Kathleen Russell

The Americleft Study: An Inter‐Center Study of Treatment Outcomes for Patients With Unilateral Cleft Lip and Palate Part 4. Nasolabial Aesthetics

Objective To compare the nasolabial aesthetics for individuals with nonsyndromic complete unilateral cleft lip and palate between the ages of 5 and 12 years. Design Retrospective cross-sectional study. Setting Four cleft centers in North America. Subjects A total of 124 subjects with repaired complete unilateral cleft lip and palate who were treated at the four centers. Methods After ethics approval was obtained, 124 preorthodontic frontal and profile patient images were scanned, cropped to show the nose and upper lip, and coded. Using the coded images, four nasolabial features that reflect aesthetics (i.e., nasal symmetry, nasal form, vermilion border, and nasolabial profile) were rated by five examiners using the rating system reported by Asher-McDade et al. (1991). Intrarater and interrater reliabilities were determined using weighted kappa statistics. Mean ratings, by center, were compared using analysis of variance. Results Intrarater reliability scores were good to very good and interrater reliability scores were moderate to good. Total nasolabial scores were Center B = 2.98, Center C = 3.02, Center D = 2.80, and Center E = 2.87. No statistically significant differences among centers were detected for both total aesthetic scores and for any of the individual aesthetic components. Conclusion There were no significant differences in nasolabial aesthetics among the centers evaluated. Overall good to fair nasolabial aesthetic results were achieved using the different treatment protocols in the four North American centers.

The Americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palate. Part 2. Dental arch relationships.

Objective To compare maxillomandibular relationships for individuals with nonsyndromic complete unilateral cleft lip and palate using the Goslon Yardstick for dental models. Design Retrospective cohort study. Setting Five cleft palate centers in North America. Subjects A total of 169 subjects with repaired complete unilateral cleft lip and palate who were consecutively treated at the five centers. Methods Ethics approval was obtained. A total of 169 dental models of patients between 6 and 12 years old with complete unilateral cleft lip and palate were assessed using the Goslon Yardstick. Weighted kappa statistics were used to assess intrarater and interrater reliabilities; whereas, analysis of variance and Tukey-Kramer analysis was used to compare the Goslon scores. Significance levels were set at p < .05. Results Intrarater and interrater reliabilities were very good for model ratings. One center that incorporated primary alveolar bone grafting showed especially poor Goslon scores that were significantly poorer than the remaining centers. The surgery protocols used by the other four centers did not include primary alveolar bone grafting but involved a number of different lip and palate closure techniques. Using the Goslon Yardstick assumptions, the center with the best scores would be expected to require end-stage maxillary advancement orthognathic surgery in 20% of its patients; whereas, the center with the worst scores would be likely to require this surgery in 66% of its patients. Conclusions The Goslon Yardstick proved capable of discriminating among the centers’ dental arch relationships. Possible explanations for the differences are discussed.

The Americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 1. Principles and study design

Objective The Americleft study is a North American initiative to undertake an intercenter outcome study for patients with repaired complete unilateral cleft lip and palate from five well-established North American cleft centers. Design Retrospective cohort study. Setting Five cleft palate centers in North America. Methods This is the first paper in a series of five that outlines the overall goals of the study and sets the basis for the clinical outcome studies that are reported in the following four papers. The five centers’ samples and treatment protocols as well as the methods used for each study are reported. The challenges encountered and possible mechanisms to resolve them and reduce methodological error with intercenter studies are also reviewed.

The americleft study: An inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 3. Analysis of craniofacial form

