Citations

Objective Assess if velopharyngeal gap size during speech is associated with consonant accuracy and whether hypernasality severity and compensatory misarticulation errors (CMAs) mediate this relationship. Design Cross-sectional. Setting Metropolitan hospital. Participants Eighty-one patients undergoing assessment for velopharyngeal insufficiency (VPI). Assessment(s) Each patient completed a preoperative magnetic resonance imaging protocol during sustained /i/ production and a perceptual speech evaluation. Velopharyngeal gap size was measured in millimeters. Hypernasality was rated using the Cleft Audit Protocol for Speech-Augmented-Americleft Modification. Percent consonants correct (PCC) and percent of CMAs used were derived from transcriptions of the American English sentence or phrase sample. Main Outcome Measure(s) A generalized structural equation model was used to examine whether velopharyngeal gap size is associated with PCC indirectly through hypernasality severity and CMA use. Results Larger velopharyngeal gap sizes were associated with more severe hypernasality ( P < .001), greater hypernasality severity was associated with increased CMA use ( P < .001), and greater CMA use was associated with lower PCC ( P < .001). Velopharyngeal gap size was not directly associated with PCC ( P = .400). However, the full indirect pathway from gap size to PCC through hypernasality severity and CMA use was significant ( P = .001); gap size was significantly associated with PCC when accounting for both direct and indirect pathways combined ( P = .005). Conclusions These findings suggest hypernasality severity is the key mediator between gap size and speech accuracy. Patients with more severe hypernasality may benefit from timely surgical remediation of VPI to increase overall consonant accuracy.

Objective To identify barriers and facilitators to changing provider's current approaches to evaluation and treatment decision-making for velopharyngeal insufficiency (VPI). Design Cross-sectional qualitative study. Setting Medium and large multidisciplinary cleft teams in the USA and Canada engaged in clinical research. Participants Twenty-eight surgeons and 16 speech-language pathologists from 12 cleft teams. Interventions Semi-structured interviews were conducted exploring perceived barriers and facilitators to changing VPI evaluation and treatment decision-making. Interviews were analyzed using a thematic approach guided by the Consolidated Framework for Implementation Research. Results Providers indicated that adoption of new approaches to VPI evaluation and decision-making would require evidence demonstrating improved speech outcomes or reduced risk of obstructive sleep apnea. Characteristics of persuasive evidence included prospective study design, comparable patient groups, standardized speech evaluations, and long-term follow-up. Providers favored applying new approaches to complex cases or defined subgroups, rather than broad adoption. Providers raised concerns about additional preoperative testing, perceiving it could increase treatment burden. When reflecting on prior changes in care delivery, providers indicated they started by discussing the potential change informally with team members to share information, gather input, and build consensus; in larger teams, discussions also occurred in formal meetings or through administrative hierarchies. Implementation decisions were consensus-based, although the pace of implementation varied among providers. Conclusions Providers have processes for implementing changes in their VPI evaluation and treatment decision-making, and criteria by which they make the decision to implement. These findings can inform the design of implementation strategies to accelerate clinical uptake of research findings.

Purpose: Nasopharyngoscopy is an imaging method used to visualize the velopharyngeal mechanism. To quantify the movement of the mechanism during speech, an international working group recommended standardized rating guidelines. While these guidelines are widely cited in research, the extent to which cleft teams apply them in a clinical setting is unclear. Method: This cross-sectional study examined clinical nasopharyngoscopy reports from 11 cleft teams in North America. Descriptive statistics were used to summarize the proportion of reports that included ratings for the extent of velar movement, left and right lateral pharyngeal wall movement, gap size, and a surgical recommendation. Fisher's exact and chi-square tests were used to assess differences in reporting by cleft team and whether the inclusion of quantitative ratings was associated with a surgical recommendation being included. Results: A total of 188 nasopharyngoscopy reports were included. The extent of velar movement was reported in 68% ( n = 127) of reports, the left and right lateral pharyngeal wall movement was reported in 38% ( n = 71), and the size of the gap was reported in 70% ( n = 132). Only 34% ( n = 63) of reports included all three quantitative ratings. Inclusion of all three quantitative ratings varied significantly ( p < .001) across teams. Teams either never included all the ratings ( n = 4/11) or inconsistently included all ratings ( n = 7/11). Conclusions: Findings suggest that what is documented clinically in nasopharyngoscopy reports varies both within and across teams. To achieve more consistent reporting within individual teams, providers may consider implementing standardized clinical templates. To achieve consistency across teams, these findings suggest a need for renewed efforts at developing consensus on the nasopharyngoscopy elements important to clinicians.

