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.

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.

Brazilian-Portuguese Linguistic Validation of the Velopharyngeal Insufficiency Effects on Life Outcome Instrument.

BACKGROUND: The purpose of this study was to conduct a linguistic validation of the velopharyngeal insufficiency (VPI) Effects on Life Outcome (VELO) instrument for use in Brazilian-Portuguese patients with VPI. METHODS: The original English version of the VELO instrument was translated into Brazilian-Portuguese, back-translated, and adapted among the Brazilian patients (n = 21) with VPI and their parents, based on the standardized guidelines for the cross-culture adaption process. Discrepancies in the forward and backward translation steps were computed. Comprehension rates were captured for each debriefing interview. The content validity index (CVI) per item (I-CVI) and of the scale (S-CVI universal agreement [S-CVI/UA] and averaging [S-CVI/Ave]) were calculated. RESULTS: Reconciliation of the 2 forward translations and the comparison between the back translation and the original VELO version resulted in some item wordings with discrepancies which were reviewed by the research team (translators, expert committee, and original developers of instrument). Three rounds of cognitive interviews also led to some revisions of wording. Comprehension rates of patients and their parents were 60% to 100%, 80% to 100%, and 100% in the first, second, and third rounds of cognitive interviews, respectively. The I-CVI, S-CVI/AU, and SCI/Ave for the Brazilian-Portuguese VELO version were 0.83 (or higher), 0.83 (or higher), and 0.97, respectively. CONCLUSIONS: The linguistic validation process of the VELO instrument created a cross-culturally equivalent Brazilian-Portuguese version for use in Brazilian-Portuguese speaking patients with VPI.

Measuring Patient-Reported Health-Related Quality of Life in Velopharyngeal Insufficiency: Reliability and Validity of the Brazilian Portuguese Version of the VELO Instrument.

OBJECTIVE: To test the Brazilian Portuguese velopharyngeal insufficiency (VPI) Effects on Life Outcome (VELO) instrument for reliability and validity. DESIGN: Cross-sectional methodological study. SETTING: Tertiary craniofacial medical center. PARTICIPANTS: Participants with VPI (VPI group, n = 60), with cleft and without VPI (no VPI/cleft group, n = 60), and with no cleft nor VPI (no VPI/no cleft group, n = 60) and their parents (n = 180). INTERVENTIONS: All patients with VPI 8+ years old and their parents completed the Brazilian-Portuguese VELO instrument and other questionnaires (Pediatric Quality of Life Inventory4.0, PedsQL4.0; Pediatric Voice-Related Quality of Life, PVRQOL; and Intelligibility in Context Scale, ICS) at baseline; patients with VPI and their parents completed the VELO instrument again 2 weeks later. MAIN OUTCOME MEASURES: The VELO instrument was tested for internal consistency, test-retest reliability, discriminant validity (participants with VPI against participants with no VPI), concurrent validity against other questionnaires, criterion validity against hypernasality severity, and construct validity against nasal air emission and overall velopharyngeal competence (speech construct) and velopharyngeal gap (anatomic construct). RESULTS: The VELO had excellent internal consistency (Cronbach α 0.99 for parents and 0.98 for participants with VPI) and test-retest reliability (all intraclass correlation coefficient > 0.87). The VELO discriminated well between VPI group and unaffected groups (all P < .05). The VELO was significantly correlated with the PedsQL4.0, PVRQOL, and ICS (-r > 0.75; P < .001). The VELO met criterion validity, speech construct validity, and anatomic construct validity (r > 0.7; P < .001). CONCLUSIONS: The Brazilian-Portuguese VELO instrument demonstrated reliability (internal consistency and test-retest) and validity (discriminant, concurrent, criterion, and construct).

Morbidity and mortality among neonates with esophageal atresia and/or tracheoesophageal fistula in the United States.

