Risk Assessment of Sleep Disordered Breathing in Cleft Lip and/or Palate.

OBJECTIVE: Children with cleft lip and/or palate (CL/P) are at increased risk for Sleep Disordered Breathing (SDB), particularly Obstructive Sleep Apnea (OSA). At our institution, routine screening for SDB is performed using the Chevrin Pediatric Sleep Questionnaire (PSQ). This analysis is a practice audit looking at the outcomes of screening children with CL/P. DESIGN/SETTING/PATIENTS/PARTICIPANTS: A single-center, retrospective analysis was done of all non-syndromic patients with CL/P over the age of 36 months over a 4-year period. Children with known OSA were eliminated from analysis. MAIN OUTCOME MEASURES: Univariate logistic regression was used to assess predictors for SDB (PSQ score > 8) amongst various patient, disease, and treatment characteristics. Outcomes of those screened were tracked. RESULTS: Of the 239 patients in the study cohort, 43 (18%) had positive PSQs. These subjects were more likely to have class III dental occlusion with maxillary retrusion (OR = 2.65, 95% CI: 1.2-5.8, p = 0.02). There were no differences amongst age, type of cleft, Veau classification, BMI, or history of pharyngeal surgery. One third of the group did not complete recommended testing. Twenty-five subjects with positive sleep screening underwent subsequent polysomnography and 21 (84%) had OSA. CONCLUSION: Routine screening reveals a significant proportion of patients with CL/P with symptoms suggestive of OSA. While several patients did not complete confirmatory testing, those who completed a PSG had a high rate of identification of OSA. After excluding children with known OSA, patients with SDB are also likely to have class III dental occlusion and maxillary retrusion.

Speech Outcomes Following Operative Management of Velopharyngeal Dysfunction (VPD) in Non-Syndromic Post-Palatoplasty Cleft Palate Patients.

OBJECTIVE: Approximately 30% of patients with a history of repaired cleft palate (CP) go on to suffer from velopharyngeal dysfunction (VPD). This study discusses the operative management of VPD and postoperative speech outcomes in a cohort of CP patients. SETTING: An academic tertiary pediatric care center. METHODS: Retrospective cohort study. PATIENTS: Patients with history of repaired CP (Veau I-IV) who underwent operative management of VPD between January 1st, 2010 and December 31st, 2020. Operative modalities were posterior pharyngeal flap (PPF), sphincter pharyngoplasty (SPP), Furlow palate re-repair, and buccal myomucosal flap palate lengthening (PL). OUTCOME MEASURES: The primary outcome measure is postoperative speech improvement evaluated by the Pittsburgh Weighted Speech Scale (PWSS). RESULTS: 97 patients met inclusion criteria. 38 patients with previous straight-line primary palatoplasty underwent Furlow re-repair; these patients were significantly younger (7.62 vs 11.14, P < .001) and were more likely to have severe VPD per PWSS (OR 4.28, P < .01, 95% CI 1.46-12.56) when compared to VPD patients with previous Furlow repair. 21.1% of these patients required an additional non-revisional VPD procedure. The remaining patients underwent a non-revision procedure (26 PPF, 22 SPP, 11 PL); all experienced significant (P < .001 on paired t-test) reductions in PWSS total and subgroup VPD severity scores without difference in improvement between operation types. SPP was statistically associated with all-cause complication (OR 2.79, 95% CI 1.03-7.59, P < .05) and hyponasality (OR 3.27, 95% CI 1.112-9.630, P < .05). CONCLUSION: Furlow re-repair reduced need for additional VPD operations. Speech outcomes between non-revisional operations are comparable, but increased complications were seen in SPP.

Orofacial Manifestations of Stickler Syndrome: An Analysis of Speech Outcome and Facial Growth After Cleft Palate Repair.

