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1.
Am J Hum Genet ; 103(5): 679-690, 2018 11 01.
Article in English | MEDLINE | ID: mdl-30401457

ABSTRACT

Non-secretor status due to homozygosity for the common FUT2 variant c.461G>A (p.Trp154∗) is associated with either risk for autoimmune diseases or protection against viral diarrhea and HIV. We determined the role of FUT2 in otitis media susceptibility by obtaining DNA samples from 609 multi-ethnic families and simplex case subjects with otitis media. Exome and Sanger sequencing, linkage analysis, and Fisher exact and transmission disequilibrium tests (TDT) were performed. The common FUT2 c.604C>T (p.Arg202∗) variant co-segregates with otitis media in a Filipino pedigree (LOD = 4.0). Additionally, a rare variant, c.412C>T (p.Arg138Cys), is associated with recurrent/chronic otitis media in European-American children (p = 1.2 × 10-5) and US trios (TDT p = 0.01). The c.461G>A (p.Trp154∗) variant was also over-transmitted in US trios (TDT p = 0.01) and was associated with shifts in middle ear microbiota composition (PERMANOVA p < 10-7) and increased biodiversity. When all missense and nonsense variants identified in multi-ethnic US trios with CADD > 20 were combined, FUT2 variants were over-transmitted in trios (TDT p = 0.001). Fut2 is transiently upregulated in mouse middle ear after inoculation with non-typeable Haemophilus influenzae. Four FUT2 variants-namely p.Ala104Val, p.Arg138Cys, p.Trp154∗, and p.Arg202∗-reduced A antigen in mutant-transfected COS-7 cells, while the nonsense variants also reduced FUT2 protein levels. Common and rare FUT2 variants confer susceptibility to otitis media, likely by modifying the middle ear microbiome through regulation of A antigen levels in epithelial cells. Our families demonstrate marked intra-familial genetic heterogeneity, suggesting that multiple combinations of common and rare variants plus environmental factors influence the individual otitis media phenotype as a complex trait.


Subject(s)
Fucosyltransferases/genetics , Genetic Variation/genetics , Otitis Media/genetics , Animals , COS Cells , Cell Line , Chlorocebus aethiops , Ear, Middle/microbiology , Exome/genetics , Female , HEK293 Cells , Humans , Male , Mice , Mice, Inbred C57BL , Microbiota/physiology , Otitis Media/microbiology , Pedigree , Galactoside 2-alpha-L-fucosyltransferase
2.
Hum Mutat ; 40(8): 1156-1171, 2019 08.
Article in English | MEDLINE | ID: mdl-31009165

ABSTRACT

A genetic basis for otitis media is established, however, the role of rare variants in disease etiology is largely unknown. Previously a duplication variant within A2ML1 was identified as a significant risk factor for otitis media in an indigenous Filipino population and in US children. In this report exome and Sanger sequencing was performed using DNA samples from the indigenous Filipino population, Filipino cochlear implantees, US probands, Finnish, and Pakistani families with otitis media. Sixteen novel, damaging A2ML1 variants identified in otitis media patients were rare or low-frequency in population-matched controls. In the indigenous population, both gingivitis and A2ML1 variants including the known duplication variant and the novel splice variant c.4061 + 1 G>C were independently associated with otitis media. Sequencing of salivary RNA samples from indigenous Filipinos demonstrated lower A2ML1 expression according to the carriage of A2ML1 variants. Sequencing of additional salivary RNA samples from US patients with otitis media revealed differentially expressed genes that are highly correlated with A2ML1 expression levels. In particular, RND3 is upregulated in both A2ML1 variant carriers and high-A2ML1 expressors. These findings support a role for A2ML1 in keratinocyte differentiation within the middle ear as part of otitis media pathology and the potential application of ROCK inhibition in otitis media.


Subject(s)
Down-Regulation , Gene Expression Profiling/methods , Mutation , Otitis Media/genetics , Sequence Analysis, DNA/methods , alpha-Macroglobulins/genetics , Adolescent , Adult , Child , Child, Preschool , Female , Finland , Gene Expression Regulation , Genetic Predisposition to Disease , Humans , Infant , Male , Middle Aged , Pakistan , Pedigree , Philippines , Sequence Analysis, RNA , Signal Transduction , United States , Young Adult
3.
J Pediatr ; 211: 179-184.e1, 2019 08.
Article in English | MEDLINE | ID: mdl-31084917