BACKGROUND: Although rare, esophageal atresia (EA) and/or tracheoesophageal fistula (TEF) can lead to severe morbidity and mortality. A known complication of EA and/or TEF repair is vocal fold motion impairment (VFMI). OBJECTIVE: To characterize the prevalence of VFMI among repaired EA and/or TEF at a national level and to identify factors associated with in-patient mortality and common clinical endpoints in this population (tracheostomy and gastrostomy tube dependence). METHODS: This is a retrospective cohort study of inpatient neonate admissions with EA and/or TEF in the 2016 Kids' Inpatient Database (KID). Patients with a history of EA and/or TEF were identified with ICD-10 codes. VFMI prevalence was assessed by observed frequencies and percentages. Clinical endpoints included gastrostomy (G-)tube placement, tracheostomy status, and inpatient mortality. Potential predictors of these three primary outcomes included cardiac anomalies, airway disorders, chromosomal anomalies, surgery within 24 h of admission, prematurity, and low birthweight (LBW). Chi-squared analysis was performed between each parameter and the primary outcomes. Significant associations (p-value<0.05) were further assessed with multivariate logistic regression. RESULTS: Of 1062 neonates with history of EA and/or TEF, 24 (2.3%) had VFMI. Among this population, 27 patients underwent tracheostomies and 63 required G-tubes. There was a significantly higher likelihood of tracheostomy in patients with VFMI compared to patients without (16.7% versus 2.2%; p < 0.01). After multivariate analysis, birthweight less than 1.5 kg (BW < 1.5 kg; OR = 2.5, 95% CI: 1.03-6.10), extreme prematurity (OR = 7.5, 95% CI: 2.00-28.07), cardiac anomalies (OR = 1.8, 95% CI: 1.01-3.04) and chromosomal anomalies (OR = 4.2, 95% CI: 2.39-7.44) were significantly associated with in-hospital mortality. Bronchopulmonary dysplasia (BPD; OR = 4.7, 95% CI: 1.73-12.78) and BW < 1.5 kg (OR = 2.4, 95% CI: 1.05-5.49) were significant predictors for G-tube status. For tracheostomy, BPD (OR = 15.8, 95% CI: 4.70-52.83) and subglottic stenosis (SS, OR = 11.2, 95% CI: 1.85-68.02) remained significant predictors after multivariate regression. CONCLUSION: The national prevalence of VFMI among neonates following EA and/or TEF repair was 2.3% and was associated with an increased likelihood of tracheostomy in the KID 2016. Significant predictors of in-hospital mortality were cardiac anomalies, chromosomal anomalies, extreme prematurity, and BW < 1.5 kg. BPD and SS were significant predictors of tracheostomy, and BPD and BW < 1.5 kg were significant predictors for G-tube requirements.

Childhood Respiratory Conditions: Stridor.

The most important issue in diagnosing stridor in infants and children is determining whether it is acute or chronic. The most common cause of acute stridor is croup, a viral infection with a straightforward diagnosis when patients have typical signs and symptoms (ie, inspiratory or biphasic stridor, barking cough, hoarse voice or cry, chest wall retractions). Management of mild and severe cases includes steroids. Patients with severe croup should undergo evaluation in an urgent care center or emergency department; hospital admission may be required. When symptoms are atypical for croup, physicians should consider other causes of acute stridor, including foreign body aspiration, bacterial tracheitis, and epiglottitis. The most common cause of chronic stridor is laryngomalacia, an abnormality in the laryngeal structures that causes the collapse of supraglottic structures over the airway. Symptoms typically develop in the first 1 to 2 months of life, are positional, and do not interfere with growth and development. However, if the diagnosis is in doubt or if the laryngomalacia is severe and the patient has cyanosis, worsening stridor after feeding, or inadequate weight gain, consultation with an otolaryngology subspecialist can be helpful. Most infants outgrow symptoms as the airway enlarges, but some may need pharmacotherapy for gastroesophageal reflux disease and careful feeding until this occurs. Others may need supraglottoplasty. Other causes of chronic stridor include vocal fold paralysis, subglottic hemangiomas, and glottic webs.

The Quest for a DISE Protocol.

