Impact of Neighborhood Deprivation and Social Vulnerability on Outcomes and Interventions in Patients with Cleft Palate.

OBJECTIVE: To examine whether neighborhood disadvantage impacts length of follow-up, interventions, and outcomes for patients with cleft palate. DESIGN: Retrospective cohort. SETTING: Cleft Palate Craniofacial Institute Database at St. Louis Children's Hospital. PATIENTS/PARTICIPANTS: Patients with cleft palate following in St. Louis Children's Hospital Cleft Palate Multidisciplinary Team Clinic. INTERVENTIONS: Primary palatoplasty between 2012-2017. Patients were divided into quartiles across area deprivation index (ADI) and social vulnerability index (SVI), two validated, composite metrics of neighborhood disadvantage, to examine whether living in neighborhoods from different deprivation quartiles impacts outcomes of interest. MAIN OUTCOME MEASURE: Follow-up through age 5, surgeries and surgical complications, speech, developmental, and behavioral outcomes. RESULTS: 205 patients were included. 39% of patients belonged to the most deprived ADI quartile, while 15% belonged to the most vulnerable SVI quartile. There were no differences between ADI or SVI quartiles in number of operations received (p ≥ 0.40). Patients in the most deprived ADI quartile were significantly more likely to have speech/language concerns (OR 2.32, 95% CI [1.20-4.89], p = 0.01). Being in a more vulnerable SVI quartile was associated with developmental delay (OR 2.29, 95% CI [1.04-5.15], p = 0.04). ADI and SVI quartile did not impact risk of loss to follow-up in the isolated and combined cleft lip and palate subgroups (p ≥ 0.21). CONCLUSIONS: Neighborhood disadvantage impacts speech and developmental outcomes in patients with cleft palate despite comparable length of follow-up in multidisciplinary team clinic.

Impact of a Palatal Fistula After Cleft Palate Repair on Velopharyngeal Closure.

PURPOSE: A palatal fistula is an adverse outcome of cleft palate repair. It is unknown if a palatal fistula will influence velopharyngeal closure, even after repair of the fistula. This study determines the effect of a soft palate fistula on the risk of developing velopharyngeal insufficiency. METHODS: A retrospective chart review was conducted on patients who underwent primary cleft palate repair between 2000 and 2015, with complete records at 4 years of age. Fistulae involving the secondary palate following primary palatoplasty were classified as the soft or hard palate. A forced-entry multivariate logistic regression model was built to detect predictors of velopharyngeal dysfunction. RESULTS: Records of 329 patients were analyzed with a mean follow-up of 8.7 years. A palatal fistula was identified in 89/329 patients (27%) and 29/329 patients (9%) underwent an independent fistula repair. Of the patients with fistula, 44% were located in the hard palate only and 56% had soft palate involvement. Compared to patients without a fistula, rates of velopharyngeal dysfunction were significantly higher in patients with a fistula involving the soft palate (OR 3.875, CI: 1.964-7.648, P < .001) but not in patients with a hard palate fistula (OR 1.140, CI: 0.497-2.613, P = .757). Veau class, age at primary repair, and syndromic status were not significant predictors of VPI (0.128≤P ≤ .975). CONCLUSIONS: A palatal fistula involving the soft palate is a significant predictor for development of velopharyngeal dysfunction after primary palatoplasty. Surgical intervention, at the time of fistula repair, to add vascularized tissue may be indicated to prophylactically decrease the risk of velopharyngeal dysfunction.

Multidisciplinary Optimal Outcomes Reporting and Team Clinic Retention in Isolated Nonsyndromic Cleft Palate.

OBJECTIVE: Optimal Outcomes Reporting was recently introduced to categorize outcomes after cleft palate repair. We seek to propose an expanded version of Optimal Outcomes Reporting and to determine if correlation exists between the expanded outcomes and persistence with team care follow-up through age 9. DESIGN: Retrospective cohort study. SETTING: Cleft team at large pediatric hospital. PATIENTS: Patients with isolated nonsyndromic cleft palate (n = 83) born from 2001-2012. MAIN OUTCOME MEASURES: Patients who continued to present at age 5 or greater were assessed for optimal outcomes. Optimal outcomes were: surgery - no fistula or velopharyngeal insufficiency; otolaryngology - no obstructive sleep apnea or signs of chronic middle ear disease; audiology - no hearing loss; speech-language pathology - no assessed need for speech therapy. RESULTS: Of the 83 patients identified, 41 were assessed for optimal outcomes. Optimal outcome in any discipline was not associated with follow-up through age 9 (0.112 ≤ p ≤ 0.999). For all disciplines, the group with suboptimal outcomes had a higher proportion of patients from geographic areas in the most disadvantaged quartile of social vulnerability index, with the strongest association in the group with suboptimal speech outcome (OR 6.75, 95% CI 0.841-81.1). CONCLUSIONS: Optimal outcomes and retention in team clinic were not statistically significantly associated, but clinically relevant associations were found between patients in the most disadvantaged quartile of social vulnerability and their outcomes. A patient-centered approach, including caregiver education about long-term care for patients with cleft palate, would allow for enhanced resource utilization to improve retention for patients of concern.

