolabial profile, center G (median score = 2.67) had better scores than center C (median = 3.00). For total nasolabial score, center G (median = 2.67) had better scores than all other centers (B median = 2.83, C median = 3, D median = 2.83). CONCLUSION: The protocol followed by center G, the only center that performed NAM and primary nasal reconstruction, produced better results in all categories when compared to center C, the only center that did not perform presurgical orthopedics or lip/nose revisions. When compared to centers that performed traditional presurgical orthopedics and surgical revisions (B and D), center G was not consistently better in all categories. As with other uncontrolled, retrospective intercenter studies, it is not possible to attribute the outcomes to a specific protocol component.

Active Presurgical Infant Orthopedics for Unilateral Cleft Lip and Palate: Intercenter Outcome Comparison of Latham, Modified McNeil, and Nasoalveolar Molding.

OBJECTIVE: To compare dental arch relationship, craniofacial form, and nasolabial aesthetic outcomes among cleft centers using distinct methods of presurgical infant orthopedics (PSIO). DESIGN: Retrospective cohort study. SETTING: Four cleft centers in North America. PATIENTS: One hundred ninety-one children with repaired complete unilateral cleft lip and palate (CUCLP). MAIN OUTCOME MEASURES: Dental arch relationship was assessed using the GOSLON Yardstick. Craniofacial form was assessed by 12 cephalometric measurements. Nasolabial aesthetics were assessed using the Asher-McDade system. Assessments were performed between 6 and 12 years of age. RESULTS: The center that used no PSIO achieved the most favorable dental arch relationship and maxillomandibular relationship, with a median GOSLON score of 2.3 ( P < .01) and an ANB angle of 5.1° ( P < .05). The proportion of children assigned a GOSLON score of 4 or 5, predictive of the need for orthognathic surgery in adolescence, was 16% at the center that used no PSIO and no secondary surgery, compared to 76% at the centers that used the Latham appliance and early secondary lip and nose surgery ( P < .01). The center that used no PSIO and no secondary surgery achieved significantly less favorable nasolabial aesthetic outcomes than the centers using Latham appliance or nasoalveolar molding (NAM) ( P < .01). CONCLUSIONS: Effects of active PSIO are multifaceted and intertwined with use of revision surgery. In our study, centers using either the Latham appliance combined with early revision surgery or the NAM appliance without revision surgery achieved better nasolabial aesthetic outcomes but worse maxillary growth, compared to a center using no PSIO and secondary surgery.

Burden of Care of Various Infant Orthopedic Protocols for Improvement of Nasolabial Esthetics in Patients With CUCLP.

OBJECTIVES: 1. To evaluate the orthodontic burden of care of nasoalveolar molding (NAM) and modified McNeil for the treatment of patients with complete unilateral cleft lip and palate (CUCLP). 2. To compare the esthetic outcomes of each with those of centers not utilizing infant orthopedics (IO). DESIGN: Retrospective cohort study. SETTING: Institutional. PARTICIPANTS: Four cohorts with repaired CUCLP (n = 149) from 3 centers. INTERVENTIONS: Two cohorts were treated in the same center and had either traditional infant orthopedics (TIO) or NAM and 2 were treated in centers not employing IO. MAIN OUTCOME MEASURES: Burden of care data for the IO groups were compared using t tests. Frontal and profile photographs at approximately age 5 were collected for ratings of nasolabial esthetics, using a modification of the Asher-McDade method. Intrarater and interrater reliabilities were determined using weighted κ statistics. Median ratings were compared using a Kruskal-Wallis test. RESULTS: The burden of care of NAM was significantly greater than TIO for both the number of visits (9.9 vs 6.6, [ P < .001]); and treatment duration (127 vs 112 days, [ P < .05]). Significant differences in nasolabial esthetic ratings were noted among the 3 centers. No significant differences were observed in the nasolabial esthetic outcomes between the NAM and TIO groups. CONCLUSIONS: 1. NAM required more visits and longer overall duration compared with TIO. 2. The center employing IO showed favorable nasolabial esthetics compared to those not utilizing IO. 3. No significant differences were found in the nasolabial esthetics of patients who have received NAM compared with TIO.

The Americleft Project: Plaster Dental Casts Versus Digital Images for GOSLON Yardstick Ratings When Used in Intercenter Comparisons.

The purpose of this investigation was to determine reliability and validity of GOSLON Yardstick ratings using plaster casts versus photo galleries of digital images in actual intercenter comparisons. The dental arch relationships of 112 patients with complete unilateral cleft lip and palate from 3 North American cleft/craniofacial centers were rated in 2 separate studies. In the first, plaster casts were used. For a later intercenter comparison, the same dental casts were scanned, digital bases added, and two-dimensional photographic galleries (6 views) were created for each set of casts. Three raters experienced with the GOSLON Yardstick carried out 2 separate ratings of the plaster casts in the first study, then of the photographic gallery of scanned digital images of the same casts in the second study. Inter- and intrarater reliabilities were calculated using the Weighted Kappa statistic. Average scores for each patient were calculated and compared between methods with correlation statistics and a Bland-Altman plot. Kruskal-Wallis test was used to compare results between centers using both media. Reliability using both methods was very good and comparable between methods. Mean weighted Kappas were: inter-rater = 0.815 (plaster) versus 0.891 (photo); and intrarater = 0.866 (plaster) versus 0.891 (photo). There was a highly significant correlation (r = 0.920). Mean difference between centers was 0.033 of a GOSLON category. The level of significance of the differences found between centers with both methods was identical, confirming the interchangeability of both media presentations.