OBJECTIVE: The objective of this quality initiative project was to modify our existing institutional drug-induced sleep endoscopy (DISE) protocol so that the surgeon could consistently determine obstructive breathing patterns while minimizing children's discomfort. METHODS: A quality initiative study utilizing the well-described plan-do-study-act (PDSA) process was conducted at a tertiary hospital for children with polysomnogram-documented obstructive sleep apnea who were undergoing DISE. A 4-point Likert measurement tool was created. Change in each Likert rating with subsequent PDSA cycle was tested with the Wilcoxon rank sum test (Mann-Whitney), and change across all PDSA cycles was tested with the Kruskal-Wallis equality-of-populations rank test. RESULTS: After a series of 4 PDSA cycles with 81 children, the DISE protocol was streamlined from 14 to 9 steps. There was significant improvement for all aspects of the DISE, with a final overall median rating of 1 (excellent) for intravenous (IV) placement, scope insertion, and anesthesiologist and surgeon satisfaction (P < .01). DISCUSSION: For sleep surgeons, DISE is quickly becoming what bronchoscopy is to the airway surgeon. Utilizing inhalational agents to obtain IV access and insert the flexible scope in the rapid "on-off" fashion optimizes DISE success regardless of the primary sedation medication and allows ample time for these agents to dissipate. IMPLICATIONS FOR PRACTICE: Adoption of a DISE protocol that includes nasal premedication and inhalational volatile gases for IV and scope insertion at the onset provides a more predictable level of sedation that is well tolerated by the patient, enabling the otolaryngologist to create an obstructive sleep apnea treatment plan.

Bone Grafting of Alveolar Clefts.

The goals of alveolar cleft repair include (1) stabilization of the maxilla, (2) permitting tooth eruption, (3) eliminating the oronasal fistula, (4) improving aesthetics, and (5) improving speech. Alveolar cleft repair should be considered one of the steps of a larger comprehensive orthodontic management plan. In conjunction with closure of the oronasal fistula, a variety of grafting materials can be used in the alveolar cleft. Autogenous grafts have been found to have greater efficacy compared with allogenic or xenogeneic bone, substitute bone, and alloplasts but with more donor site morbidity.

Personality in Children With Vocal Fold Nodules: A Multitrait Analysis.

Purpose Vocal fold nodules (VNs) are bilateral, symmetrical, callous-like lesions secondary to phonotrauma and possibly related to specific personality traits. This case-control study examined the relation between personality and VNs in children within the context of the Trait Theory of VNs. Method Parents of children with VNs (N = 39, M = 7.43, SD = 2.01 years) and two medical control groups (i.e., voice disordered controls, but not VNs [VDCs; N = 40, M = 7.09, SD = 2.01 years] and vocally normal controls [VNCs; N = 40, M = 7.6, SD = 1.54 years]) completed the Inventory of Child Individual Differences, a personality instrument that describes the Big Five superfactors as well as 15 lower order personality traits. Results Children with VNs, as compared with VNCs, were (a) emotionally reactive (i.e., higher N-Neuroticism, p < .005, Cohen's d = 0.53), (b) Antagonistic, Strong-Willed, and less Compliant (i.e., lower A-Agreeableness, p < .014, Cohen's d = 0.59), and (c) Distractible and Disorganized (i.e., lower C-Conscientiousness, p < .009, Cohen's d = 0.62). Both voice disordered groups displayed elevated scores on the personality superfactor of Neuroticism (N; and the "Negative Emotions" lower order trait). Conclusions The combination of personality traits identified in this study (i.e., high N, low A and C) may play a central role in VNs development and possibly attenuate voice therapy success. Children with VNs displayed a similar personality typology as women with VNs, with the exception of elevated Extraversion (E), thereby providing support for the relevance of the Trait Theory of VNs in both children and adults. Clinicians treating children with voice disorders, including VNs, should consider their underlying personality traits in assessment and management.

Radiofrequency Ablation vs Electrocautery Blinded Randomized Trial: Impact on Clinically Meaningful Outcomes.

