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.

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.

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.

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.

Objective Describe surgical decision making and outcomes in a series of patients with persistent VPI after pharyngeal flap placement that were all treated with revision palatoplasty. Design Retrospective, case series. Participants Five patients with nonsyndromic cleft palate and persistent hypernasality following a pharyngeal flap. On MRI, 3 patients presented with an incohesive levator veli palatini muscle, and 4 patients had a pharyngeal flap that was below the palatal plane. Interventions One patient underwent straight-line intravelar veloplasty (IVVP), 2 patients underwent pharyngeal flap take-down and IVVP, and 2 patients underwent pharyngeal flap take-down and palate lengthening with buccal myomucosal flaps. Main Outcome Measure Pre- and postoperative resonance. Results Four of five patients (80%) achieved normal resonance 12 months postoperation. Conclusions Revision palatoplasty may be an effective approach for treating patients with persistent hypernasality following a pharyngeal flap. MRI may aid in surgical selection based on patient-specific anatomical findings.

Objective Evaluate the effectiveness of a digital home monitoring program for infants with cleft palate with or without cleft lip (CP ± L), compared to monitoring through in-person clinic visits. Design Retrospective cohort study. Setting One metropolitan pediatric hospital. Patients Eight-eight infants with CP ± L: 41 infants received digital home monitoring and 47 infants were monitored solely through in-person visits. Interventions Beginning in September 2022, all infants with CP ± L were enrolled in a digital home monitoring program, in which caregivers weighed their child weekly at home and submitted those weights, along with subjective evaluations of their child's feeding, using a secure website. Submissions were monitored by the cleft team nurse coordinator. Main Outcome Measures The primary outcome was the incidence of malnutrition at 4 months of age. The secondary outcome was the average number of in-person clinic visits during the first 4 months of life. Results There was not a significant difference ( P = .764) in the incidence of malnutrition between infants monitored in-person compared to infants in the home monitoring program (13% vs 17%). Infants in the home monitoring program had fewer in-person visits with speech–language pathology (5.4 vs 3.9; P < .001). Across groups, malnutrition was associated with Child Protective Services involvement ( P = .001) and presence of a syndrome ( P = .014). Conclusions The digital home monitoring program did not decrease the incidence of malnutrition, but it did reduce the number of speech–language pathology in-person visits. The program appears to distinguish infants who are gaining weight appropriately from those with feeding challenges, but it does not fully address the multifactorial contributors to malnutrition.

Objective To assess the quality of nasopharyngoscopy video recordings used for velopharyngeal insufficiency (VPI) surgical planning and identify factors that limit evaluation of velopharyngeal closure. Design Prospective observational study. Setting Metropolitan-based hospitals with craniofacial clinics in the United States and Canada. Participants One-hundred and forty-two (142) patients with VPI across 10 hospitals. Assessment(s) Nasopharyngoscopy video recordings used for VPI surgical planning . Main Outcome Measure(s) Ratability of nasopharyngoscopy video recordings, with “ratable” defined as the video (1) visualized the velum, lateral pharyngeal wall, and posterior pharyngeal walls at some point during speech production and (2) contained an oral speech sample at the phrase level or above. Results One-hundred and forty-two (142) nasopharyngoscopy video recordings were obtained from patients undergoing VPI evaluation, of which 59.9% (n=85) were ratable. A multilevel logistic regression model was used to identify factors that influenced the quality of nasopharyngoscopy video recordings. Factors associated with unratable nasopharyngoscopy videos were age ( P =.030), sex ( P =.005*), type of scope camera used ( P =.039), presence of compensatory misarticulations ( P =.008), and a limited speech sample ( P =.040). Conclusions A substantial proportion of nasopharyngoscopy video recordings obtained during VPI evaluation are not sufficient for rating velopharyngeal closure. Lack of ratability could impact the surgery selected to treat VPI. Younger patients, those with limited speech samples, or patients with extensive compensatory articulations may be more successful in completing other VPI imaging techniques, such as videofluoroscopy or magnetic resonance imaging.