ABSTRACT

OBJECTIVES: To examine weight changes relative to surgical success in children with Down syndrome and obstructive sleep apnea (OSA). STUDY DESIGN: Retrospective chart review of children with Down syndrome undergoing tonsillectomy from 2005 to 2016 for OSA at a tertiary care children's hospital. Only patients with pre-and postoperative polysomnogram within 6 months of tonsillectomy were included. Demographics, weight, height, and polysomnogram data were collected. Body mass index (BMI), expressed as a percentage of the 95th percentile (%BMIp95), was calculated for 24 months prior to and following surgery. Pre-and postoperative OSA severity were also recorded. The postoperative obstructive/hypopnea index identified subjects with resolution of obstruction (obstructive/hypopnea index <2 events/hour) or persistent mild/moderate/severe obstructive apnea. Regression analyses were used to compare %BMIp95 pre- and post-tonsillectomy with %BMIp95 by OSA status following tonsillectomy. RESULTS: A total of 78 patients with Down syndrome whose mean age was 5.29 years at time of tonsillectomy were identified. There was no difference between best-fit curves of %BMI p95 pre-and post-tonsillectomy. There was no difference between best-fit curves of %BMI p95 in patients who saw resolution of OSA after tonsillectomy vs patients with residual OSA. CONCLUSIONS: Tonsillectomy neither alters the BMI trajectory of children with Down syndrome, nor changes differentially the risk for obesity in children whose OSA did or did not resolve after surgery.


Subject(s)
Body Mass Index , Down Syndrome/epidemiology , Pediatric Obesity/epidemiology , Tonsillectomy , Adolescent , Child , Child, Preschool , Female , Humans , Infant , Male , Regression Analysis , Retrospective Studies , Sleep Apnea, Obstructive/surgery
4.
Cleft Palate Craniofac J ; 56(7): 890-895, 2019 08.
Article in English | MEDLINE | ID: mdl-31282194

ABSTRACT

OBJECTIVE: To determine whether nonsupine sleep improves obstructive sleep apnea (OSA) in infants with cleft palate undergoing polysomnography (PSG). DESIGN: Retrospective chart review. SETTING: Tertiary care pediatric hospital. PATIENTS: Twenty-seven infants (1 month to 1 year) with cleft palate with or without cleft lip (CP ± L) undergoing PSG testing for suspected OSA were included. MAIN OUTCOME MEASURES: Polysomnography measures included obstructive apnea-hypopnea index (OAHI), central apnea-hypopnea index (CAHI), oxygen saturation (SpO2) nadir, SpO2, and end-tidal carbon dioxide (ETCO2). RESULTS: Twenty-three PSGs with at least 20 minutes of sleep in both the supine and the nonsupine positions were analyzed. The supine OAHI (mean: 16.8 events/hour; standard deviation [SD]: 18.5) and nonsupine OAHI (mean: 12.6 events/hour; SD: 12.6) did not differ significantly (P = .10). The supine CAHI (mean: 1.9 events/hour; SD: 2.7) and nonsupine CAHI (mean: 3.1 events/hour; SD: 3.7; P = .15), the supine SpO2 nadir (mean: 81.2%; SD: 6.3) and nonsupine SpO2 nadir (mean: 81.8%; SD: 5.3; P = .70), the supine mean SpO2 (mean: 95.5%; SD: 1.9) and nonsupine mean SpO2 saturation (mean: 95.3%; SD: 2.4; P = .34), and the supine ETCO2 (mean: 45.4 mm Hg; SD: 5.3) and nonsupine ETCO2 (mean: 42.5 mm Hg; SD: 10.1; P = .24) were also similar. CONCLUSIONS: There were no significant improvements in OSA metrics during nonsupine sleep in infants with CP ± L. Prior to recommending nonsupine positioning which increases infant's exposure to sudden infant death syndrome risk, we advocate obtaining a PSG to verify an objective improvement in OSA.


Subject(s)
Cleft Lip , Cleft Palate , Posture , Sleep Apnea, Obstructive , Sleep , Cleft Palate/complications , Humans , Infant , Infant, Newborn , Polysomnography , Retrospective Studies , Sleep Apnea, Obstructive/etiology
5.
J Clin Sleep Med ; 20(2): 237-243, 2024 Feb 01.
Article in English | MEDLINE | ID: mdl-37858282

