Factors Associated with Delayed Palatoplasty Before, During, and After the COVID-19 Pandemic.

BACKGROUND: Cleft palatoplasty is typically performed around 10 to 12 months of age in the US, and delays can negatively affect speech development. Early during COVID-19, elective surgeries were canceled. The aims of this study were to (1) identify overall risk factors for greater age at palatoplasty and (2) analyze delays in palatoplasty during COVID-19. METHODS: This study was part of a larger prospective, multicenter comparative study of speech outcomes in palatoplasty. Participants underwent palatoplasty between March 2019 and September 2022 at 18 pediatric hospitals in the United States. Ages were corrected for prematurity. Dates of palatoplasty were divided into 4 periods corresponding to different phases of the pandemic. Factors analyzed included region, language, adoption status, sex, ethnicity, race, rurality, health insurance type, and cleft type. Analyses were performed using ANOVA, Student's test, and multivariable linear regression, with a P value of ≤0.05 being significant. RESULTS: Nine hundred twenty-eight participants were included. Average corrected age at palatoplasty was 374 days. In univariable analysis, palatoplasty was performed later in children who were Hispanic (P=0.003), of a race other than White, Black, or Asian (P<0.001), and without private insurance (P<0.001). On multivariable regression, predictors of delayed palatoplasty were Hispanic ethnicity (P=0.015), from other race (P<0.001), and without private insurance (P<0.001). During COVID-19, disproportionate delays occurred in patients who were female, of other races, from nonrural areas, and on Medicaid. CONCLUSIONS: Palatoplasty was performed later in vulnerable populations. Some of these populations were also disproportionately affected by COVID-19 delays. Providers should be aware of these differences as they pertain to equitable access to craniofacial care. LEVEL OF EVIDENCE: III.

Risk of Malocclusion Among Patients Undergoing Single-Stage Versus Two-Stage Cleft Palate Repair.

OBJECTIVE: Evaluate impact of single-stage versus staged palate repair on the risk of developing malocclusion among patients with cleft palate (CP). DESIGN: Retrospective cohort study 2000-2016. SETTING: Academic, tertiary children's hospital. PATIENTS: Patients undergoing CP repair between 1999-2015. INTERVENTIONS: CP repair, categorized as either single-stage or staged. MAIN OUTCOME MEASURE: Time to development of Class III malocclusion. RESULTS: 967 patients were included; 60.1% had a two-stage CP repair, and 39.9% had single-stage. Malocclusion was diagnosed in 28.2% of patients. In the model examining all patients at ≤5 years (n = 659), patients who were not white had a higher risk of malocclusion (HR 2.46, p = 0.004) and staged repair was not protective against malocclusion (HR 0.98, p = 0.91). In all patients >5 years (n = 411), higher Veau classification and more recent year of birth were significantly associated with higher hazard rates (p < 0.05). Two-staged repair was not protective against developing malocclusion (HR 0.86, p = 0.60). In the model examining patients with staged repair ≤5 years old (n = 414), higher age at hard palate closure was associated with reduced malocclusion risk (HR 0.67, p < 0.001) and patients who were not white had increased risk (HR 2.56, p = 0.01). In patients with staged repair >5 years old, more recent birth year may be associated with a higher risk of malocclusion (HR 1.06, p = 0.06) while syndrome may be associated with lower risk of malocclusion diagnosis (HR 0.46, p = 0.07). CONCLUSION: Our data suggests that staged CP repair is not protective against developing Class III malocclusion.

Cleft Palate Repair Postoperative Management: Current Practices in the United States.

OBJECTIVE: To describe current postoperative management practices following cleft palate repair. DESIGN: A survey was administered to cleft surgeons to collect information on their demographic characteristics, surgical training, surgical practice, and postoperative management preferences. SETTING: Eighteen tertiary referral hospitals across the United States.Participants: Surgeons (n = 67) performing primary cleft palate repair. RESULTS: Postoperative diet restrictions were imposed by 92% of surgeons; pureed foods were allowed at one week after surgery by 90% of surgeons; a regular diet was allowed at one month by 80% of surgeons. Elbow immobilizers and/or mittens were used by 85% of surgeons, for a median duration of two weeks. There was significant disagreement about postoperative use of bottles (61% allow), sippy cups (68% allow), pacifiers (29% allow), and antibiotics (45% prescribe). Surgeon specialty was not associated with any aspect of postoperative management (p > 0.05 for all comparisons). Surgeon years in practice, a measure of surgeon experience, was associated only with sippy cup use (p < 0.01). The hospital at which the surgeon practiced was associated with diet restrictions (p < 0.01), bottle use (p < 0.01), and use of elbow immobilizers or mittens (p < 0.01); however, many hospitals still had disagreement among their surgeons. CONCLUSIONS: Surgeons broadly agree on diet restrictions and the use of elbow immobilizers or mittens following palate repair. Almost all other aspects of postoperative management, including the type and duration of diet restriction as well as the duration of immobilizer use, are highly individualized.

