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  • 1
    In: Otolaryngology–Head and Neck Surgery, Wiley, Vol. 168, No. 1 ( 2023-01), p. 74-81
    Abstract: Caregivers frequently report poor quality of life (QOL) in children with sleep‐disordered breathing (SDB). Our objective is to assess the correlation between caregiver‐ and child‐reported QOL in children with mild SDB and identify factors associated with differences between caregiver and child report. Study Design Analysis of baseline data from a multi‐institutional randomized trial Setting Pediatric Adenotonsillectomy Trial for Snoring, where children with mild SDB (obstructive apnea‐hypopnea index 〈 3) were randomized to observation or adenotonsillectomy. Methods The Pediatric Quality of Life Inventory (PedsQL) assessed baseline global QOL in participating children 5 to 12 years old and their caregivers. Caregiver and child scores were compared. Multivariable regression assessed whether clinical factors were associated with differences between caregiver and child report. Results PedsQL scores were available for 309 families (mean child age, 7.0 years). The mean caregiver‐reported PedsQL score was higher at 75.2 (indicating better QOL) than the mean child‐reported score of 67.9 ( P 〈 .001). The agreement between caregiver and child total PedsQL scores was poor, with intraclass correlation coefficients of 0.03 (95% CI, –0.09 to 0.15) for children 5 to 7 years old and 0.21 (95% CI, 0.03‐0.38) for children 8 to 12 years old. Higher child age and health literacy were associated with closer agreement between caregiver and child report. Conclusion Caregiver‐ and child‐reported global QOL in children with SDB was weakly correlated, more so for young children. In pediatric SDB, child‐perceived QOL may be poorer than that reported by caregivers. Further research is needed to assess whether similar trends exist for disease‐specific QOL metrics.
    Type of Medium: Online Resource
    ISSN: 0194-5998 , 1097-6817
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2008453-5
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  • 2
    In: The Laryngoscope, Wiley, Vol. 131, No. 4 ( 2021-04)
    Abstract: Subglottic stenosis (SGS) is the most common type of laryngeal stenosis in neonates. SGS severity is currently graded based on percent area of obstruction (%AO) via the Myer‐Cotton grading scale. However, patients with similar %AO can have widely different clinical courses. Computational fluid dynamics (CFD) based on patient‐specific imaging can quantify the relationship between airway geometry and flow dynamics. We investigated the effect of %AO and axial position of SGS on work of breathing (WOB) in neonates using magnetic resonance imaging. Methods High‐resolution ultrashort echo‐time MRI of the chest and airway was obtained in three neonatal patients with no suspected airway abnormalities; images were segmented to construct three‐dimensional (3D) models of the neonatal airways. These models were then modified with virtual SGSs of varying %AO and axial positioning. CFD simulations of peak inspiratory flow were used to calculate patient‐specific WOB in nonstenotic and artificially stenosed airway models. Results CFD simulations demonstrated a relationship between stenosis geometry and WOB increase. WOB rapidly increased with %AO greater than about 70%. Changes in axial position could also increase WOB by approximately the same amount as a 10% increase in %AO. Increased WOB was particularly pronounced when the SGS lumen was misaligned with the glottic jet. Conclusion The results indicate a strong, predictable relationship between WOB and axial position of the stenotic lumen relative to the glottis, which has not been previously reported. These findings may lead to precision diagnosis and treatment prediction tools in individual patients. Level of Evidence 4 Laryngoscope , 131:E1220–E1226, 2021
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2026089-1
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  • 3
    In: The Laryngoscope, Wiley, Vol. 131, No. 6 ( 2021-06)
