Citations

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.

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 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.

Purpose: The purpose of this study was to investigate the relationship between perceptual ratings of hypernasality made during connected speech and velopharyngeal (VP) gap size measured in millimeters in the sagittal plane during sustained vowel production using magnetic resonance imaging (MRI). Method: A retrospective cross-sectional analysis was completed. A subgroup of 110 participants from another study with an M age of 10.1 years presenting for management of VP insufficiency was included. Perceptual ratings of hypernasality during connected speech and measurement of gap size during sustained /i/ production on MRI were performed by raters blinded to the participants' medical and surgical history. Results: There was a moderate-to-strong, positive correlation ( r = .61; p < .001) between hypernasality ratings and VP gap size measured on MRI using sustained /i/. The odds of a higher hypernasality rating increased as the gap size increased (odds ratio = 1.34; 95% CI [1.20, 1.49]; p < .001). The predicted probability for hypernasality ratings of none/minimal/mild steadily decreased as the gap size increased indicating that lower ratings of hypernasality were associated with smaller gap sizes. For the rating of “moderate” hypernasality, the predicted probability of the rating steadily increased up to 8 mm and then decreased as the gap size continued to increase. The predicted probability for a hypernasality rating of “severe” consistently increased as the gap size increased. Conclusions: Hypernasality ratings made at the connected speech level were significantly associated with VP gap size as measured during sustained vowel production. These findings suggest sustained vowel production elicited on MRI may adequately characterize VP gap size in the evaluation of VP insufficiency.

Assess agreement of hypernasality and audible nasal emission (ANE) ratings between audio-recordings and a clinic setting. Cross-sectional study using retrospective clinical recordings. Audio-recording ratings by two trained speech language pathologists. Percent agreement and intra- and inter-rater reliability of perceptual ratings. Intra-rater reliability (AC2) of 167 audio-recorded speech samples for the primary and secondary raters, respectively, was 0.82 and 0.79 for hypernasality; for ANE, it was 0.57 and 0.75. Inter-rater reliability was 0.77 for hypernasality and 0.63 for ANE. When comparing ratings made from audio-recording versus the original clinical ratings, intra-rater reliability was 0.85 and 0.61 (primary and secondary rater, respectively) for hypernasality and 0.21 and 0.34 for ANE. Ratings for hypernasality made from audio recordings were consistent with clinical evaluation, while ratings of ANE were not. ANE ratings made from audio recordings may not be a valid measure of velopharyngeal insufficiency speech characteristics.

Background: Palate re-repair has been proposed as an effective treatment for velopharyngeal insufficiency (VPI) with a low risk of obstructive sleep apnea (OSA). The authors conducted a systematic review and meta-analysis to determine the proportion of patients achieving normal speech resonance following palate re-repair for VPI, the proportion developing OSA, and the criteria for patient selection that are associated with increased effectiveness. Methods: PubMed, Embase, and Scopus were searched from inception through April 2018 for English language articles evaluating palate re-repair for the treatment of VPI in patients with a repaired cleft palate. Inclusion criteria included reporting of hypernasality, nasal air emission, nasometry, additional VPI surgery, and/or OSA outcomes. Meta-analysis was conducted using random effects models. Risk of bias was assessed regarding criteria for patient selection, blinding of outcome assessors, and validity of speech assessment scale. Results: Eighteen studies met inclusion criteria. The incidence of achieving no consistent hypernasality follow palate re-repair was 61% (95% confidence interval [CI]: 44%-75%). The incidence of additional surgery for persistent VPI symptoms was 21% (95% CI: 12%-33%). The incidence of OSA was 28% (95% CI: 13%-49%). Criteria for selecting patients to undergo re-repair varied, with anterior/sagittal position of palatal muscles (33%) and small velopharyngeal gap (22%) being the most common. No specific patient selection criteria led to superior speech outcomes ( P = .6572). Conclusions: Palate re-repair achieves normal speech resonance in many but not all patients with VPI. Further research is needed to identify the specific examination and imaging findings that predict successful correction of VPI with re-repair.