ABSTRACT

STUDY OBJECTIVES: Clinical practice guidelines recommend screening all children with Down syndrome for obstructive sleep apnea with polysomnography by age 4 years. Because persistent obstructive sleep apnea (obstructive apnea-hypopnea index > 1 event/h) following adenotonsillectomy (T&A) is common in children with Down syndrome, it is important to know whether clinicians can rely on parental assessment postoperatively. The primary objective is to identify accuracy of parental perception of nighttime breathing following T&A compared with preoperative assessment. METHODS: Patients with Down syndrome who underwent T&A along with polysomnography prior to and after the surgical procedure were included. Parents completed a 3-question pre- and postsurgery survey regarding nighttime symptoms. The responses were categorized into 3 groups: infrequent (< 3 nights/wk), sometimes (> 3 nights/wk but < 6 nights/wk), or frequent (≥ 6 nights/wk) on at least 1 question. The primary end point was identifying the accuracy of parental perception of nighttime breathing in children with Down syndrome following T&A. RESULTS: A total of 256 children met inclusion criteria, of which 117 (46%) were included. A total of 71 (68%) children had an obstructive apnea-hypopnea index > 5 events/h preoperatively compared with 55 (47%) postoperatively. There was no association between parents' perception of symptoms and obstructive sleep apnea categorization postoperatively (P > .05) or of parents' perception of symptoms improving and obstructive sleep apnea categorization improving postoperatively (P > .05). CONCLUSIONS: Despite previous experience, parents are unable to predict nighttime breathing patterns following a T&A. We recommend obtaining a polysomnogram rather than relying on parental assessment to determine whether a T&A has been successful. CITATION: Chabuz CA, Lackey TG, Pickett KL, Friedman NR. Accuracy of parental perception of nighttime breathing in children with Down syndrome after adenotonsillectomy. J Clin Sleep Med. 2024;20(2):237-243.


Subject(s)
Down Syndrome , Sleep Apnea, Obstructive , Tonsillectomy , Child , Humans , Child, Preschool , Down Syndrome/complications , Down Syndrome/surgery , Tonsillectomy/methods , Adenoidectomy/methods , Sleep Apnea, Obstructive/complications , Sleep Apnea, Obstructive/surgery , Sleep Apnea, Obstructive/diagnosis , Parents , Respiration , Perception
6.
Otolaryngol Head Neck Surg ; 170(1): 277-283, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37668178

ABSTRACT

OBJECTIVE: To analyze the growth trajectory of children with obesity before and after adenotonsillectomy (T&A). We hypothesize that T&A will not affect the growth trajectory but children in a multidisciplinary weight management program (MWMP) will have a healthier growth trajectory. STUDY DESIGN: Retrospective review. SETTING: Tertiary Children's Hospital. METHODS: Body mass index (BMI) trajectories of nonsyndromic children with obesity and obstructive sleep apnea (OSA) who underwent T&A were analyzed. A linear mixed effects model was fit to the BMI expressed as a percentage of the 95th percentile (%BMIp95 ) data. Covariates included demographic variables, pre- and postoperative participation in an MWMP, baseline obesity class, and time. We explored clinically meaningful interactions. BMI slope estimates before and after surgery were calculated and compared for baseline obesity classification and postoperative MWMP visits. RESULTS: A total of 177 patients, 58% male with a mean age of 9.7 years at the time of surgery, were studied. Higher baseline obesity class (II and III), time, the interaction between obesity class III and elapsed time relative to surgical date, and the interaction between obesity class III and the postsurgical period were all significantly associated with the outcome of %BMIp95 (P < .05). There was a significantly higher %BMIp95 trajectory following surgery in patients with baseline obesity class III who did not have any postoperative MWMP visits (P < .001). Preoperative obesity visits, however, were not significantly associated with postoperative growth. CONCLUSION: The association between T&A and weight trajectory depends upon obesity class and participation in a MWMP. Coordinated care of children with obesity between otolaryngologists and an MWMP may improve OSA and obesity outcomes. LEVEL OF EVIDENCE: The level of evidence: 3.


Subject(s)
Sleep Apnea, Obstructive , Tonsillectomy , Child , Humans , Male , Female , Polysomnography , Adenoidectomy , Obesity/complications , Retrospective Studies
7.
Laryngoscope ; 134(5): 2449-2454, 2024 May.
Article in English | MEDLINE | ID: mdl-37971081

ABSTRACT

OBJECTIVE: To determine if children with greater social vulnerability are more likely to experience a prolonged oxygen requirement (POR) following adenotonsillectomy to inform the need for overnight monitoring prior to discharge. METHODS: A previously published prospective study assessing children observed overnight following adenotonsillectomy for obstructive sleep-disordered breathing was reanalyzed including social vulnerability index (SVI). The outcome was POR beyond 3 h following extubation. Logistic regression was used to assess the association of SVI components with POR. SVI components were assessed as quartiles of cohort values. Final adjusted models included race, asthma, Down syndrome, and pre-operative SpO2. RESULTS: A total of 462 children had SVI data available and were included. 354 (76.6%) were > = 3 years of age. Overall, 351 (76%) did not have a POR. The median overall SVI percentile was 26.5 (Q1 10.4, Q3 60.1). When categorized by SVI quartiles, there was a statistically significant difference with POR for overall SVI percentile (p = 0.007), SVI household composition percentile (p = 0.033), and median SVI housing/transportation percentile (p = 0.005). Individuals with an overall SVI in the 4th quartile (greatest vulnerability) were 2.63 times more likely to experience a POR than those in the 1st quartile (lowest social vulnerability) in adjusted logistic regression (95% OR CI 1.23-5.62; p = 0.01). CONCLUSIONS: There is a significant association between greater neighborhood-level social vulnerability and a POR following adenotonsillectomy. We propose that a child's SVI be considered when planning for the perioperative course following adenotonsillectomy. LEVEL OF EVIDENCE: 3 Laryngoscope, 134:2449-2454, 2024.


