Ronald R. Hathaway

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.

The Americleft Project: A Proposed Expanded Nasolabial Appearance Yardstick for 5- to 7-Year-Old Patients With Complete Unilateral Cleft Lip and Palate (CUCLP)

Objective To develop a yardstick of reference photographs for nasolabial appearance assessments of 5- to 7-year-old patients with complete unilateral cleft lip and palate (CUCLP). Design Blind retrospective analysis of clinical records and comparison to historical controls. Patients Subjects were two groups of 6- to 12-year-olds (n = 124 and n = 135) and one group of 5- to 7-year-olds (n = 149) with nonsyndromic CUCLP from three previous Americleft studies, including cohorts from seven different cleft/craniofacial centers. Interventions All patients received the infant management protocols of their respective centers. Eleven trained and calibrated judges (five participated in all three studies) did blind ratings of nasolabial appearance using the Asher-McDade method. Main Outcome Measures Patients receiving the most consistent ratings between judges, selected first from the groups of 6- to 12-year-olds, were used to create a pilot yardstick for eventual use in the third study of 5- to 7-year-olds. For each of the Asher-McDade categories, 8 of the 5- to 7-year-old patients receiving the most consistent scores between raters were ranked by 10 judges for a final elimination to leave three per category. Results Using this method of successive changes in rating methods, a new reference yardstick for nasolabial appearance rating was established and linked to the original Asher-McDade method as well as the single examples in a previously published yardstick for patients with CUCLP. Pilot testing using the yardstick improved reliabilities. Conclusions Use of an expanded nasolabial yardstick of reference photographs representative of the range of possibilities of each of the five Asher-McDade categories is now available to see if reliability of these ratings can be improved.

Primary alveolar cleft bone grafting in unilateral cleft lip and palate: arch dimensions at age 8.

The purpose of this investigation is to determine whether primary alveolar cleft bone grafting in infants with unilateral cleft lip and palate (N = 17) leads to less favorable dental arch dimensions at age 8 when compared with other 8-year-old patients with unilateral cleft lip and palate who received no alveolar bone grafting procedures (N = 49). Dental casts were obtained for the primary grafted group, and arch lengths and widths were digitally recorded with a reflex microscope. These arch dimensions were then compared with the reported data for a nongrafted group and a noncleft group of 8-year-old children. The major findings were: 1) that the dental arches of both cleft groups generally demonstrated a significant diminution in length and width (P < 0.05) compared with the noncleft groups, and 2) that the patients who underwent primary alveolar cleft bone grafting showed no statistically significant difference for any arch dimension (P < 0.05) when compared with the nongrafted group lacking this additional surgical procedure.

Craniofacial correction of giant frontoethmoidal encephalomeningocele.

The surgical treatment of a very large anterior encephalocele in an infant is presented. Because of the large size of the encephalocele, a combined transfacial-transcranial approach was used for correction of the associated intracranial, cranioorbitonasal bone, and facial skin deformities.

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.

Primary alveolar cleft bone grafting in unilateral cleft lip and palate: craniofacial form at age 8.

Counterpart analysis can be advantageous for the clinician interested in the underlying determinants of the craniofacial form for any given person. This analysis was performed for a group of patients who underwent primary alveolar cleft bone grafting (N = 18) and a group of patients who did not undergo grafting (N = 19) who were 8 years of age (+/- 6 months). The primary grafting group more frequently noted maxillary retrusion, but of a nonsignificant magnitude. Also, the primary grafting group had greater mean magnitudes of mandibular opening as a compensatory adjustment in some patients, but this could not be generalized to all patients who had underdone primary grafting. The mean magnitude of craniofacial vertical shortening was also greater for some patients who had undergone primary grafting, but it, too, did not exhibit a generalized pattern for all patients who had undergone primary alveolar cleft bone grafting procedures. This study emphasizes the great diversity of craniofacial skeletal adjustments made within each group of patients with unilateral cleft lip and palate and cautions the clinician against generalizations concerning a particular treatment protocol.