    Abstract: To assess the ability of ultra‐short echo time (UTE)‐MRI to detect subglottic stenosis (SGS) and evaluate response to balloon dilation. To correlate measurements from UTE‐MRI with endotracheal‐tube (ETT)‐sizing and to investigate whether SGS causes change in airway dynamics. Study Design Animal research study. Methods Eight adult New‐Zealand white rabbits were used as they approximate neonatal airway‐size. The airways were measured using ETT‐sizing and 3D UTE‐MRI at baseline, 2 weeks post‐cauterization induced SGS injury, and post‐balloon dilation treatment. UTE‐MR images were acquired to determine airway anatomy and motion. Airways were segmented from MR images. Cross‐sectional area (CSA), major and minor diameters ( D major and D minor ), and eccentricity were measured. Results Post‐injury CSA at SGS was significantly reduced (mean 38%) compared to baseline ( P = .003) using UTE‐MRI. ETT‐sizing correlated significantly with MRI‐measured CSA at the SGS location ( r = 0.6; P   〈  .01), particularly at the post‐injury timepoint ( r = 0.93; P   〈  .01). Outer diameter from ETT‐sizing (OD) correlated significantly with D major ( r = 0.63; P   〈  .01) from UTE‐MRI at the SGS location, especially for the post‐injury timepoint ( r = 0.91; P   〈  .01). Mean CSA of upper trachea did not change significantly between end‐expiration and end‐inspiration at any timepoint (all P   〉  .05). Eccentricity of the upper trachea increased significantly post‐balloon dilation ( P   〈  .05). Conclusions UTE‐MRI successfully detected SGS and treatment response in the rabbit model, with good correlation to ETT‐sizing. Balloon dilation increased CSA at SGS, but not to baseline values. SGS did not alter dynamic motion for the trachea in this rabbit model; however, tracheas were significantly eccentric post‐balloon dilation. UTE‐MRI can detect SGS without sedation or ionizing radiation and may be a non‐invasive alternative to ETT‐sizing. Level of Evidence NA Laryngoscope , 131:E1971–E1979, 2021
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2026089-1
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  • 4
    In: Journal of Clinical Medicine, MDPI AG, Vol. 9, No. 11 ( 2020-11-20), p. 3723-
    Abstract: General anesthesia decreases the tone of upper airway muscles in a dose-dependent fashion, potentially narrowing the pharyngeal airway. We examined the effects of adding ketamine on the airway configuration after dexmedetomidine administration in spontaneously breathing children with normal airways. 25 children presenting for Magnetic Resonance Imaging (MRI) of the brain/spine under general anesthesia were prospectively recruited in the study. Patients were anesthetized with dexmedetomidine bolus (2 mcg over 10 min) followed by dexmedetomidine infusion (2 mcg·kg−1·h) and ketamine and permitted to breathe spontaneously via the native airway. MR-CINE images of the upper airway were obtained with dexmedetomidine infusion alone (baseline) and 5, 10, and 15 min after administering ketamine bolus (2 mg·kg−1) in two anatomical axial planes at the nasopharynx and the retroglossal upper airway. Airway lumen is segmented with a semi-automatic image processing approach using a region-growing algorithm. Outcome measures of cross-sectional area, transverse and anterior-posterior diameters of the airway in axial planes at the level of the epiglottis in the retroglossal airway, and in the superior nasopharynx were evaluated for changes in airway size with sedation. Airway dimensions corresponding to the maximum, mean, and minimum sizes during a respiratory cycle were obtained to compare the temporal changes in the airway size. The dose-response of adding ketamine to dexmedetomidine alone condition on airway dimensions were examined using mixed-effects of covariance models. 22/25 patients based on inclusion/exclusion criteria were included in the final analysis. The changes in airway measures with the addition of ketamine, when compared to the baseline of dexmedetomidine alone, were statistically insignificant. The modest changes in airway dimensions are clinically less impactful and within the accuracy of the semi-automatic airway segmentation approach. The effect sizes were small for most airway measures. The duration of ketamine seems to not affect the airway size. In conclusion, adding ketamine to dexmedetomidine did not significantly reduce upper airway configuration when compared to dexmedetomidine alone.