Buccal Myomucosal Flap Repair for Velopharyngeal Dysfunction.

BACKGROUND: Velopharyngeal dysfunction (VPD) is the incomplete separation of the nasal and oral cavities during speech sound production that can persist following primary palatoplasty. Surgical technique used in management of VPD (palatal re-repair versus pharyngeal flap or sphincter pharyngoplasty) is often dictated by the preoperative velar closing ratio and closure pattern. Recently, buccal flaps have increased in popularity in management of VPD. Here, the authors investigate the effectiveness of buccal myomucosal flaps in the treatment of VPD. METHODS: A retrospective review was performed of all patients undergoing secondary palatoplasty with buccal flaps at a single center between 2016 and 2021. Preoperative and postoperative speech outcomes were compared. Speech assessments included perceptual examinations, graded on a four-point scale of hypernasality, and speech videofluoroscopy, from which the velar closing ratio was obtained. RESULTS: A total of 25 patients underwent buccal myomucosal flap procedures for VPD at a median of 7.1 years after primary palatoplasty. Patients had significantly increased velar closing postoperatively (95% versus 50%; P < 0.001) and improved speech scores ( P < 0.001). Three patients (12%) had continued hypernasality postoperatively. There were no occurrences of obstructive sleep apnea. CONCLUSIONS: Treatment of VPD with buccal myomucosal flaps leads to improved speech outcomes without the risk of obstructive sleep apnea. Traditionally, palatal re-repair techniques have been used for smaller preoperative velopharyngeal gaps; however, the addition of buccal flaps allows for anatomical velar muscle correction for patients with a larger preoperative velopharyngeal gap. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Evaluation of Discipline-Specific Outcomes Through a Multidisciplinary Team Clinic for Patients With Isolated Cleft Palate.

OBJECTIVE: To describe the incidence and timing of provider-specific interventions for children with isolated cleft palate. DESIGN: This was a retrospective cohort study involving review of medical records. SETTING: Multidisciplinary team care clinic at a tertiary academic children's hospital between January 2000 and July 2019. PATIENTS: Patients with isolated nonsyndromic cleft palate seen by an American Cleft Palate-Craniofacial Association-approved team; 138 children were included. MAIN OUTCOME MEASURES: Study outcomes included incidence of secondary velopharyngeal management, tympanostomy tube insertion, speech therapy, hearing loss, dental/orthodontic treatment, and psychology interventions. Provider-specific outcomes were calculated for patients at ages 0 to 3, 3 to 5, and >5 years. RESULTS: Median follow-up time was 7.0 years (interquartile range: 3.3-11.8 years). At their last team assessment, 42% of patients still had conductive hearing loss. The rate of tympanostomy tube insertions not done alongside a palatoplasty was highest for ages 3 to 5 and dropped after new American Academy of Otolaryngology-Head and Neck Surgery Foundation guidelines in 2013 (P = .015); 54% of patients received speech-language therapy during follow-up. Palatoplasty, psychology, and dental/orthodontic treatment were all less common than speech or ENT treatment (P < .01). Secondary palatoplasty was performed in 31 patients (22%). Patients who received speech, dental/orthodontic, or psychology intervention followed up longer than those who did not (9.8 vs 2.1 years, P < .001). CONCLUSION: Half of the patients terminated team follow-up by age 7, suggesting that burden of care outweighed perceived benefits of continued follow-up for many families. These results can be used to adjust protocols for children with isolated cleft palate.

The Reliability of Visual Ratings of Velopharyngeal Physiology for Speech.