PURPOSE: The purpose of this study was to characterize airway problems, speech outcomes, and facial growth in patients with Stickler syndrome undergoing cleft palate repair. METHODS: A retrospective, longitudinal study was performed at the Children's Hospital of Philadelphia on 25 patients with Stickler syndrome and 53 nonsyndromic patients with clefts of the secondary palate repaired between 1977 and 2000. Airway problems were characterized by the incidence of Pierre Robin Sequence (PRS) and the necessity for surgical airway management. Speech was analyzed using the Pittsburgh weighted values for speech symptoms associated with velopharyngeal incompetence (VPI). Longitudinal anthropometric measurements represented up to 12 years of longitudinal cephalofacial growth. RESULTS: Seventy-two percent of patients with Stickler syndrome were diagnosed with PRS, 55.6% of whom required surgical airway management. Conversely, 20.8% of nonsyndromic patients were diagnosed with PRS (P < 0.0001), 18% of whom required surgical intervention (P < 0.05). Speech outcomes were poorer in patients with Stickler syndrome with 40% demonstrating borderline VPI and 13.3% demonstrating VPI, compared with 21.8% and 9.1%, respectively, in the nonsyndromic group. Both groups exhibited significantly shallower upper and mid facial depths and wider upper facial breadths when compared with normal standards of facial growth. Although there was a tendency toward decreased facial depths in patients with Stickler syndrome relative to nonsyndromic patients, the differences were nonsignificant. CONCLUSIONS: Patients with Stickler syndrome show significant potential for early airway compromise and a poorer prognosis for speech outcome after cleft palate repair. Their cephalofacial growth does not differ significantly from that of nonsyndromic cleft palate patients.

Long Term Speech Outcomes Following Midface Advancement in Syndromic Craniosynostosis.

Midface advancement by distraction osteogenesis (DO) is commonly performed in patients with craniosynostosis for indications including midface hypoplasia, exorbitism, obstructive sleep apnea, class III malocclusion, and overall aesthetic facial deficiency. There is evidence to suggest that maxillary LeFort I advancement increases the risk of velopharyngeal dysfunction in the cleft palate population, yet few studies have investigated changes in speech following LeFort III or monobloc midface advancement in patients with syndromic craniosynostosis. The purpose of this study was to examine the effect of midface DO on speech as indicated by the Pittsburgh Weighted Speech Score in patients with Apert, Crouzon, and Pfeiffer Syndrome. Among 73 midface advancement cases performed during the study period, 19 cases met inclusion criteria. Overall, the highest post-advancement Pittsburgh Weighted Speech Score (PWSS) was significantly higher than the pre-advancement PWSS (0.52 versus 2.42, P = 0.01), indicating an acute worsening of VPI post-advancement. Specifically, the PWSS components nasal emission and nasality were significantly higher post-advancement than pre-advancement (nasal emission: 1.16 versus 0.21, P = 0.02) (nasality: 0.68 versus 0.05, P = 0.04). However, there was no significant difference between pre-advancement PWSS and the latest post-advancement PWSS (P = 0.31). Midface distraction is associated with an acute worsening of VPI post-operatively that is followed by improvement, and often resolution over time. Future work with additional patient accrual is needed to determine the effect of different advancement procedures and syndromes on VPI rates and profundity.

Change in Initial Tympanostomy Tube Placement Timing Yields Decreased Burden of Care Without Increased Complications in Patients With Cleft Lip and Palate.

OBJECTIVE: To report a practice audit of the consequences of a change in protocol in the timing of placement of tympanostomy tubes in infants with cleft lip and palate. PARTICIPANTS: All children with a diagnosis of cleft lip and palate, treated between November 1998 and May 2006 under the old protocol, and between December 2012 and July 2016 under a new protocol. Under the old protocol, tympanostomy tubes were first inserted at the time of lip repair at around age 2 months. Under the new protocol, tympanostomy tubes were deferred until the time of palate repair around the age of 9 months. Children with syndromic diagnoses other than Stickler syndrome and Van der Woude syndrome, and children who failed newborn hearing screen were excluded. MAIN OUTCOME MEASURES: Incidence of otorrhea from birth to 6 months after palate repair and presence of hearing loss at ages 1 and 2. RESULTS: Deferral of tympanostomy tubes until the time of palate repair decreases the burden of care due to otorrhea as compared to early tympanostomy tubes at the time of lip repair. There was no significant difference in the incidence of hearing thresholds at or below 15 dB at age 1 or 2. CONCLUSIONS: Placement of tympanostomy tubes at the time of palate repair balances the goals of minimizing the adverse effects from middle ear effusion and minimizing the burden of care on our patients and their families.