OBJECTIVE: To analyze patients' return to normal activity, pain scores, narcotic use, and adverse events after undergoing tonsillectomy or adenotonsillectomy with monopolar electrocautery or radiofrequency ablation. STUDY DESIGN: Randomized double-blinded clinical trial based on prospective parallel design. SETTING: Academic medical center and tertiary children's hospital between March 2018 and July 2019. METHODS: Inclusion criteria included patients aged ≥3 years with surgical indication of recurrent tonsillitis or airway obstruction/sleep-disordered breathing. Patients were randomly assigned to monopolar electrocautery or radiofrequency ablation. Patients were blinded to treatment assignment. Survey questions answered via text or email were collected daily until postoperative day 15. The primary outcome was the patient's return to normal activity. Secondary outcomes included daily pain score, total amount of postoperative narcotic use, and adverse events. RESULTS: Of the 236 patients who met inclusion criteria and were randomly assigned to radiofrequency ablation or monopolar electrocautery, 230 completed the study (radiofrequency ablation, n = 112; monopolar electrocautery, n = 118). There was no statistically significant difference between the groups in the number of days for return to normal activity (P = .89), daily pain scores over 15 postoperative days (P = .46), postoperative narcotic use (P = .61), or return to hospital for any reason (P = .60), including bleeding as an adverse event (P = .13). CONCLUSIONS: As one of the largest randomized controlled trials examining instrumentation in tonsillectomy, our data do not show a difference between monopolar electrocautery and radiofrequency ablation with regard to return to normal activity, daily pain scores, total postoperative narcotic use, or adverse events.

Inter-rater reliability in diagnosis and treatment of type one laryngeal cleft: A blinded observational study.

INTRODUCTION: Type one laryngeal cleft (T1LC) has been implicated as a major contributor to aspiration in non-syndromic pediatric dysphagia. Despite an increasing incidence, there remains controversy in diagnosis and treatment algorithms. OBJECTIVES: The primary objective of this study was to evaluate the inter-rater reliability (IRR) for the diagnosis and treatment of T1LC. METHODS: A retrospective analysis was conducted to identify children evaluated for a T1LC from 2016 to 2017 at a single tertiary care center. The microlaryngoscopy video recordings depicting palpation of the interarytenoid region with a right-angle probe were reviewed. These recordings were shown to blinded pediatric otolaryngologists and each surgeon's determination of the presence or absence of a T1LC as well as recommended treatment (observation, injection laryngoplasty, or endoscopic cleft repair) was recorded and compared against the other blinded surgeons. Fleiss's kappa was calculated to evaluate IRR in both diagnosis and treatment. RESULTS: Eight pediatric otolaryngologists were included in the study with a mean post-training experience of 15 years (range 1-35 years). The inter-rater percent agreement in diagnosis of our patient population was 28.6% (range 3.7-71%) with a kappa value of 0.31 (p < 0.0001). In regard to management, the inter-rater percent agreement in treatment was 11.4% (range 0-35%) with a kappa value of 0.14 (p = 0.01). CONCLUSION: This study highlights the challenges and variation that exists among surgeons in diagnosing and managing potential T1LC. Further standardizing the endoscopic examination and treatment algorithm may reduce diagnostic and treatment discordance.

The Case of the Missing Nose: Congenital Arhinia Case Presentation and Management Recommendations.

OBJECTIVES: To discuss the presentation and management of infants with arhinia or congenital absence of the nose. METHODS: This case report describes an infant with arhinia that was diagnosed prenatally. In addition to a discussion of the case, a review of the literature was completed to define appropriate postnatal work-up and management. RESULTS: The patient is a term male infant, diagnosed with arhinia on ultrasound and magnetic resonance imaging (MRI) performed at 21-weeks gestational age. Upon birth, the patient was subsequently intubated, followed by tracheostomy due to complete nasal obstruction. Through a genetics evaluation, the patient was found to be heterozygous for the SMCHD1 gene, with hypomethylation at the D4Z4 locus. Plans for reconstruction will be based on future imaging and the development of any nasal patency, however, the patient's family plans to utilize a prosthetic nose until the patient is older. CONCLUSION: Arhinia is a rare condition causing respiratory distress in the neonatal period. While stabilization of the airway is the first priority, further management is not clearly defined given the rarity of the malformation. This case discusses stabilization of the airway with a review of treatment and reconstructive options.