Subject(s)
Sleep Apnea, Obstructive , Tonsillectomy , Child , Humans , Prospective Studies , Social Vulnerability , Adenoidectomy , Sleep Apnea, Obstructive/surgery , Oxygen
8.
Otolaryngol Head Neck Surg ; 170(3): 905-912, 2024 Mar.
Article in English | MEDLINE | ID: mdl-37937707

ABSTRACT

OBJECTIVE: Girls with Turner syndrome (TS) often have features that have been associated with obstructive sleep-disordered breathing (oSDB). However, little is known about oSDB in TS. Herein, we aimed to characterize oSDB in young patients with TS and identify associated risk factors. STUDY DESIGN: Retrospective cross-sectional study. SETTING: Tertiary care pediatric hospital. METHODS: We reviewed medical records for patients diagnosed with TS seen at our institution between October 1, 2007 and December 31, 2019 with the first outpatient visit before age 6 years. The prevalence of oSDB was compared to the general pediatric population with 1-sample binomial proportion tests. Clinical characteristics were compared between those diagnosed with oSDB and those without oSDB, and risk factors for oSDB were identified. RESULTS: Of 151 patients with TS, 73 (48%) were diagnosed with oSDB which is 4-fold higher than the general pediatric population (12%, P < 0.0001). In the multivariable model, adenoid, tonsillar, and inferior turbinate hypertrophy, birthweight, failure to thrive, and older age at the last clinic visit were all associated with increased odds for oSDB. CONCLUSION: Young children with TS have a high prevalence of oSDB and thus should be screened for oSDB. Polysomnography should be performed in those with associated risk factors and symptoms oSDB. Treatment of oSDB is imperative as individuals with TS are already at increased risk of behavioral problems, neurocognitive deficits, and growth impairment that may be worsened with oSDB.


Subject(s)
Sleep Apnea, Obstructive , Turner Syndrome , Female , Child , Humans , Child, Preschool , Prevalence , Turner Syndrome/complications , Turner Syndrome/epidemiology , Retrospective Studies , Cross-Sectional Studies , Sleep Apnea, Obstructive/diagnosis
9.
Otolaryngol Head Neck Surg ; 170(4): 1158-1166, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38169046

ABSTRACT

OBJECTIVE: The Food and Drug Administration recently approved upper airway stimulation (UAS) for children with Down Syndrome and persistent obstructive sleep apnea who meet certain inclusion and exclusion criteria. Although there is a robust experience with this therapy in the adult population, established protocols used in adults are not directly transferrable to a complex pediatric population. This review aims to combine the protocols from several institutions for patient selection and postimplantation optimization, including a protocol for Drug-Induced Sleep Endoscopy in children with Down Syndrome, preactivation threshold measurements, device titration, and follow-up sleep studies. STUDY DESIGN: Expert panel development of best Practice algorithm. SETTING: Multi-institutional investigator review. METHODS: An expert panel was assembled of pediatric otolaryngologists with extensive experience in hypoglossal nerve stimulation in children with Down Syndrome. Thirty statements were created during an initial drafting session. A modified Delphi method was used assess consensus among the panel. RESULTS: After 2 rounds of Delphi surveys, 29 statements met criteria for consensus. One statement did not meet consensus. The statements were grouped into several categories to facilitate presentation. CONCLUSIONS: A standardized approach to UAS for children with Down Syndrome must take into account the unique challenges inherent to treating a complex pediatric population with a high rate of sensory processing disorders. This expert panel has met consensus on several statements that will guide clinicians as this novel therapy is adopted.