The Effects of a Nutrition Media Literacy Intervention on Parents' and Youths' Communication about Food.

Interventions addressing links between media exposure and obesity risk for school-age youth have not explicitly addressed the role of family communication about media. Youths' influence attempts on parents to purchase advertised foods can create conflict and negatively affect parental food choices. This study tested whether a family-based media literacy curriculum improves parents' media management skills and decreases youths' susceptibility to appealing but unrealistic food marketing. A matched-group pretest/posttest field experiment of parent-youth dyads with control group (N = 100 dyads, youth M = 11 years of age) tested the six-session curriculum. Hypotheses were analyzed using a Bayesian structural equation model. The curriculum increased parents' active negative mediation to foster youths' critical thinking about food marketing, b* = 0.35, 95% CCI [0.17, 0.50], increased parent Efficacy for making healthy dietary changes for their families, b* = 0.59, 95% CCI [0.41, 0.75], and fostered family discussion about nutrition labels (total effect = 0.22). Additionally, cumulative influences of Perceived Desirability and Wishful Identification on youths' requests for marketed foods were reduced (total effect = 0.04). Media literacy education can empower parents and improve youths' critical thinking to reduce effects of food marketing on families and improve use of media to obtain nutrition information.

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.

Emergency department length of stay in critical nonoperative trauma.

BACKGROUND: Prolonged emergency department (ED) stays correlate with negative outcomes in critically ill nontrauma patients. This study sought to determine the effect of ED length of stay (LOS) on trauma patients. MATERIALS AND METHODS: Two hundred forty-one trauma patients requiring direct intensive care unit (ICU) admission were identified. Patients requiring immediate operative intervention were excluded. Odds ratios (ORs) of outcomes for patients transferred to ICU in ≤90 min were compared with patients transferred in >90 min, adjusting for Injury Severity Score (ISS). RESULTS: One hundred two of 241 patients (42%) were transferred to the ICU in ≤90 min. Increased ED LOS was associated with decreased complications (OR 0.545, 95% confidence interval 0.312-0.952). Although the result was not statistically significant, patients with an ISS >15 were less likely to have long ED stays (OR 0.725, 95% CI 0.407-1.290). No significant difference was seen in mortality. No difference in duration of intubation was observed for patients intubated in the ED versus the ICU. For the subgroup with ISS ≤15, there was a significant decrease in ICU LOS for patients who remained in the ED >90 min (5.5 d versus 2.7 d, P = 0.02). No other differences in LOS were identified. CONCLUSIONS: In a mature trauma center with standardized activation protocols and focused resource allocation in the ED trauma bay, trauma activation and subsequent management appear to mitigate the negative effects of prolonged ED LOS seen in other critically ill populations.

The Effect of Furlow Palatoplasty Timing on Speech Outcomes in Submucous Cleft Palate.