Objective To compare craniofacial morphology for individuals with nonsyndromic complete unilateral cleft lip and palate between the ages of 6 and 12 years. Design Retrospective cohort study. Setting Four North American cleft palate centers. Subjects A total of 148 subjects with repaired complete unilateral cleft lip and palate who were consecutively treated at the four centers. Methods The 148 preorthodontic lateral cephalometric radiographs were scanned, scaled, digitized, and coded to blind the examiners to radiograph origin. On each radiograph, 18 (angular and ratio) cephalometric measurements were performed. Measurement means, by center, were compared using analysis of variance and Tukey-Kramer analysis. Results Significant differences were found for sagittal maxillary prominence among the four centers. The most significant difference was seen between Center B (lowest SNA) and Center C (highest SNA). Similar differences were seen at the soft tissue level, with Center C showing a significantly larger ANB angle compared with Centers B and D. Center C was also shown to have statistically greater mean soft tissue convexity than Centers B, D, and E. The mean nasolabial angle in Center B was significantly more acute than in Centers C, D, and E. No statistically significant differences were seen for mandibular prominence, vertical dimensions, or dental inclinations. Conclusion Significant differences were seen among the centers for hard and soft tissue maxillary prominence, but not for mandibular prominence, vertical dimensions, or dental inclinations. A modest but statistically significant (p < .001) negative correlation was found between Goslon scores and ANB angle (r = –.607).

The Americleft Study: An Inter‐Center Study of Treatment Outcomes for Patients With Unilateral Cleft Lip and Palate Part 5. General Discussion and Conclusions

Objective To summarize the Americleft study regarding treatment outcomes for patients with complete unilateral cleft lip and palate (CUCLP). Setting Five cleft palate centers in North America. Subjects One hundred sixty-nine subjects, between the ages of 6 years and 12 years, with repaired CUCLP who were consecutively treated at the five centers. Methods Study consisted of model comparisons assessing maxillomandibular relationship using the GOSLON Yardstick (169 patients from all 5 centers), soft and hard tissue craniofacial morphologic comparisons using lateral cephalometric analyses (148 patients from four of the centers), and nasolabial esthetics assessments (125 patients from four of the centers). Results Significant differences were found between the center with the best GOSLON scores and the remaining centers. These differences also corresponded to those found in the craniofacial morphologic cephalometric assessment. Sagittal maxillary prominence was found to be significantly better for the center with the best GOSLON scores, while no significant differences were seen among the centers for mandibular prominence, vertical dimensions, or dental inclinations. No differences were seen for nasolabial esthetics between the centers. Conclusions Challenges experienced while undertaking the inter-center retrospective study are reviewed. Aspects of treatment that could potentially make the outcome of treatment less optimal included primary alveolar bone grafting and extensive treatment protocols. Differences in the outcomes identified between the centers were restricted to the maxilla, and no differences were identified for mandibular prominence, vertical dimensions, or dental inclinations.

Canine Eruption in Patients With Complete Cleft Lip and Palate

Objective: To describe erupting maxillary canine positions in patients with bone-grafted alveolar clefts. Sample: The sample consisted of 101 cleft sites from patients with complete unilateral or bilateral cleft lip and palate who had early (≤9 years) or late (>9 years) secondary alveolar bone grafts. Methods: Canine position was assessed using panoramic radiographs taken before and after alveolar bone grafts. Vertical canine positions were assessed using the long axis of the maxillary permanent canine relative to a 90° vertical reference line. Lateral canine positions were defined using the relationship between the canine tip and the midplane of the lateral incisor root. Anomalous lateral incisors were recorded. Statistical analysis included Students t tests and chi-square tests. Results: Patients with alveolar clefts had a 20-fold increased risk for canine impaction, based on erupting canine positions. Abnormal vertical canine positions decreased following early and late alveolar bone grafts (p < .05), whereas abnormal lateral canine positions increased following late alveolar bone grafts (p < .01). Of the cleft sites with altered canine positions, 61% also had a lateral incisor anomaly. Based on canine position, the non–cleft-side canine had the same risk for impaction as the cleft-side canine. Conclusions: Patients with alveolar clefts have a significantly higher risk for canine impaction compared with patients without clefts. Timing of alveolar bone grafts and lateral incisor anomalies influenced the risk for canine impaction. An alveolar bone graft should be planned in accordance with maxillofacial development, including the eruption of teeth adjacent to the cleft.

A population-based evaluation of antenatal diagnosis of orofacial clefts.