Management of Velopharyngeal Insufficiency in Cleft Patients With and Without Multidisciplinary Team Care.

OBJECTIVE: To evaluate the effect of an American Cleft Palate-Craniofacial Association (ACPA)-approved multidisciplinary team on velopharyngeal insufficiency (VPI) diagnosis and treatment. DESIGN: Retrospective cohort setting; tertiary children's hospital patients; children with cleft palate repair identified through procedure codes. MAIN OUTCOME MEASURES: Velopharyngeal insufficiency diagnosis was assigned based on surgeon or team assessment. Age at diagnosis and surgery was recorded. Difference in age and rate of VPI diagnosis and surgery was analyzed with t test. Multivariate linear and logistic regression adjusted for confounding variables. RESULTS: Nine hundred forty patients were included with 71.5% cared for by an ACPA-approved multidisciplinary team. More (38.8% ) team care patients were found to have a diagnosis of VPI in comparison to 10% in independent care (P < .001). Team care was associated with an almost 6-fold increase in VPI diagnosis (P < .001). Team care was associated with a higher proportion of speech surgery (21% vs 10%, P < .001). Among children receiving team care, each visit was associated with 25% increased odds of being diagnosed with VPI (P < .001) and 20% increased odds of receiving speech surgery (P < .001). Age at VPI diagnosis and speech surgery were similar between groups (P = .55 and .29). DISCUSSION: Team care was associated with more accurate detection of VPI, resulting in more VPI speech therapy visits and surgical management. A higher number of team visits were similarly associated. CONCLUSION: Further studies of the clinical implication of timely and accurate VPI diagnosis, including quality of life assessments, are recommended to provide stronger guidance on team visit and evaluation planning.

Oronasal Fistula Risk After Palate Repair.

OBJECTIVE: To assess risk factors for oronasal fistula, including 2-stage palate repair. DESIGN: Retrospective analysis. SETTING: Tertiary children's hospital. PATIENTS: Patients with non-submucosal cleft palate whose entire cleft repair was completed at the study hospital between 2005 and 2013 with postsurgical follow-up. INTERVENTIONS: Hierarchical binary logistic regression assessed predictive value of variables for fistula. Variables tested for inclusion were 2 stage repair, Veau classification, sex, age at surgery 1, age at surgery 2, surgeon volume, surgeon, insurance status, socioeconomic status, and syndrome. Variables were added to the model in order of significance and retained if significant at a .05 level. MAIN OUTCOME MEASURE: Postoperative fistula. RESULTS: Of 584 palate repairs, 505 (87%) had follow-up, with an overall fistula rate of 10.1% (n = 51). Among single-stage repairs (n = 211), the fistula rate was 6.7%; it was 12.6% in 2-stage repairs (n = 294, P = .03). In the final model utilizing both single-stage and 2-stage patient data, significant predictors of fistula were 2-stage repair (odds ratio [OR]: 2.5, P = .012), surgeon volume, and surgeon. When examining only single-stage patients, higher surgeon volume was protective against fistula. In the model examining 2-stage patients, surgeon and age at hard palate repair were significant; older age at hard palate closure was protective for fistula, with an OR of 0.82 (P = .046) for each additional 6 months in age at repair. CONCLUSIONS: Two-stage surgery, surgeon, and surgeon volume were significant predictors of fistula occurrence in all children, and older age at hard palate repair was protective in those with 2-stage repair.

Family Experience With Pierre Robin Sequence: A Qualitative Study.

OBJECTIVE: To identify concepts and constructs important to parents of children with Pierre Robin Sequence (PRS). DESIGN: Qualitative study. SETTING: All children received some care at a tertiary hospital with additional care at outside facilities. Interviews were conducted in nonclinical locations, including remote locations. PARTICIPANTS: Parents of children <5 years old with a diagnosis of PRS. Prior treatments included observation, positioning, nasal trumpet, mandibular distraction osteogenesis, tracheostomy, and gastrostomy. INTERVENTION: Semi-structured interviews with individuals (4) and with groups (focus groups, 4) were conducted using open-ended questions and non-leading prompts. Transcripts were analyzed with iterative open and axial coding. Concepts and constructs were identified and refined into codes and central themes. Interviews were conducted until thematic saturation was achieved. RESULTS: Sixteen parents were interviewed. Their experiences were coded into 5 main themes, which can be summarized as: (1) child's symptoms/well-being, (2) parents' grief/isolation, (3) family stress, (4) relationships with providers, and (5) psychological and technical growth. Difficulty with feeding, weight gain, and breathing problems were core physical issues described by participants with associated intense fear. Participants described frustration from not only lack of care coordination, slow diagnoses, and poor communication but also gratitude for providers who served as advocates. Participants described gradual development of knowledge/competencies. CONCLUSIONS: Families of children with PRS have experiences that profoundly affect their lives. Child's physical symptoms/well-being and parents' psychosocial well-being provide content for a future PRS-specific quality-of-life instrument. Concepts that emerged also provide a framework to improve parents' experience and enhance their children's quality of care.