BACKGROUND: Because some patients with submucous cleft palate (SMCP) are asymptomatic, surgical treatment is conventionally delayed until hypernasal resonance is identified during speech production. We aim to identify whether speech outcomes after repair of a SMCP is influenced by age of repair. METHODS: We retrospectively studied nonsyndromic children with SMCP. Speech results, before and after any surgical treatment or physical management of the palate were compared using the Pittsburgh Weighted Speech Scoring system. RESULTS: Furlow palatoplasty was performed on 40 nonsyndromic patients with SMCP, and 26 patients were not surgically treated. Total composite speech scores improved significantly among children repaired between 3 and 4 years of age (P = 0.02), but not older than 4 years (P = 0.63). Twelve (86%) of 14 patients repaired who are older than 4 years had borderline or incompetent speech (composite Pittsburgh Weighted Speech Scoring ≥3) compared with 2 (29%) of 7 repaired between 3 and 4 years of age (P = 0.0068), despite worse prerepair scores in the latter group. Resonance improved in children repaired who are older than 4 years, but articulation errors persisted to a greater degree than those treated before 4 years of age (P = 0.01.) CONCLUSIONS: Submucous cleft palate repair before 4 years of age appears associated with lower ultimate rates of borderline or incompetent speech. Speech of patients repaired at or after 4 years of age seems to be characterized by persistent misarticulation. These findings highlight the importance of timely diagnosis and management.

International Adoptees With Cleft Lip and/or Palate: Lessons Learned From a Growing Population.

BACKGROUND: International adoptees with cleft lip and palate (CLP) are a growing population in the United States. They represent a clinical challenge, presenting at various ages and stages of cleft repair. METHODS: A retrospective review of patients seen at the CLP Program at the Children's Hospital of Philadelphia (CHOP) between 1998 and 2012 with a history of international adoption was performed. Demographics, surgical histories, and long-term speech outcomes were reviewed. RESULTS: Seventy-four female and 77 male patients were evaluated. Patients were adopted at an average age of 2.3 years (range, 0.4-8.6 years); 80.8% (n = 122) of patients were adopted from China. The rate of international cleft adoption increased by approximately 1.5 patients per year (r = 0.7739, P < 0.001); 13.2% (n = 19) of all subjects with cleft palates had oronasal fistulas (ONFs) that required repair. The ONF rates for primary palatoplasties at CHOP were significantly lower compared to both preadoption repairs (P = 0.002) and postadoption repairs at outside hospitals (P = 0.01); 14.8% (n = 21) of all patients had secondary surgeries for velopharyngeal incompetence (VPI). Rates of secondary surgery for VPI were also significantly lower for primary palatoplasties at CHOP compared to both preadoption repairs (P = 0.0018) and postadoption repairs at outside hospitals (P = 0.0033). CONCLUSIONS: International adoptees with CLP are a growing population and are clinically challenging with high ONF rates and high secondary surgery rates for VPI. We recommend expedited repair of unoperated cleft palates in adoptees older than 18 months. Adopted patients with CLP should be rigorously evaluated for the need for speech therapy and secondary surgeries to correct for VPI.

A 35-year experience with syndromic cleft palate repair: operative outcomes and long-term speech function.

BACKGROUND: Associated comorbidities can put syndromic patients with cleft palate at risk for poor speech outcomes. Reported rates of velopharyngeal insufficiency (VPI) vary from 8% to 64%, and need for secondary VPI surgery from 23% to 64%, with few studies providing long-term follow-up. The purpose of this study was to describe our institutional long-term experience with syndromic patients undergoing cleft palatoplasty. METHODS: A retrospective review was conducted of all patients with syndromic diagnoses undergoing primary Furlow palatoplasty from 1975 to 2011. Outcomes included postoperative oronasal fistula (ONF) and need for secondary VPI surgery. Speech scores for verbal patients 5 years or older were collected via the Pittsburgh scale for speech assessment. Aggregate scores categorized the velopharyngeal mechanism as competent, borderline, or incompetent. Outcomes were analyzed by patient and operative factors. RESULTS: One hundred thirty-two patients were included with average age at repair of 20.7 months. Cleft type was 9% submucosal, 16% Veau class I, 50% class II, 12% class III, and 13% class IV. Forty-five syndromes were recorded, most commonly Stickler syndrome (n = 32) and 22q11.2 deletion syndrome [22q11.2DS (n = 19)]. Forty-four patients also had associated Pierre Robin sequence (PRS). The overall ONF rate was 4.5% and was highest in Veau class IV clefts (P = 0.048). Seventy-six patients were included in speech analysis, with an average age at last assessment of 10.4 years. Overall, 60.5% of patients had a competent velopharyngeal mechanism, 23.7% borderline, and 15.8% incompetent mechanism. Fifty percent of 22q11.2DS patients had borderline speech and none had competent speech, compared to 73.3% with Stickler syndrome (P = 0.01) and 71.4% of patients with associated PRS (P = 0.02). Secondary VPI surgery was performed in 11.4% of patients overall. Patients with PRS (13.6%) and with Stickler syndrome (15.6%) had secondary VPI surgery, compared to 31.6% of patients with 22q11.2DS (P = 0.01). CONCLUSIONS: This study demonstrates low rates of postoperative ONF after modified Furlow palatoplasty in syndromic patients. Speech outcomes were comparable to nonsyndromic patients at our institution, but patients with 22q11.2DS consistently had borderline-incompetent speech and a 3-fold higher incidence of secondary VPI surgery.