Subject(s)
Down Syndrome , Electric Stimulation Therapy , Sleep Apnea, Obstructive , Adult , Humans , Child , Down Syndrome/complications , Patient Selection , Sleep Apnea, Obstructive/therapy , Nose , Endoscopy/methods , Electric Stimulation Therapy/methods , Hypoglossal Nerve
10.
Int J Pediatr Otorhinolaryngol ; 176: 111799, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38081110

ABSTRACT

OBJECTIVE: Postviral olfactory dysfunction (OD) including corona 2019 viral disease (COVID-19) OD occurs in both adults and children. Despite limited reports of efficacy in treating adult postviral including COVID-19 OD with olfactory training (OT), its effects on children in general, and post-COVID-19 in specific, is unknown. The study aimed at evaluating the effects of OT in a COVID-19 OD pediatric cohort. METHODS: A single-arm prospective study of pediatric COVID-19 OD subjects confirmed by the University of Pennsylvania Smell Identification Test (UPSIT), was conducted. All subjects underwent OT by sniffing 4 odorants (lavender, orange, peppermint, and eucalyptus) for 1 min twice a day for 3 months. Subjects underwent an odorant identification test (OIT) of the 4 odorants each visit. A repeat UPSIT was administered at the 4th visit. RESULTS: The study enrolled a total of 37 subjects [11 males/26 females with mean age/standard deviation (std) of 15.6(2.1) years]. The time interval between COVID-19 and entry was 5.3(2.4) months. The mean pre/post study UPSIT score improvement was 2.3(4.7), p = .09. OIT scores between entry and 3 subsequent visits showed a mean improvement of 1.8(1.5), 1.8(1.9) and 2.3(1.9) odorants, respectively, with P < .001 for all 3 comparisons. CONCLUSIONS: OT subjects were predominantly female teens with substantial OD lasting greater than 5 months. OT did not affect OD as measured by UPSIT but OIT scores improved during OT. We postulate that OT likely has a role in pediatric post-COVID OD recovery, but UPSIT likely is too rigid to detect disparate odorant improvement.


Subject(s)
COVID-19 , Olfaction Disorders , Adult , Male , Adolescent , Humans , Female , Child , Smell , Prospective Studies , Olfactory Training , Olfaction Disorders/diagnosis , Olfaction Disorders/etiology , Olfaction Disorders/therapy , Odorants
11.
Cureus ; 16(5): e60214, 2024 May.
Article in English | MEDLINE | ID: mdl-38868294

ABSTRACT

INTRODUCTION: Aerosol mitigation equipment implemented due to COVID-19 has increased noise levels in the operating room (OR) during otolaryngological procedures. Intraoperative sound levels may potentially place personnel at risk for occupational hearing loss. This study hypothesized that cumulative intraoperative noise exposures with aerosol mitigation equipment exceed recommended occupational noise exposure levels. METHODS: Sound levels generated by the surgical smoke evacuator (SSE) during adenotonsillectomy were measured using a sound level meter and compared to surgery without SSE. RESULTS: Thirteen adenotonsillectomy surgeries were recorded. Mean sound levels with the SSE were greater than the control (72 ± 3 A-weighted decibels (dBA) vs. 68 ± 2 dBA; p=0.015). Maximum noise levels during surgery with SSE reached 82 ± 3 dBA. CONCLUSION: Surgeons performing adenotonsillectomy with aerosol mitigation equipment are exposed to significant noise levels. Intraoperative sound levels exceeded international standards for work requiring concentration. Innovation is needed to reduce cumulative OR noise exposures.

12.
J Clin Sleep Med ; 19(1): 171-177, 2023 01 01.
Article in English | MEDLINE | ID: mdl-36081330

ABSTRACT

STUDY OBJECTIVES: To examine children with Down syndrome with residual obstructive sleep apnea (OSA) to determine if they are more likely to have positional OSA. METHODS: A retrospective chart review of children with Down syndrome who underwent adenotonsillectomy at a single tertiary children's hospital was conducted. Children with Down syndrome who had a postoperative polysomnogram with obstructive apnea-hypopnea index (OAHI) > 1 event/h, following adenotonsillectomy with at least 60 minutes of total sleep time were included. Patients were categorized as mixed sleep (presence of ≥ 30 minutes of both nonsupine and supine sleep), nonsupine sleep, and supine sleep. Positional OSA was defined as an overall OAHI > 1 event/h and a supine OAHI to nonsupine OAHI ratio of ≥ 2. Group differences are tested via Kruskal-Wallis test for continuous variables and Fisher's exact tests for categorical. RESULTS: There were 165 children with Down syndrome who met inclusion criteria, of which 130 individuals had mixed sleep. Patients who predominately slept supine had a greater OAHI than mixed and nonsupine sleep (P = .002). Sixty (46%) of the mixed-sleep individuals had positional OSA, of which 29 (48%) had moderate/severe OSA. Sleeping off their backs converted 14 (48%) of these 29 children from moderate/severe OSA to mild OSA. CONCLUSIONS: Sleep physicians and otolaryngologists should be cognizant that the OAHI may be an underestimate if it does not include supine sleep. Positional therapy is a potential treatment option for children with residual OSA following adenotonsillectomy and warrants further investigation. CITATION: Lackey TG, Tholen K, Pickett K, Friedman N. Residual OSA in Down syndrome: does body position matter? J Clin Sleep Med. 2023;19(1):171-177.