Association of Allergic Rhinitis With Change in Nasal Congestion in New Continuous Positive Airway Pressure Users.

Importance: Nasal congestion occurring after continuous positive airway pressure (CPAP) treatment initiation impairs CPAP adherence. Allergic rhinitis is associated with worsening nasal congestion in patients who are exposed to nonallergic triggers. Use of CPAP presents potential nonallergic triggers (eg, humidity, temperature, pressure, and airflow). Objective: To compare nasal congestion among CPAP users with allergic rhinitis, nonallergic rhinitis, and no rhinitis. We hypothesize that CPAP patients with baseline allergic rhinitis are more likely to experience a worsening of nasal congestion (or less improvement in nasal congestion) compared with patients with no baseline rhinitis. Design, Setting, and Participants: This prospective cohort study included consecutive patients newly diagnosed with obstructive sleep apnea in a tertiary sleep center who were using CPAP therapy 3 months after diagnosis. Baseline rhinitis status was assigned as allergic rhinitis, nonallergic rhinitis, or no rhinitis, based on questionnaire responses and past allergy testing. Data were collected from 2004 to 2008 and analyzed from July 2019 to February 2020. Main Outcomes and Measures: At baseline before CPAP exposure and again 3 months later, subjective nasal congestion was measured with the Nasal Obstruction Symptom Evaluation (NOSE) scale and a visual analog scale (VAS), each scored from 0 to 100 (100 = worst congestion). Changes in nasal congestion were tested over 3 months for the whole cohort, within each rhinitis subgroup (paired t test), and between rhinitis subgroups (multivariate linear regression). Results: The study cohort comprised 102 participants, of whom 61 (60%) were male and the mean (SD) age was 50 (13). The study included 23 (22.5%) participants with allergic rhinitis, 67 (65.7%) with nonallergic rhinitis, and 12 (11.8%) with no rhinitis. Nasal congestion improved from baseline to 3 months in the whole cohort (mean [SD] NOSE score, 38 [26] to 27 [23], mean [SD] change, -10 [23]; 95% CI, -15 to -6; mean [SD] VAS score, 41 [27] to 32 [28]; mean [SD] change, -10 [26]; 95% CI, [-15 to -4]) and in each rhinitis subgroup. Adjusted improvement in nasal congestion at 3 months was significantly less in the allergic rhinitis subgroup compared with the no rhinitis subgroup (positive difference means less improvement) compared with baseline: NOSE score 14 (95% CI, 1 to 28) and VAS score 15 (95% CI, 0 to 30). Conclusions and Relevance: Initiation of CPAP was associated with improved subjective nasal congestion, but less improvement in patients with baseline allergic rhinitis. Baseline allergic rhinitis may predict which patients are more vulnerable to potential congestive effects of CPAP.

Development of a Sleep Apnea-Specific Health State Utility Algorithm.

Importance: With the increasing emphasis on economic evaluations, there is a need for additional methods of measuring patient utility in the obstructive sleep apnea population. Objective: To develop and validate a utility scoring algorithm for a sleep apnea-specific quality-of-life instrument. Design, Setting, and Participants: Development and validation were conducted at 2 tertiary referral sleep centers and associated sleep clinics and included patients with newly diagnosed obstructive sleep apnea from a randomized clinical trial and an associated observational cohort study. Baseline participants were randomly divided into a model development group (60%) and a cross-validation group (40%). Main Outcomes and Measures: Utility scoring of the Symptoms of Nocturnal Obstruction and Related Events (SNORE-25) was mapped from the SF-6D utility index through multiple linear regression in the development sample using the Akaike information criterion to determine the best model. Results: A total of 500 participants (development, n = 300; validation, n = 200) were enrolled; the analyzed sample of 500 participants included 295 men (59%), and the mean (SD) age was 48.6 (12.8) years, with a range of 18 to 90 years. The mean (SD) SF-6D utility among participants with untreated sleep apnea was 0.61 (0.08; range, 0.40-0.85) with similar utility across sleep apnea severity groups. The best-fit model (the SNORE Utility Index) was the natural log conversion of the instrument subscales (r2 = 0.32 in the development sample). The SNORE Utility Index retained this association within the validation sample (r2 = 0.33). Conclusions and Relevance: The SNORE Utility Index provides a validated, disease-specific, preference-weighted utility instrument that can be used in future studies of patients with obstructive sleep apnea.

