Chee-Williams, Jessica L.

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.

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: There remains significant uncertainty about the fistula rate following palate repair with the Furlow double opposing Z-plasty (Furlow DOZ) technique compared with a straight-line/mucosal incision with intravelar veloplasty (IVVP) technique. Moreover, the relative impact of technique and surgical skill on fistula incidence remains unclear. Methods: A prospective, observational study of cleft palate repair was conducted. Enrollment occurred at 18 U.S.-based cleft centers, including 64 cleft surgeons. The primary outcome was postoperative fistula. Results: Overall, 1219 participants underwent cleft palate repair, 1178 of whom presented for follow-up. Of these, 581 (49.3%) underwent palate repair with the Furlow DOZ technique, 483 (41.0%) with the straight-line/IVVP technique, and 114 (9.7%) with an alternative technique. The overall fistula incidence was 5.3% (62 of 1162 analyzed for fistula occurrence). The fistula incidence was significantly lower in the Furlow DOZ group (2.4% [14 of 564]) than in the straight-line/IVVP group (7.8% [37 of 427]), with a propensity score–weighted risk difference of 5.7% (95% CI, 1.6% to 9.9%; P = 0.007). The fistula incidence varied substantially among surgeons: the top quartile’s fistula rate was 0% (0 fistulas among 417 repairs), whereas the bottom quartile’s fistula rate was 18.9% (31 fistulas among 164 repairs). Conclusions: Fistula incidence following cleft palate repair was significantly lower when using the Furlow DOZ technique, compared with the straight-line/IVVP technique. The effect of surgical technique was smaller, however, than the variation observed between the top and bottom quartiles of surgeons. These findings suggest that changing surgical technique may lead to small changes in fistula incidence, but greater opportunities exist to improve outcomes by improving surgical skill.

Objective: Describe changes in symptoms of velopharyngeal insufficiency (VPI)—hypernasality, audible nasal emission, and VPI-related quality of life—following LeFort I osteotomy in individuals with cleft palate +/- cleft lip. Design: Single-center, retrospective cohort study. Participants: Twenty-one patients with a history of cleft palate repaired in infancy were included in this study. Ten patients had no prior history of VPI surgery and eleven had a pharyngeal flap in place at the time of LeFort I osteotomy. The mean age at time of surgery was 17.2 years. Main Outcome Measure: Pre- and postoperative hypernasality severity, frequency of audible nasal emission, and total score on the VPI Effects on Life Outcomes youth questionnaire (VELO-Y) before and after LeFort osteotomy I. Fischer exact test was used to assess differences between groups for categorical variables and a t test was used for continuous variables. Results: Patients with no prior history of VPI management had a significant increase in hypernasality severity compared with those with a pharyngeal flap in place at the time of surgery ( P =0.004). Patients without a prior history of VPI management also had a significant increase in frequency of audible nasal emission, while patients with a pharyngeal flap had decreased audible nasal emission ( P =0.004). There was not a significant difference between groups for change in the total score on the VELO-Y ( P =0.16). Conclusions: Findings from this study may improve preoperative counseling provided to patients and caregivers on the risk of acquiring VPI after LeFort I osteotomy based on prior VPI surgical history.

Objective To explore if observations on nasopharyngoscopy influence the surgical procedure selected for the management of velopharyngeal insufficiency (VPI). Design Cross-sectional survey. Participants Seventeen surgeons with experience treating VPI. Interventions Review of twenty-four nasopharyngoscopy videos with a combination of velopharyngeal closure patterns and gap sizes. Main Outcome Measures Surgical procedure was selected after watching a nasopharyngoscopy video. Chi-square tests were used to evaluate if, for each surgeon, procedure selection varied based on closure pattern and gap size combination. Variable importance scores were obtained from a random forest analysis to quantify contribution of nasopharyngoscopy observations on procedure selection. Results Four procedures accounted for 86% of all surgeries selected: Furlow double-opposing Z-Plasty (33%), palatal lengthening using buccal myomucosal flaps (20%), pharyngeal flap (19%), and sphincter pharyngoplasty (14%). Four surgeons (23.5%) significantly varied their surgical selection when different closure pattern and gap size combinations were shown on nasopharyngoscopy. Ten surgeons (58%) selected the same procedure in ≥ 50% of the 24 nasopharyngoscopy videos. Individual surgeon preference was the strongest predictor of surgical selection, accounting for 57.9% of surgical selection variance. Velopharyngeal gap size accounted for 36.5% of the variance in surgical selection, yet the specific surgeries selected based on gap size varied widely across surgeons. Closure pattern was a weak predictor, contributing 5.6% to surgical selection variance. Conclusions Nasopharyngoscopy findings influence VPI surgical procedure selection in a highly individualized, surgeon-specific manner. Among factors observed on nasopharyngoscopy, velopharyngeal gap size has a much larger influence on procedure selection than closure pattern.

Background: Positioning the pharyngeal flap base high along the posterior pharyngeal wall is essential for optimizing speech outcomes. Objective data on where to place the flap base are lacking. Further, adenoid tissue can restrict cephalad positioning of the flap. This study aimed to improve the design of the pharyngeal flap by measuring the distance from the first cervical vertebrae (C1) to the palatal plane, and the adenoid depth in children undergoing evaluation for velopharyngeal insufficiency. Methods: This retrospective cross-sectional study analyzed magnetic resonance imaging (MRI) scans of the velopharynx and measured the distance between C1 and the palatal plane, and the adenoid depth at the level of the palatal plane in millimeters. Results: Thirty-four patients met the inclusion criteria. The mean age at the time of MRI was 7.4 years (range: 3.9–11.9 y). The anterior tubercle of C1 was below the palatal plane in 97% (n = 33) of patients. On average, this landmark was 10.5 mm (SD = 5.0) below the palatal plane. Adenoid tissue was present at the level of the palatal plane in 91% (n = 31) of patients. Conclusions: Positioning the pharyngeal flap base at C1 is too low to aid with velopharyngeal closure. Further, adenoid tissue is frequently present at the level of velopharyngeal closure, limiting superior positioning of the pharyngeal flap base. When this occurs, surgeons should consider adenoidectomy before pharyngeal flap surgery. Preoperative MRI may be beneficial for planning pharyngeal flap positioning relative to C1 and assessing adenoid tissue at the palatal plane.

Purpose: The aim of this study was to investigate whether successful surgical management of velopharyngeal insufficiency (VPI) aids in the remediation of compensatory misarticulation errors (CMAs) among children with VPI and CMAs. Method: Fourteen participants with VPI and use of CMAs from a larger study were included in this retrospective cohort study. The mean age at the time of preoperative evaluation was 8.9 years ( SD = 1.1). Perceptual ratings of hypernasality, phonetic transcription, and anatomic measurements from magnetic resonance imaging were performed by raters blinded to the participants' medical and surgical history. The mean percentage of CMAs produced on the American English Sentence Sample was calculated. The Wilcoxon signed-ranks test was used to compare the change in CMA use pre- and postoperatively. During the study period, 71% ( n = 10) of participants received speech therapy. Results: Nine participants had resolved hypernasality after surgery, and five had persistent hypernasality. Among those with resolved hypernasality, the mean percentage of CMAs significantly decreased from 14.6% preoperatively to 1.1% postoperatively ( p = .028). For participants with persistent hypernasality, the mean percentage of CMAs decreased from 27.6% to 22%; this change was not significant ( p = .586). Conclusions: Correction of VPI may aid in the remediation of CMAs as participants have more normal anatomy to achieve velopharyngeal closure. These findings suggest correction of VPI may reduce the amount of speech therapy needed to treat CMAs.