Sitzman, Thomas J

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.

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.

Introduction The levator veli palatini (LVP) muscle has two segments with distinct roles in velopharyngeal function. Previous research suggests longer extravelar segments with shorter intravelar segments may lead to a more advantageous mechanism for velopharyngeal closure. The purpose of this study was to examine whether the distribution of the LVP intravelar and extravelar segments differs between children with cleft palate with and without VPI and controls. Methods The study included 97 children: 37 with cleft palate +/- lip with VPI, 37 controls, and 19 with cleft palate with normal resonance. Measures included mean LVP length, mean extravelar LVP length, and intravelar LVP length. Results Overall mean LVP length was similar ( P = .267) between controls and children with cleft palate (with and without VPI). However, there was a significant difference ( P < .001) between group for both intravelar and extravelar LVP lengths: the intravelar segment was significantly longer in those with VPI compared to controls and children with cleft palate and normal resonance; and the extravelar segment was significantly shorter in those with VPI compared to controls and children with cleft palate and normal resonance. Conclusions Results from this study demonstrate a significant difference between the distribution of the functional segments of the LVP among children with VPI, with a more disadvantageous distribution of the muscle segments among those with VPI.

Objective Compare the effectiveness of palatoplasty and pharyngoplasty procedures at resolving hypernasality in patients with 22q11.2 deletion syndrome (22q). Design Retrospective cohort study. Setting Metropolitan children's hospital. Patients Fourteen patients with 22q presenting for management of velopharyngeal insufficiency. Interventions Palatoplasty or pharyngoplasty procedure. Main Outcome Measure Resolution of hypernasality 12 months postoperatively. Results Both procedure groups had a mean preoperative velopharyngeal gap of 6.2 mm during phonation. No patient who underwent palatoplasty achieved resolution of hypernasality; 1/7 patients had worse hypernasality, 4/7 had no change, and 2/7 had improved hypernasality. In contrast, hypernasality was resolved in 6/7 patients in the pharyngoplasty group, which was significantly ( P = .03) higher than the palatoplasty group. Conclusions In patients with 22q, palatoplasty procedures may be less effective than pharyngoplasty procedures at resolving hypernasality. This may be due to underlying anatomic or physiologic differences, such as increased pharyngeal depth and hypodynamic muscles.

Objective To investigate whether flexible nasopharyngoscopy, when performed in addition to magnetic resonance imaging (MRI), influences the type of surgery selected or success of surgery in patients with velopharyngeal insufficiency (VPI). Design Cohort study. Setting A metropolitan children's hospital. Patients Patients with non-syndromic, repaired cleft palate presenting for management of VPI. Interventions MRI and nasopharyngoscopy or MRI alone for preoperative imaging of the velopharyngeal mechanism. Main Outcome Measures (1) Surgical selection and (2) resolution of hypernasality. All speech, MRI, and nasopharyngoscopy measurements were performed by raters blinded to patients’ medical and surgical history. Results Of the 25 patients referred for nasopharyngoscopy, 76% completed the exam. Of the 41 patients referred for MRI, the scan was successfully completed by 98% of patients. Completion of nasopharyngoscopy was significantly (p=0.01) lower than MRI. Surgical selection did not significantly differ (p=0.73) between the group receiving MRI and nasopharyngoscopy and the group receiving MRI alone, nor was there a significant difference between these groups in the proportion of patients achieving resolution of hypernasality postoperatively (p=0.63). Percent total velopharyngeal closure assessments on nasopharyngoscopy and MRI were strongly correlated (r=0.73). Conclusions In patients receiving MRI as part of their preoperative VPI evaluation, the addition of nasopharyngoscopy did not result in a difference in surgical selection or resolution of hypernasality. Routine inclusion of nasopharyngoscopy may not be necessary for the evaluation of velopharyngeal anatomy when MRI is available.

Background Collection of high-quality videorecorded speech samples is essential for speech outcomes research. Solution Cleft palate team SLPs were trained to collect standard videorecorded speech samples in the clinic setting across 20 sites in North America. Standard training and equipment was provided. What We Did That is New Quality management procedures were developed and utilized to verify video quality and protocol adherence. Over 97% of speech samples collected by trained SLPs met defined quality standards.

Objective Surgical treatment of velopharyngeal insufficiency (VPI) includes a wide array of procedures. The purpose of this study was to develop a classification for VPI procedures and to describe variations in how they are performed. Design/participants/setting/outcomes: We completed an in-depth review of the literature to develop a preliminary schema that encompassed existing VPI procedures. Forty-one cleft surgeons from twelve hospitals across the USA and Canada reviewed the schema and either confirmed that it encompassed all VPI procedures they performed or requested additions. Two surgeons then observed the conduct of the procedures by surgeons at each hospital. Standardized reports were completed with each visit to further explore the literature, refine the schema, and delineate the common and unique aspects of each surgeon's technique. Results Procedures were divided into three groups: palate-based surgery; pharynx-based surgery; and augmentation. Palate-based operations included straight line mucosal incision with intravelar veloplasty, double-opposing Z-plasty, and palate lengthening with buccal myomucosal flaps. Many surgeons blended maneuvers from these three techniques, so a more descriptive schema was developed classifying the maneuvers employed on the oral mucosa, nasal mucosa, and muscle. Pharynx-based surgery included pharyngeal flap and sphincter pharyngoplasty, with variations in design for each. Augmentation procedures included palate and posterior wall augmentation. Conclusions A comprehensive schema for VPI procedures was developed incorporating intentional adaptations in technique. There was substantial variation amongst surgeons in how each procedure was performed. The schema may enable more specific evaluations of surgical outcomes and exploration of the mechanisms through which these procedures improve speech.

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.