The Americleft Project: A Modification of Asher-McDade Method for Rating Nasolabial Esthetics in Patients With Unilateral Cleft Lip and Palate Using Q-sort.

The purpose of this study was to investigate ways to improve rater reliability and satisfaction in nasolabial esthetic evaluations of patients with complete unilateral cleft lip and palate (UCLP), by modifying the Asher-McDade method with use of Q-sort methodology. Blinded ratings of cropped photographs of one hundred forty-nine 5- to 7-year-old consecutively treated patients with complete UCLP from 4 different centers were used in a rating of frontal and profile nasolabial esthetic outcomes by 6 judges involved in the Americleft Project's intercenter outcome comparisons. Four judges rated in previous studies using the original Asher-McDade approach. For the Q-sort modification, rather than projection of images, each judge had cards with frontal and profile photographs of each patient and rated them on a scale of 1 to 5 for vermillion border, nasolabial frontal, and profile, using the Q-sort method with placement of cards into categories 1 to 5. Inter- and intrarater reliabilities were calculated using the Weighted Kappa (95% confidence interval). For 4 raters, the reliabilities were compared with those in previous studies. There was no significant improvement in inter-rater reliabilities using the new method. Intrarater reliability consistently improved. All raters preferred the Q-sort method with rating cards rather than a PowerPoint of photos, which improved internal consistency in rating compared to previous studies using the original Asher-McDade method. All raters preferred this method because of the ability to continuously compare photos and adjust relative ratings between patients.

Structural Fat Grafting to Improve Reconstructive Outcomes in Secondary Cleft Lip Deformity.

OBJECTIVE: To describe the technique and results of structural fat grafting in cleft lip revision, including patient satisfaction and aesthetic outcome. DESIGN: Retrospective case series. SETTING: Multidisciplinary cleft care center. PATIENTS: All patients who underwent structural fat grafting between June 2006 and September 2012 for cleft lip revision, with appropriate photographic follow-up included. Twenty-two cases were reviewed; 18 had sufficient data to be included. INTERVENTIONS: Patients underwent structural fat grafting for cleft lip revision, most commonly injecting fat under deficient philtral columns, the nostril base, and upper lip. MAIN OUTCOME MEASURES: Blinded observers rated outcomes using the Asher-McDade nasolabial appearance rating scale. Patients completed questionnaires assessing their satisfaction. A paired Student's t-test was used to test outcomes for significance (alpha = 0.05). RESULTS: Patients were an average of 16 years old (range: 6-43); average length of follow up was 11.7 months. Overall symmetry and aesthetics were improved based on the nasal form (P = 0.006) and vermillion border (P = 0.04) when rated using the Asher-McDade scale. No complications were recorded. Patients were significantly happier with their appearance after fat grafting (P < 0.001) and were uniformly positive when questioned about the ease of the surgery and rate of recovery. CONCLUSIONS: Structural fat grafting is a safe and effective way to improve symmetry and enhance facial proportions in patients with cleft lip. Given the high degree of patient satisfaction, few complications, and durable results, fat grafting offers many advantages in cleft lip revision.

Reliability of the SWAG-The Standardized Way to Assess Grafts Method for Alveolar Bone Grafting in Patients With Cleft Lip and Palate.

OBJECTIVE: The objective of this study was to test a new method, a Standardized Way to Assess Grafts (SWAG), to rate alveolar bone graft (ABG) outcomes for patients with cleft lip and palate. DESIGN: This was a retrospective comparison using the SWAG scale. SETTING: This study took place in four cleft palate centers with different treatment protocols. METHODS: A total of 160 maxillary occlusal radiographs taken 3 to 18 months post-ABG for sequentially treated patients with cleft lip and palate were assessed using the SWAG scale. Radiographs were scanned, standardized, blinded, and rated by 6 calibrated orthodontists to assess vertical thirds, bony root coverage, and complete bony fill. All radiographs were rated twice, 24 hours apart, by the same raters. MAIN OUTCOMES: Intra- and interrater reliabilities were assessed. RESULTS: Intrarater reliability was good to very good (.760; .652-.834), and interrater reliability was moderate to good (.606; .569-.681), comparable to previously published methods. CONCLUSIONS: Rater reliabilities were shown to be comparable to or better than existing methods. The SWAG method was validated for ABG assessments in the mixed and permanent dentitions based on reliabilities in an intercenter outcome comparison.