Objective Assess the patient compliance rate for nasopharyngoscopy as documented in clinical reports. Design Cross-sectional. Setting Thirteen cleft teams in North America. Patients Patients aged 3 to 21 years old with a repaired cleft palate. Interventions Nasopharyngoscopy. Main Outcome Measure Patient compliance rate for nasopharyngoscopy. Results Patient compliance was documented in 128 of 158 reports (81%). Of the 128 reports, patient compliance was reported as “good/excellent” in 65% (n = 83), “marginal/fair” across 10% (n = 13), “poor” in 16% (n = 20), and “other” in 9% (n = 12). Patients with “poor” compliance had lower rates of documented imaging ratings, however 55% (n = 11) of reports included a surgical recommendation. Conclusions Among children completing nasopharyngoscopy for velopharyngeal insufficiency surgery planning, at least one-fourth of reports indicate significant compliance issues that limits velopharyngeal port imaging. These findings suggest that some cleft teams and surgeons may be proceeding with surgical management of velopharyngeal insufficiency without adequate visualization or ratings of velopharyngeal anatomy.

Objective To assess the relationship in ratings of velopharyngeal closure between connected speech and sustained speech, as visualized on nasopharyngoscopy. Design Prospective observational study. Setting Craniofacial clinics in the United States and Canada. Participants Fifty-two participants with velopharyngeal insufficiency (VPI) who underwent nasopharyngoscopy and produced both connected speech and sustained speech of either /i/or /s/ during the procedure. Main outcome measure(s) Correlations and differences between connected speech and sustained speech for velopharyngeal closure percentage, velar movement, lateral pharyngeal wall movement, and velopharyngeal closure pattern. Results There were moderate to very strong correlations observed between connected speech and sustained /i/ for velopharyngeal closure percentage ( P < .01); extent of velar movement ( P < .01), and left ( P < .01) and right ( P < .01) lateral pharyngeal wall movement. Findings were similar between connected speech and sustained /s/. Differences between ratings made during connected and sustained speech were small: for velopharyngeal closure percentage, there was a 5% difference in the mean between connected speech and sustained /i/ (77.5% vs 72.5%) and a 7% difference for /s/ (79.3% vs 86.7%). Overall differences across ratings ranged from 2% to 12%. Closure pattern was rated the same for both connected speech and sustained /i/ in 90% of videos (n = 44/49; P = .38) and the same for connected speech and sustained /s/ in 95% of videos (n = 42/44; P = 1.00). Conclusions Ratings of velopharyngeal movement during nasopharyngoscopy are similar between connected speech and sustained /i/ and /s/ suggesting elicitation of only sustained /i/ and /s/ may be adequate in most cases when assessment of connected speech is not possible.

Purpose: The purpose of this study was to compare the height of velopharyngeal (VP) closure in relation to the palatal plane between individuals with velopharyngeal insufficiency (VPI) with or without cleft palate and individuals without cleft palate and with normal speech, considering the effects of age and sex. Method: Magnetic resonance imaging was used to analyze the height of VP closure in the midsagittal view during phonation of /i/ and /s/ sounds among 105 individuals with VPI and 45 individuals without cleft palate and normal speech. Results: Independent-samples t tests demonstrated that individuals with VPI display a significantly lower mean height of VP closure (3.7 mm) compared to individuals without cleft palate and normal speech during phonation of /i/ and /s/ sounds ( p < .001). Analysis of variance was used to evaluate the impact of age and sex on the height of VP closure. Significant differences were observed between the height of VP closure in individuals with VPI and those without cleft palate and normal speech across the age span during the production of /i/ and /s/ sounds. However, nonsignificant differences were found between the height of VP closure in males and females of the VPI group and those without cleft palate and normal speech during the production of these sounds. Conclusions: The results revealed the variations of the height of VP closure in relation to the palatal plane. These findings provide insights into the presurgical planning for individuals with VPI undergoing palatal surgeries, especially the location of pharyngeal flap insertion.

Obstructive sleep disordered breathing (oSDB), including obstructive sleep apnea (OSA), frequently coexists with cleft lip and palate, velopharyngeal insufficiency (VPI), and other craniofacial conditions. Moreover, oSDB is often a key factor in decisions for primary or revision cleft palate or velpharyngeal surgery. Due to the frequency with which cleft and craniofacial team members care for patients with oSDB, this article provides a primer for OSA evaluation and management for team members. oSDB can have a significant impact on quality of life, health outcomes, surgical planning, and global development for all children, especially those with cleft and craniofacial conditions. In this review, oSDB screening and testing methods are discussed, including interpretation of OSA testing. Considerations for specific craniofacial populations are included, such as those with 22q11.2 deletion syndrome and Pierre-Robin sequence. The impact of VPI surgery on OSA is discussed. Finally, approaches to OSA management are reviewed, with attention to how standard management approaches may shift in children with cleft and craniofacial conditions. As awareness of oSDB in children has increased, including increased access to diagnostic testing, studies have shown certain patient populations are more likely to have OSA and would benefit from screening. Children with craniofacial conditions, including cleft palate and velopharyngeal insufficiency, have altered risk profiles which impact management considerations for OSA. This article provides an introduction to key concepts in oSDB screening, diagnositics, and management, which can be valuable for any professional assisting in the care of children with a cleft or craniofacial condition.