Do patients with Pierre Robin sequence have worse outcomes after cleft palate surgery?

BACKGROUND: Pierre Robin sequence (PRS) is a congenital condition characterized by micrognathia, glossoptosis, airway obstruction, and often clefting of the soft and hard palate. The purpose of this study was to compare oronasal fistula rates, long-term speech outcomes, and rates of secondary surgery for velopharyngeal insufficiency (VPI) after primary cleft palate repair using the modified Furlow technique between nonsyndromic patients with and without PRS. This study also sought to determine if type of airway management in infancy correlated with speech outcomes in patients with PRS. METHODS: A retrospective review was performed of all nonsyndromic patients with clefts of the palate only who underwent repair between 1981 and 2006 at The Children's Hospital of Philadelphia using the modified Furlow technique. Patient outcomes were evaluated by the rate of postoperative oronasal fistula, speech scores at a minimum of age 5 years using the Pittsburgh Weighted Values for Speech Symptoms Associated with VPI, and the need for secondary pharyngeal surgery for VPI. RESULTS: Fifty-five patients with PRS and 129 without PRS were included in this study. There was no significant difference in oronasal fistula rate between groups. Speech outcomes were worse in patients with PRS with 52.7% demonstrating a competent velopharyngeal mechanism, and 30.9% a borderline mechanism, compared to 72.1% and 19.4% in the non-PRS group, respectively (P = 0.035). In addition, only 76.3% of patients with PRS had no or mild hypernasality compared to 91.5% of patients without PRS (P = 0.01). The rates of nasal emission, articulation errors associated with VPI, and secondary surgery for VPI did not differ between groups. Of the patients with PRS, 36 were managed in infancy with positioning alone and 15 underwent surgical intervention for airway obstruction, and there were no significant differences in speech scores or rates of secondary surgery for VPI between these subgroups. CONCLUSIONS: Nonsyndromic patients with PRS had worse speech outcomes after modified Furlow cleft palate repair, but no significant differences were seen in the rates of secondary surgery for VPI or postoperative oronasal fistula. In addition, initial airway management in patients with PRS did not correlate with speech outcomes.

Speech Outcomes following Mandibular Distraction Osteogenesis for Robin Sequence: Midchildhood Results.