BACKGROUND: Analyze intrarater and interrater reliability for evaluating endoscopic images of velopharyngeal (VP) physiology. METHOD: Speakers produced 9 speech stimuli representing 4 stimulus types: sustained phonemes, repetitions of "puh," single words, and short phrases. The 37-speaker participants included 16 patients with VP dysfunction and 21 control participants. Five raters independently rated the video images for degree of VP opening, location of opening, and pattern of closure. Outcome measures included intrarater and interrater measures of reliability and the effects of raters and stimulus type on ratings. RESULTS: Intrarater reliability was acceptable, and ratings were logically consistent. Fixed effects regression coefficients for the patient and the control groups showed that raters were a significant source of variability for degree of opening and pattern of closing. Stimulus type was not a significant source of variation for any metric for the controls, but stimulus type was a significant determinant for degree of opening for patients. The degree of opening was larger for sustained phonemes than for the other speech stimuli. Ratings for degree of opening were most similar for repeated "puh." CONCLUSIONS: Interrater reliability needs to be improved so that the assessment procedure produces more consistent findings among clinicians, thus strengthening our evidence base for this procedure. Interrater additional research is needed to understand how the stimulus affects ratings of VP physiology, to identify stimuli that yield the most useful clinical information, and to understand how training affects the ratings of VP physiology.

Evaluation of Multidisciplinary Team Clinic for Patients With Isolated Cleft Lip.

OBJECTIVE: To report the incidences of secondary lip and nose operations, otolaryngology procedures, speech-language therapy, neurodevelopmental concerns, and dental and orthodontic issues in children with isolated cleft lip to inform multidisciplinary cleft team protocols. SETTING: An American Cleft Palate-Craniofacial Association-approved team at a tertiary academic children's hospital. DESIGN: Retrospective cohort study of patients evaluated through longitudinal clinic visits by a multidisciplinary cleft palate and craniofacial team between January 2000 and June 2018. PATIENTS, PARTICIPANTS: Children with nonsyndromic cleft lip with or without cleft alveolus (n = 92). RESULTS: Median age at final team visit was 4.9 years (interquartile range: 2.4-8.2 years). Secondary plastic surgery procedures were most common between ages 3 and 5 (135 per 1000 person-years), and the majority of these procedures were minor lip revisions. The rate of tympanostomy tube insertion was highest before age 3 (122 per 1000 person-years). By their final team visit, 88% of patients had normal hearing and 11% had only slight to mild conductive hearing loss. No patients had speech errors attributable to lip abnormalities. Psychological interventions, learning disabilities, and dental or orthodontic concerns were uncommon. CONCLUSIONS: Most patients with isolated cleft lip may not require long-term, longitudinal evaluation by cleft team specialists. Cleft teams should develop limited follow-up protocols for these children to improve resource allocation and promote value-based care in this patient population.

Progressive Tightening of the Levator Veli Palatini Muscle Improves Velopharyngeal Dysfunction in Early Outcomes of Primary Palatoplasty.

BACKGROUND: Management of the levator veli palatini with intravelar veloplasty has been shown to improve speech resonance. The senior author has introduced a more aggressive procedure where the levator is separately dissected, overlapped, and tightened. This study compares resonance results from four levator management protocols: non-intravelar veloplasty, Kriens intravelar veloplasty, radical intravelar veloplasty, and overlapping intravelar veloplasty. METHODS: Retrospective chart review was conducted on 252 patients who underwent primary palatoplasty with speech follow-up at 3 years of age. Velopharyngeal function was evaluated with perceptual speech examinations, and subjects were scored on a four-point scale (0 = normal resonance; 1 = occasional hypernasality/nasal emission/turbulence/grimacing, no further assessment warranted; 2 = mild hypernasality/intermittent nasal turbulence/grimacing, velopharyngeal imaging suggested; and 3 = severe hypernasality, surgical intervention recommended). Fisher's exact test was used to compare outcomes. RESULTS: A single surgeon performed all the non-intravelar veloplasty (n = 92), Kriens intravelar veloplasty (n = 103), and radical intravelar veloplasty (n = 31), whereas the senior author performed the overlapping intravelar veloplasty (n = 26). Cleft severity proportions were equivalent across the four methods (p = 0.28). Patients who underwent overlapping intravelar veloplasty demonstrated significantly better velopharyngeal function, and none required further imaging or secondary surgery compared with the other three procedures (p < 0.001). CONCLUSIONS: Speech resonance outcomes at 3 years of age are improved and need for secondary velopharyngeal dysfunction management is reduced with more aggressive levator dissection and reconstruction during primary one-stage palatoplasty. Results were best when the muscle was overlapped. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Evaluation of two palate repair techniques for the surgical management of velopharyngeal insufficiency.