Increased Risk of Velopharyngeal Insufficiency in Patients Undergoing Staged Palate Repair.

OBJECTIVE: To evaluate the association of 2-stage cleft palate (CP) surgery on velopharyngeal insufficiency (VPI) incidence, speech surgeries, and cleft-related surgical burden. DESIGN: Retrospective cohort with follow-up of 4 to 19 years. SETTING: Academic, tertiary children's hospital. PATIENTS: Patients who underwent CP surgery between 2000 and 2017. Exclusions included submucous CP or age at last contact under 3.9. INTERVENTIONS: Cleft palate surgery, completed in either a single-stage or 2-stage repair. MAIN OUTCOME MEASURE(S): Rates of VPI diagnosis and speech surgery and total cleft surgeries; t tests, tests of proportion, and linear and logistic regression were performed. Total cleft-related surgeries were examined in a subset (n = 418) of patients with chart reviews. RESULTS: A total of 1047 patients were included; 59.6% had 2-stage CP repair, 40.4% had single-stage repair. Approximately 32% of children with 2-stage CP repair were diagnosed with VPI, as opposed to 22% of single-stage patients (P < .001). Children with 2-stage CP repair were 1.8 times as likely to be diagnosed with VPI (P < .001). Speech surgery rates were similar across groups. Patients who had 2-stage repair received an average of 2.3 more cleft-related procedures, when excluding prosthesis management procedures. CONCLUSION: Our data show an increased risk of VPI diagnosis and increased surgical burden among patients receiving 2-stage CP repair.

Ecuadorian Spanish translation and validation of the VELO quality of life instrument.

OBJECTIVES: Adapt the Spanish translation of VPI Effects on Life Outcome (VELO) instrument into Ecuadorian Spanish; test the resulting instrument for reliability and validity. METHODS: A cross-sectional, prospective design, set at a humanitarian mission within a community hospital. Linguistic validation: native Ecuadorian-Spanish speakers modified the Spanish VELO to Ecuadorian Spanish. Cognitive interviews were conducted with children with cleft palate (CP) and their parents (n = 50), guiding instrument modifications. An expert panel reviewed changes, resulting in the VELO-Ecuadorian dialect (VELO-Ec). INSTRUMENT ASSESSMENT: 88 participants with CP (88 parents, 46 children) and 33 non-cleft controls (33 adult, 11 children) completed the VELO-Ec, Spanish-Pediatric Voice Handicap Index (pVHI), and Spanish-Intelligibility in Context Scale (ICS). Internal consistency was assessed with Cronbach's alpha; test-retest reliability was assessed by calculating the intraclass correlation coefficient (ICC); standard error of measurement (SEM) was calculated. Concurrent validity was assessed with Pearson correlations of VELO-Ec with pVHI and ICS. Discriminant validity assessment used an established ICS cutoff. Construct validity was assessed by grouping patients by parent report of hypernasality and early vs. late cleft repair (>24 months) using the Wilcoxon Rank-Sum test. RESULTS: VELO-Ec showed excellent internal consistency (alpha 0.96) and test-retest reliability (ICC = 0.85, 95% CI 0.68-0.93, SEM 5.71). It had strong concurrent validity, correlating with ICS (r = 0.75, p < 0.001) and pVHI (r = -0.79, p < 0.001). Discriminant validity was strong with better VELO-Ec scores among subjects with normal vs. abnormal ICS score (median 95 & 61, p < 0.001). Strong construct validity was identified: those with parent-reported hypernasality had worse VELO-Ec scores than those without (median 59 & 75, p < 0.001). Those with repair before or after 24 months had similar VELO-Ec scores (p = 0.882). CONCLUSION: The VELO-Ec is a valid and reliable measure of VPI-related quality of life, useful to clinicians and researchers treating Ecuadorian CP patients, especially in areas with limited resources such as on humanitarian missions.

Male exposure to bisphenol A (BPA) and semen quality in the Home Observation of Periconceptional Exposures (HOPE) cohort.

BACKGROUND: Exposure to bis-phenol A (BPA) has been associated with reduced semen quality. The objective of this study was to examine associations between BPA measured in serial daily first-morning urine samples and semen quality parameters among men trying to conceive. METHODS: This prospective, preconception cohort included 161 men ages 18-40 without known subfertility. Men collected daily, first morning urine during their female partner's fertile window. Semen samples were collected through intercourse after the fertile window. RESULTS: Samples from 161 men were analyzed. Higher geometric mean (GM) BPA exposures (ng/mL) were found among men with abnormal sperm tail morphology (GM = 3.12, 95% CI = 2.43, 4.01) compared to men with normal morphologic findings (GM = 2.39, 95% CI = 2.17, 2.74). There was no association with sperm count. CONCLUSION: Higher exposure to BPA was associated with abnormal sperm tail morphology in this prospective, pre-conception cohort.