BACKGROUND: The aim of this study was to compare midchildhood speech outcomes in patients with nonsyndromic Robin sequence with cleft palate (RSCP) treated with mandibular distraction osteogenesis (MDO) to patients with nonsyndromic Veau class I and Veau class II cleft palate (CP). METHODS: The authors performed a retrospective review of patients with nonsyndromic Robin sequence from 2000 to 2017, comparing those who underwent MDO to patients with nonsyndromic CP. Demographics, operative details, length of hospital stay, complications, and Pittsburgh Weighted Speech Scale scores were collected. RESULTS: Thirty-three patients met inclusion criteria in the MDO group with 127 patients as controls. Despite similar median age (RSCP, 4.5 years; CP only, 4.6 years) and Veau cleft type at early evaluation, there was a significant increase in composite Pittsburgh Weighted Speech Scale score within the MDO cohort ( P ≤ 0.002); specifically, with worse visible nasal emission ( P ≤ 0.007), hypernasality ( P ≤ 0.001), and compensatory articulation ( P ≤ 0.015). However, these differences were not present at age-matched midchildhood evaluation (median, RSCP, 6.5; CP only, 7.1; P ≥ 0.092). Median age-matched follow-up was 6.4 years in the MDO group and 7.1 years in the control group ( P ≥ 0.136). There was also no difference in the rate of secondary speech surgery at midchildhood evaluation ( P ≥ 0.688). CONCLUSIONS: The authors' retrospective comparison of speech outcomes in RSCP versus CP only demonstrates no difference in midchildhood speech, conflicting with recent reports. Although patients with Robin sequence treated with MDO had worse visible nasal emission, hypernasality, and compensatory articulation in early childhood, this appears to have resolved in the interim without additional intervention. Longitudinal follow-up is needed to fully understand the speech ramifications of RSCP. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.

Speech Outcomes and Rates of Secondary Speech Surgery from Childhood to Skeletal Maturity Following Modified-Furlow Palatoplasty.

BACKGROUND: This study updates our institutional experience with modified Furlow palatoplasty, evaluating speech outcomes and incidence of secondary speech surgery throughout development and at skeletal maturity. METHODS: Nonsyndromic patients undergoing primary modified Furlow palatoplasty between 1985 and 2005 with post-operative speech evaluations were retrospectively reviewed. Secondary speech surgery and Pittsburgh Weighted Speech Scale (PWSS) scores prior to secondary speech or orthognathic surgeries were assessed in the 5-7, 8-11, 12-14, and 15+ age groups and analyzed by Veau cleft type. RESULTS: Five hundred fifty-one patients with 895 total speech assessments were analyzed. Of 364 patients followed to age 15 or older, 19.8% underwent secondary speech surgery. Speech assessment of patients aged 15 or older without prior secondary speech surgery showed competent velopharyngeal mechanisms in 77% of patients. PWSS nasal emission scores were worse in the 5-7 age range (p=0.02), while resonance scores remained stable throughout development (p=0.2). Patients with Veau type I or II clefts had worse overall PWSS classifications in the age 5-7 and 8-11 age groups (p=0.01, p=0.03), with greater odds of secondary speech surgery relative to those with Veau type III (OR 2.9, p<0.001) or IV clefts (OR 3.6, p=0.001). CONCLUSIONS: Most patients undergoing primary modified Furlow palatoplasty do not require secondary speech surgery and achieve socially acceptable speech at skeletal maturity. However, Veau type I and II clefts are associated with increased risk for early velopharyngeal dysfunction and secondary speech surgery. Incidence of secondary speech surgery was 19.8%, an increase from our previously reported rate of 8%.

The role of media literacy in shaping adolescents' understanding of and responses to sexual portrayals in mass media.

This study was a theory-based, pretest-posttest quasi-experiment conducted in the field (N = 922) to determine whether and how a media literacy curriculum addressing sexual portrayals in the media would influence adolescents' decision-making processes regarding sex. Results of the evaluation, based on the Message Interpretation Process Model, indicated that participants who received media literacy training better understood that media influence teens' decision making about sex and were more likely to report that sexual depictions in the media are inaccurate and glamorized. In addition, participants who received media literacy lessons were more likely than were control group participants to believe that other teens practice abstinence and reported a greater ability to resist peer pressure. An interaction effect existed between gender and condition on attitudes toward abstinence, suggesting that the lessons helped girls and boys in somewhat different ways. Overall, the results indicated that media literacy strengthened key aspects of participants' logic-oriented decision-making process.

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 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 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 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).