Objective To explore current approaches to evaluation and treatment decision-making for velopharyngeal insufficiency (VPI) following cleft palate repair. Design Cross-sectional, qualitative study. Setting/participants Participants included 28 surgeons and 17 speech-language pathologists (SLPs) from 12 cleft teams in the United States and Canada. Interventions Semi-structured, qualitative interviews were conducted exploring participants’ current approaches to VPI evaluation and treatment decision-making. Data were analyzed using thematic analysis. Results Participants viewed VPI evaluation as a joint effort of surgeons and SLPs. SLPs were entrusted to conduct formal speech evaluations. All teams completed a perceptual evaluation of speech resonance that included spontaneous speech and phrase repetition. Teams varied in their use of patient questionnaires and nasometry. Most teams performed nasoendoscopy as part of their evaluation, although providers noted limited cooperation in younger children and those with developmental delay. Treatment decision-making occurred either as a joint effort between the evaluating surgeon and SLP or exclusively by the surgeon. Most surgeons employed a personalized approach to treatment decision-making, with 19 distinct approaches described. Elements frequently considered in surgical procedure selection included velopharyngeal gap size and closure pattern as seen on nasoendoscopy. Providers were satisfied with their current approach, although most identified opportunities for improvement. Conclusions Cleft teams have individualized approaches to VPI evaluation and treatment decision-making, with the only common element being completion of a perceptual speech evaluation by an SLP. These findings suggest that efforts to implement new approaches will need to be customized to each team's current process.

Objective Prior work has identified numerous barriers to standardized outcome measurement in cleft care. The objective of this study was to evaluate a multifaceted implementation strategy to collect standardized outcomes for children with cleft lip and/or palate (CL/P). Design A one-armed, single-site pilot study of a multifaceted implementation strategy. Setting A metropolitan children's hospital. Patients Five-year-old children with CL/P. Interventions A multifaceted implementation strategy for standardized outcome measurement. Main Outcomes Measures The primary outcome measure was penetration of the intervention, defined as the proportion of children for whom the standardized measures of aesthetic, dental, and speech outcomes were collected. Penetration pre-implementation and 12 months post-implementation were compared. The secondary outcome was acceptability of standardized outcome measurement among providers and staff, evaluated with the Acceptability of Intervention Measure. Results The implementation strategy resulted in high penetration of standardized outcome measurement: measurement of aesthetic outcomes increased from 7% to 82% ( p < .0001); measurement of dental outcomes increased from 0% to 56% ( p < .0001); measurement of speech outcomes increased from 29% to 77% ( p = .0001). Acceptability of standardized outcome measurement was high, with 93% of cleft team providers and staff (n = 14/15) reporting that they somewhat or completely agreed that standardized outcome measurement met their approval, was appealing, was welcomed, and was liked. Penetration continued to improve after the implementation period, with high rates of outcome measure collection sustained for four years following implementation. Conclusions Deployment of a multifaceted implementation strategy led to substantial increases in the collection of aesthetic, dental, and speech outcomes among children with CL/P.

Background: Surgeons pursuing improvement in the aesthetic outcomes of their cleft lip repairs may benefit from a granular scale evaluating individual objectives of the repair. Methods: A working group of 9 surgeons convened to develop an assessment scale for nasolabial aesthetics after unilateral cleft lip repair. The group identified objectives of the repair that could be evaluated using two-dimensional facial photographs. Scale items were developed to appraise success or failure in achieving each objective. Scale items were iteratively tested and refined. The scale was subsequently implemented as part of a Continuing Medical Education course that included self-evaluation and peer-to-peer education, culminating in the formation of individual plans for improvement. Results: Twelve distinct objectives of unilateral cleft lip repair were identified, of which 10 could be evaluated using photographs routinely obtained in clinical practice. A comprehensive scale was developed, incorporating these 10 objectives. Each scale item takes the form of a binary (yes/no) question evaluating a specific aesthetic concept, with accompanying reference images. Intrarater reliability for each item ranged from moderate to substantial (kappa value, 0.57 to 0.81). Interrater reliability ranged from fair to substantial (kappa value, 0.27 to 0.81). When implemented in a Continuing Medical Education course, the scale enabled surgeons to identify specific opportunities for improvement in their repair and specific surgical maneuvers to adopt in pursuit of these improvements. Conclusions: A new scale for evaluating outcomes of unilateral cleft lip repair is presented. The scale provides specific, actionable evaluations for individual objectives of the repair.