BACKGROUND: The Furlow palatoplasty is commonly used for the correction of velopharyngeal insufficiency in cleft patients. An alternative procedure is introduced involving a single Z-plasty with overlapping intravelar veloplasty (Woo palatoplasty). This study compared the results of both techniques in the correction of velopharyngeal insufficiency. METHODS: After institutional review board approval, a retrospective chart review was performed of all patients who had undergone secondary palatoplasty for the correction of velopharyngeal insufficiency. All nonsyndromic patients with imaging data were evaluated. Data elements included preoperative and postoperative velopharyngeal gap size and perceptual speech examination results. RESULTS: Fifty-two subjects were included: 30 subjects had undergone Furlow palatoplasty and 22 underwent Woo palatoplasty. Overall, a larger proportion of Woo (95 percent) than Furlow subjects (63 percent) did not require secondary surgery (p = 0.005). However, mean presurgery closure was significantly different between groups (p = 0.042). For a more refined assessment, only those with 80 percent or greater preoperative closure were evaluated. Successful results were achieved in 67 percent (10 of 15) in Furlow and in 100 percent (19 of 19) in Woo. Again, this finding was significant (p = 0.005). Linear regression analysis suggested a significant effect of cleft type (ß = 2.3, p = 0.013) on closure after repair, with decreased closure in cases with isolated cleft palate. CONCLUSIONS: The Woo palatoplasty compared favorably with Furlow palatoplasty for correction of velopharyngeal insufficiency. The technique appears to be a viable alternative for palatal re-repair, especially in circumstances when Furlow palatoplasty cannot be performed. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Effects of aural atresia on speech development and learning: retrospective analysis from a multidisciplinary craniofacial clinic.

IMPORTANCE Aural atresia (AA) is associated with maximal conductive hearing loss in affected ears, and children with bilateral AA require amplification. Some recent research has suggested an increased risk for speech and learning problems among children with unilateral sensorineural hearing loss. OBJECTIVE To investigate whether increased risk for speech and learning problems exists among children with AA. DESIGN Retrospective medical record review. SETTING Multidisciplinary craniofacial clinic. PARTICIPANTS Children with unilateral or bilateral AA. INTERVENTIONS Records review, including evaluations by audiologists, speech pathologists, and psychologists. MAIN OUTCOME MEASURES Rates of speech and/or language delay, prevalence of speech therapy and educational interventions, and parental report of psychosocial problems. RESULTS A total of 74 patients were identified who met inclusion and exclusion criteria: 48 with right-sided AA, 19 with left-sided AA, and 7 with bilateral AA. Children with AA demonstrated high rates of speech therapy (86% among bilateral, 43% among unilateral). Reports of school problems were more common among children with right-sided AA (31%) than those with left-sided AA (11%) or bilateral AA (0%) (P = .06). Educational interventions were common in all groups (33% right, 21% left, 43% bilateral). In the case of bilateral AA, all children who received additional interventions were enrolled in schools for the hearing impaired, without any identified learning deficiencies. CONCLUSIONS AND RELEVANCE Children with unilateral AA may be at greater risk of speech and learning difficulties than previously appreciated, similar to children with unilateral sensorineural hearing loss. Whether amplification may alleviate this risk is unclear and warrants further study.

The effect of timing of surgery for velopharyngeal dysfunction on speech.

The timing of surgery for velopharyngeal dysfunction has been based on assumptions about the relation between age, speech development, and velopharyngeal dysfunction. Cleft palate teams often counsel parents to have an intervention for velopharyngeal dysfunction performed earlier rather than later, believing that earlier interventions result in more rapid or better normalization of speech. The objective of this retrospective chart review study is to determine whether the age at surgical intervention for velopharyngeal dysfunction has an effect on the subsequent length of speech therapy. Of 174 patients included in the study database, 36 had velopharyngeal dysfunction for which further velopharyngeal management was required. Of the 36 patients who received surgical velopharyngeal dysfunction management, 27 had verifiable speech therapy records. These 27 patients represent the study population. The outcome measure was the total length of subsequent speech therapy until speech normalization. The data suggest that there is no relation between the age at velopharyngeal dysfunction surgical management and the amount of speech therapy needed to achieve normalization of the speech impairments secondary to velopharyngeal dysfunction after that management. In conclusion, 1) the age at surgical velopharyngeal dysfunction management (pharyngeal flap or sphincter pharyngoplasty) does not have an effect on subsequent normalization of speech as measured by the duration of speech therapy necessary to achieve normalization of the speech impairments secondary to velopharyngeal dysfunction after that management, and 2) the age at surgical velopharyngeal dysfunction management does not affect the likelihood of subsequent surgical velopharyngeal dysfunction management procedures.