Objectives: To evaluate the changes in prevalence and antenatal detection of cleft lip with or without cleft palate and isolated cleft palate and to describe the association between anomalies and rates of antenatal diagnosis in Nova Scotia from 1992 to 2002. Design: This population-based cohort study employed the Nova Scotia Atlee Perinatal Database, the Fetal Anomaly Database, and IWK Cleft Palate Database in Halifax, Nova Scotia, Canada. Outcome Measures: Cleft type, mode of diagnosis, and associated abnormalities of orofacial clefts for liveborn infants, stillbirths, and second trimester terminations of pregnancy between 1992 and 2002 were determined. Results: There were 225 fetuses identified as having orofacial clefts. The overall prevalence of clefts was 2.1 in 1000 live births, and this prevalence did not change with time. The overall antenatal detection of cleft lip with or without cleft palate was 23%; however, there was improvement in detection of cleft lip with or without cleft palate from the years 1992 to 1996 (14%) to the years 1997 to 2002 (30%, p  =  .02). No isolated cleft palates were detected antenatally. Associated structural anomalies were seen in 34.2% of cases with orofacial clefts, and chromosomal abnormalities were associated with 9.8%. Conclusions: The prevalence of orofacial clefts in Nova Scotia has not changed from 1992 to 2002. The proportion of antenatally diagnosed cleft lip with or without cleft palate in Nova Scotia is consistent with rates reported in the literature and has increased from 1992 to 2002.

A Multicenter Study Using the SWAG Scale to Compare Secondary Alveolar Bone Graft Outcomes for Patients With Cleft Lip and Palate

Objective To assess secondary alveolar bone graft (ABG) outcomes using the standardized way to assess grafts, or SWAG scale, for patients with cleft lip and palate (CLP). Design Retrospective cohort study. Setting Four cleft centers with different protocols. Methods One hundred sixty maxillary occlusal radiographs taken 3 to 18 months after secondary ABG for sequentially treated patients with CLP were assessed using the SWAG scale. Radiographs were scanned, standardized, blinded, and rated by six orthodontists using the SWAG scale. Randomized radiographs were rated twice, 24 hours apart, by the same raters. Main Outcomes Intrarater and interrater reliabilities were assessed. Means and SDs were calculated for ABG ratings using analysis of variance and Tukey tests (P < .05). Result The mean ABG age was 9.1 years (range = 7 to 10.1 years) and the mean follow-up age was 12.4 years (range = 8.2 to 20.4 years). Intrarater and interrater reliabilities were good (intrarater = 0.788, interrater = 0.705), and higher than published methods. Mean ABG ratings for the cleft centers were 4.53, 2.9, 3.63, and 5.0 and differed significantly. The two centers with the highest ABG ratings showed higher ratings for all thirds decreasing from apical to coronal. The two centers with lower ABG ratings showed poorer ABG ratings for all thirds, and the middle third received the highest ratings. Conclusions The SWAG scale overcomes the challenges of age and bone location. The SWAG method was validated for showing intercenter differences for overall bone fill as well as in vertical thirds. Surgical technique, timing, and expertise/volume were identified as possible factors related to outcome.

The Americleft Project: Burden of Care from Secondary Surgery

Background: The burden of care for children with cleft lip and palate extends beyond primary repair. Children may undergo multiple secondary surgeries to improve appearance or speech. The purpose of this study was to compare the use of secondary surgery between cleft centers. Methods: This retrospective cohort study included 130 children with complete unilateral cleft lip and palate treated consecutively at 4 cleft centers in North America. Data were collected on all lip, palate, and nasal surgeries. Nasolabial appearance was rated by a panel of judges using the Asher-McDade scale. Risk of secondary surgery was compared between centers using the log-rank test, and hazard ratios estimated with a Cox proportional hazards model. Results: Median follow-up was 18 years (interquartile range, 15–19). There were significant differences among centers in the risks of secondary lip surgery (P < 0.001) and secondary rhinoplasty (P < 0.001). The cumulative risk of secondary lip surgery by 10 years of age ranged from 5% to 60% among centers. The cumulative risk of secondary rhinoplasty by 20 years of age ranged from 47% to 79% among centers. No significant differences in nasolabial appearance were found between children who underwent secondary lip or nasal surgery and children who underwent only primary surgery (P > 0.10). Conclusions: Although some cleft centers were significantly more likely to perform secondary surgery, the use of secondary surgery did not achieve significantly better nasolabial appearance than what was achieved by children who underwent only primary surgery.

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

Abstract 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 Projects 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.