Subject(s)
Down Syndrome , Sleep Apnea, Obstructive , Tonsillectomy , Child , Humans , Down Syndrome/complications , Retrospective Studies , Sleep Apnea, Obstructive/complications , Sleep Apnea, Obstructive/surgery , Adenoidectomy
13.
Laryngoscope ; 2023 Dec 10.
Article in English | MEDLINE | ID: mdl-38073113

ABSTRACT

OBJECTIVE: Adenotonsillectomy is a common procedure performed in children, and the practice patterns at academic centers have been evolving with the publication of updated societal guidelines. In this study, we assess perioperative practice patterns at tertiary care children's hospitals for children undergoing adenotonsillectomy. METHODS: A cross-sectional 18-question survey distributed in July of 2022 recruited responses through August 25, 2022. The division chiefs of 70 pediatric otolaryngology groups at tertiary care children's hospitals across the United States and Canada were surveyed. Division chiefs submitted survey responses on behalf of the group practice patterns for children undergoing adenotonsillectomy. The main measure was survey responses from the division chiefs of pediatric otolaryngology reporting group practice. RESULTS: The survey response rate was 46%. Eighty-eight percent of groups reported an official adenotonsillectomy admission policy. Commonly reported admission criteria included age (93%) and obesity (59%). Eighty-eight percent of groups defined severe obstructive sleep apnea as apnea-hypopnea index ≥10. Only 41% of groups required a child to be observed sleeping on room air prior to ambulatory discharge. Ninety-seven percent of groups reported routinely obtaining preoperative polysomnography in a variety of clinical settings. CONCLUSIONS: Many children's hospitals report an official admission policy following adenotonsillectomy. Despite a decade passing since the initial publication of the American Academy of Otolaryngology-Head and Neck Surgery clinical practice guidelines, there remains great variability in the practice patterns for both preoperative polysomnography and postoperative monitoring and admission. These results highlight an opportunity for an improved unified approach to perioperative pediatric adenotonsillectomy practice. LEVEL OF EVIDENCE: 5 Laryngoscope, 2023.

14.
Int J Pediatr Otorhinolaryngol ; 174: 111748, 2023 Nov.
Article in English | MEDLINE | ID: mdl-37820572

ABSTRACT

OBJECTIVES: To develop and validate a consensus international pediatric sleep endoscopy scale (IPSES) for pediatric drug-induced sleep endoscopy (DISE). METHODS: Existing published DISE ratings scales were reviewed in order to develop a consensus rating scale synthesizing the most common features and adding new elements to address areas of controversy. Samples of 30 de-identified DISE video recordings were reviewed to develop and refine the scale. After the consensus scale was defined, a separate sample of 25 de-identified DISE videos were scored with the new consensus scale by the development group and a panel of independent raters. A weighted kappa statistic was used to quantify the inter-rater and intra-rater reliability of the consensus scale at each anatomic level. RESULTS: Among all raters, intra-rater reliability was most variable for the nasal airway (kappa range 0.33-0.94) and best for the lateral oropharynx (kappa range 0.68-0.95). Inter-rater reliability ranged from 0.43 for the nasal airway to 0.57 at the soft palate. CONCLUSION: The IPSES is a reliable consensus scale that reflects the most common features of existing scales and can be adopted as a universal scoring scale for pediatric DISE.


Subject(s)
Sleep Apnea, Obstructive , Humans , Child , Reproducibility of Results , Endoscopy , Palate, Soft , Sleep
15.
Genet Test Mol Biomarkers ; 27(7): 221-228, 2023 Jul.
Article in English | MEDLINE | ID: mdl-37522794

ABSTRACT

Background: Otitis media (OM) is defined as middle ear (ME) inflammation that is usually due to infection. Globally, OM is a leading cause of hearing loss and is the most frequently diagnosed disease in young children. For OM, pediatric patients with Down syndrome (DS) demonstrate higher incidence rates, greater severity, and poorer outcomes. However, to date, no studies have investigated the bacterial profiles of children with DS and OM. Method: We aimed to determine if there are differences in composition of bacterial profiles or the relative abundance of individual taxa within the ME and nasopharyngeal (NP) microbiotas of pediatric OM patients with DS (n = 11) compared with those without DS (n = 84). We sequenced the 16S rRNA genes and analyzed the sequence data for diversity indices and relative abundance of individual taxa. Results: Individuals with DS demonstrated increased biodiversity in their ME and NP microbiotas. In children with OM, DS was associated with increased biodiversity and higher relative abundance of specific taxa in the ME. Conclusion: Our findings suggest that dysbioses in the NP of DS children contributes to their increased susceptibility to OM compared with controls. These findings suggest that DS influences regulation of the mucosal microbiota and contributes to OM pathology.