Aspiration in the otherwise healthy Infant-Is there a natural course for improvement?

OBJECTIVES/HYPOTHESIS: Timing and indication for surgical intervention is a major challenge in managing pediatric oropharyngeal dysphagia. No study has evaluated a natural course of swallowing dysfunction in otherwise healthy infants. Our objective was to review the outcomes and time to resolution of abnormal swallow in infants with aspiration. STUDY DESIGN: Retrospective case series at a tertiary children's hospital. METHODS: Fifty patients under 1 year old with aspiration on a modified barium swallow study were included. Patients born <34 weeks, with medical or genetic comorbidities, or who underwent surgical intervention for aspiration were excluded. Patients were followed until aspiration resolved on a swallow study. Kaplan-Meier survival analysis was performed. RESULTS: Forty patients (25 patients [50%] by 6 months, 10 [20%] by 1 year, three [6%] by 2 years, and two [4%] at the end of the follow-up interval) were recommended a normal diet, and 10 patients (20%) were still aspirating by the end of the follow-up interval. Median time to resolution was 202 ± 7 days (range, 19-842 days), probability 48% (95% confidence interval [CI]: 0.34-0.62). The probability of resolution at 6 months was 46% (95% CI: 0.4-0.68), at 1 year was 64% (95% CI: 0.51-0.77), at 2 years was 76% (95% CI: 0.64-0.88), and at the end of the follow-up interval 81.3% (95% CI: 0.7-0.92). CONCLUSIONS: The majority of infants with aspiration and without any other major comorbidities improved within 1 year. Future research should be directed toward better understanding swallowing dysfunction in neurologically normal infants. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:514-520, 2020.

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.

Validation of the Chinese Velopharyngeal Insufficiency Effects on Life Outcomes Instrument.

OBJECTIVES/HYPOTHESIS: To translate the Velopharyngeal Insufficiency Effects on Life Outcomes (VELO) instrument into Chinese and test its psychometric properties. STUDY DESIGN: Quality of life instrument translation and validation. METHODS: The original English version of the VELO instrument was translated into Mandarin, back-translated, and adapted among the Chinese population, based on the standardized guidelines for the cross-culture adaption process. Velopharyngeal insufficiency (VPI) patients were identified by a professional speech and language pathologist. Internal reliability of the VELO instrument was assessed by the Cronbach's α coefficient. Discriminant validity was tested by the Mann-Whitney U test. Construct validity was assessed by factor analysis. RESULTS: A total of 113 patients with VPI and 72 parents of the patients were enrolled. The mean age of the VPI patients was 14.8 years. Internal reliability was excellent; Cronbach's α coefficients were 0.92 and 0.94 for VPI patients and their parents, respectively. The Chinese VELO discriminated well between the VPI group and the controls, with a mean (standard deviation) score that was significantly lower for the VPI group (74.8 [25.7]) than the control group (98.0 [15.9]) (P < .001). The total scores and scores in the emotional domain or perception domain showed differences between VPI patients and their parents. Similar to the original study, the factor loading after rotation followed hypothesized domains largely, in spite of items from several domains loaded on the same factor. CONCLUSIONS: The translated Chinese version of the VELO instrument demonstrated an acceptable reliability, discriminant validity, and construct validity. These psychometric properties suggested theoretical evidence for the further use of the VELO instrument among Chinese patients with VPI. LEVEL OF EVIDENCE: 3b Laryngoscope, 129:E395-E401, 2019.