Subject(s)
Down Syndrome , Microbiota , Otitis Media , Child , Humans , Child, Preschool , RNA, Ribosomal, 16S/genetics , Down Syndrome/genetics , Otitis Media/genetics , Ear, Middle/microbiology , Ear, Middle/pathology , Microbiota/genetics
16.
Int J Pediatr Otorhinolaryngol ; 171: 111627, 2023 Aug.
Article in English | MEDLINE | ID: mdl-37441992

ABSTRACT

OBJECTIVES: To develop consensus statements for the scoring of pediatric drug induced sleep endoscopy in the diagnosis and management of pediatric obstructive sleep apnea. METHODS: The leadership group identified experts based on defined criteria and invited 18 panelists to participate in the consensus statement development group. A modified Delphi process was used to formally quantify consensus from opinion. A modified Delphi priori process was established, which included a literature review, submission of statements by panelists, and an iterative process of voting to determine consensus. Voting was based on a 9-point Likert scale. Statements achieving a mean score greater than 7 with one or fewer outliers were defined as reaching consensus. Statements achieving a mean score greater than 6.5 with two or fewer outliers were defined as near consensus. Statements with lower scores or more outliers were defined as no consensus. RESULTS: A total of 78 consensus statements were evaluated by the panelists at the first survey - 49 achieved consensus, 18 achieved near consensus, and 11 did not achieve consensus. In the second survey, 16 statements reached consensus and 5 reached near consensus. Regarding scoring, consensus was achieved on the utilization of a 3-point Likert scale for each anatomic site for maximal observed obstructions of <50% (Score 0, no-obstruction), ≥ 50% but <90% (Score 2, partial obstruction), and ≥ 90% (Score 3, complete obstruction). Anatomic sites to be scored during DISE that reached consensus or near-consensus were the nasal passages, adenoid pad, velum, lateral pharyngeal walls, tonsils (if present), tongue base, epiglottis, and arytenoids. CONCLUSION: This study developed consensus statements on the scoring of DISE in pediatric otolaryngology using a modified Delphi process. The use of a priori process, literature review, and iterative voting method allowed for the formal quantification of consensus from expert opinion. The results of this study may provide guidance for standardizing scoring of DISE in pediatric patients.


Subject(s)
Endoscopy , Sleep Apnea, Obstructive , Child , Humans , Endoscopy/methods , Pharynx , Polysomnography/methods , Sleep , Sleep Apnea, Obstructive/diagnosis
17.
Laryngoscope ; 132(2): 461-469, 2022 02.
Article in English | MEDLINE | ID: mdl-34191279

ABSTRACT

OBJECTIVES/HYPOTHESIS: To determine the success of an adenotonsillectomy (T&A) in treating children with severe obesity utilizing a more accurate obesity scale. STUDY DESIGN: Retrospective cohort. METHODS: A retrospective cohort of children with obesity between 5 and 10 years of age who underwent a T&A at Children's Hospital of Colorado (CHCO) was used. This study also utilized publicly available data from the Childhood Adenotonsillectomy Trial (CHAT) study. The cohort was divided into three obesity classes using age- and sex-specific body mass index (BMI) expressed as a percentage of the 95th percentile (%BMIp95) and compared for operative success differences. RESULTS: There were 132 patients included in our primary analysis, with obesity distribution as follows: Class 1 to 53 patients (40%), Class 2 to 45 patients (34%), and Class 3 to 34 patients (26%). Overall, 52 patients (35.9%) experienced a cure (obstructive apnea/hypopnea index [OAHI] <1), with 27 (52%) patients in Class 1 obesity, 18 (35%) in Class 2, and 7 (13%) in Class 3. Class 3 had a significantly lower obstructive sleep apnea cure rate compared with Class 1 patients (P = .013), but after adjusting for covariates, this difference was no longer present (P > .05). There was no significant difference in the preoperative to postoperative percent change in mean oxygen saturation (P = .82 CHCO, P = .43 CHAT), oxygen nadir (P = .20 CHCO, P = .49 CHAT), or OAHI (P = .12 CHCO, P = .26 CHAT) between the obesity classes. CONCLUSION: After adjusting for covariates, children with Class 3 obesity are as likely to be cured with a T&A as those with Class 1 obesity. A T&A should be considered a first line treatment for all children with obesity. LEVEL OF EVIDENCE: 3 Laryngoscope, 132:461-469, 2022.


Subject(s)
Adenoidectomy , Obesity, Morbid/complications , Pediatric Obesity/complications , Sleep Apnea, Obstructive/etiology , Sleep Apnea, Obstructive/surgery , Tonsillectomy , Child , Cohort Studies , Female , Humans , Male , Obesity, Morbid/classification , Pediatric Obesity/classification , Retrospective Studies , Treatment Outcome
18.
Laryngoscope ; 132(6): 1289-1294, 2022 06.
Article in English | MEDLINE | ID: mdl-34551129

ABSTRACT

OBJECTIVES/HYPOTHESIS: To reanalyze the growth trajectory and assess longitudinal changes of children undergoing adenotonsillectomy (AT) versus watchful waiting (WW) enrolled in the Childhood Adenotonsillectomy Trial (CHAT) study and to determine if an AT increases the risk of obesity in children. STUDY DESIGN: Reanalysis of prospective cohort investigation. METHODS: The study analyzed publicly available data from CHAT, including 3 months visit data not previously included in a prior publication. Statistical comparisons and mixed-effects modeling were done using age- and sex-specific BMI expressed as a percentage of the 95th percentile (%BMIp95). P < .05 was considered significant. RESULTS: Children in the AT group, especially if underweight at baseline, had an increased rate of weight gain, with 100% of underweight children in the AT group becoming normal weight compared to 20% for WW. However, the rate of weight gain, as measured by the %BMIp95 trajectory for both AT and WW groups, was not significantly different when baseline weight status and obstructive sleep apnea (OSA) resolution were accounted for. Comparisons of %BMIp95 between treatment groups at baseline, 3- and 7-month follow-up visits also failed to identify statistically significant differences (P > .05). Overall for the entire cohort, resolution of OSA was associated with a decreased weight trajectory (P < .001). CONCLUSIONS: AT compared to WW is not associated with an increased risk of excessive weight gain. Otolaryngologists should be aware of this updated analysis when discussing AT surgical outcomes with families. LEVEL OF EVIDENCE: 2 Laryngoscope, 132:1289-1294, 2022.


Subject(s)
Pediatric Obesity , Sleep Apnea, Obstructive , Tonsillectomy , Adenoidectomy/adverse effects , Child , Female , Humans , Male , Pediatric Obesity/complications , Pediatric Obesity/surgery , Prospective Studies , Sleep Apnea, Obstructive/complications , Thinness/complications , Tonsillectomy/adverse effects , Weight Gain
19.
Otolaryngol Head Neck Surg ; 167(3): 590-599, 2022 09.
Article in English | MEDLINE | ID: mdl-34399647

ABSTRACT

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.


Subject(s)
Endoscopy , Sleep Apnea, Obstructive , Bronchoscopy , Child , Endoscopy/methods , Humans , Polysomnography/methods , Sleep , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/surgery
20.
Laryngoscope ; 132(8): 1675-1681, 2022 08.
Article in English | MEDLINE | ID: mdl-34672364

ABSTRACT

OBJECTIVES/HYPOTHESIS: Children who do not require oxygen beyond 3 hours after surgery and pass a sleep room air challenge (SRAC) are safe for discharge regardless of polysomnogram (PSG) results or comorbidities. STUDY DESIGN: Cross-sectional prospective study. METHODS: All children observed overnight undergoing an adenotonsillectomy for obstructive sleep-disordered breathing were prospectively recruited. Demographic, clinical, and PSG characteristics were stratified by whether the patient had required oxygen beyond 3 hours postoperatively (prolonged oxygen requirement [POR]) and compared using t test, chi-squared test, or Fisher's exact test depending on distribution. Optimal cut points for predicting POR postsurgery were calculated using receiver operating characteristic curves. The primary analysis was performed on the full cohort via logistic regression using POR as the outcome. Significant characteristics were analyzed in a logistic regression model, with significance set at P < .05. RESULTS: A total of 484 participants met the inclusion criteria. The mean age was 5.65 (standard deviation = 4.02) years. Overall, 365 (75%) did not have a POR or any other adverse respiratory event. In multivariable logistic regression, risk factors for POR were an asthma diagnosis (P < .001) and an awake SpO2 <96% (P = .005). The probability of a POR for those without asthma and a SpO2 ≥ 96% was 18% (95% confidence interval: 14-22). Age, obesity, and obstructive apnea/hypopnea index were not associated with POR. CONCLUSIONS: In conclusion, all children in our study who are off oxygen within 3 hours of surgery and passed a SRAC were safe for discharge from a respiratory standpoint regardless of age, obesity status, asthma diagnosis, and obstructive apnea/hypopnea index. Additional investigations are necessary to confirm our findings. LEVEL OF EVIDENCE: 3 Laryngoscope, 132:1675-1681, 2022.


Subject(s)
Asthma , Sleep Apnea, Obstructive , Tonsillectomy , Adenoidectomy/adverse effects , Asthma/complications , Child , Child, Preschool , Cross-Sectional Studies , Humans , Obesity/complications , Oxygen , Prospective Studies , Tonsillectomy/adverse effects
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