    Type of Medium: Online Resource
    ISSN: 2077-0383
    Language: English
    Publisher: MDPI AG
    Publication Date: 2020
    detail.hit.zdb_id: 2662592-1
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  • 5
    In: Sleep, Oxford University Press (OUP), Vol. 45, No. 5 ( 2022-05-12)
    Abstract: Obstructive sleep apnea is associated with neurobehavioral dysfunction, but the relationship between disease severity as measured by the apnea-hypopnea index and neurobehavioral morbidity is unclear. The objective of our study is to compare the neurobehavioral morbidity of mild sleep-disordered breathing versus obstructive sleep apnea. Methods Children 3–12 years old recruited for mild sleep-disordered breathing (snoring with obstructive apnea-hypopnea index & lt; 3) into the Pediatric Adenotonsillectomy Trial for Snoring were compared to children 5–9 years old recruited for obstructive sleep apnea (obstructive apnea-hypopnea 2–30) into the Childhood Adenotonsillectomy Trial. Baseline demographic, polysomnographic, and neurobehavioral outcomes were compared using univariable and multivariable analysis. Results The sample included 453 participants with obstructive sleep apnea (median obstructive apnea-hypopnea index 5.7) and 459 participants with mild sleep-disordered breathing (median obstructive apnea-hypopnea index 0.5). By polysomnography, participants with obstructive sleep apnea had poorer sleep efficiency and more arousals. Children with mild sleep-disordered breathing had more abnormal executive function scores (adjusted odds ratio 1.96, 95% CI 1.30–2.94) compared to children with obstructive sleep apnea. There were also elevated Conners scores for inattention (adjusted odds ratio 3.16, CI 1.98–5.02) and hyperactivity (adjusted odds ratio 2.82, CI 1.83–4.34) in children recruited for mild sleep-disordered breathing. Conclusions Abnormal executive function, inattention, and hyperactivity were more common in symptomatic children recruited into a trial for mild sleep-disordered breathing compared to children recruited into a trial for obstructive sleep apnea. Young, snoring children with only minimally elevated apnea-hypopnea levels may still be at risk for deficits in executive function and attention. Trial Registration Pediatric Adenotonsillectomy for Snoring (PATS), NCT02562040; Childhood Adenotonsillectomy Trial (CHAT), NCT00560859
    Type of Medium: Online Resource
    ISSN: 0161-8105 , 1550-9109
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2022
    detail.hit.zdb_id: 2056761-3
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  • 6
    In: BioMed Research International, Hindawi Limited, Vol. 2022 ( 2022-2-25), p. 1-13
    Abstract: This study proposes a Bayesian joint model with extended random effects structure that incorporates nested repeated measures and provides simultaneous inference on treatment effects over time and drop-out patterns. The proposed model includes flexible splines to characterize the circadian variation inherent in blood pressure sequences, and we assess the effectiveness of an intervention to resolve pediatric obstructive sleep apnea. We demonstrate that the proposed model and its conventional two-stage counterpart provide similar estimates of nighttime blood pressure but estimates on the mean evolution of daytime blood pressure are discrepant. Our simulation studies tailored to the motivating data suggest reasonable estimation and coverage probabilities for both fixed and random effects. Computational challenges of model implementation are discussed.
    Type of Medium: Online Resource
    ISSN: 2314-6141 , 2314-6133
    Language: English
    Publisher: Hindawi Limited
    Publication Date: 2022
    detail.hit.zdb_id: 2698540-8
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  • 7
    In: Stat, Wiley, Vol. 9, No. 1 ( 2020-01)
    Abstract: In longitudinal studies in which a medical device is used to monitor outcome repeatedly and frequently on the same patients over a prespecified duration of time, two clustering goals can arise. One goal is to assess the degree of heterogeneity among patient profiles. A second yet equally important goal unique to such studies is to determine frequency and duration of monitoring sufficient to identify longitudinal changes. Considering these goals jointly would identify clusters of patients who share similar patterns over time and characterize temporal stability within each cluster. We use a biclustering approach, allowing simultaneous clustering of observations at both patient and time levels and using a nonparametric hierarchical Bayesian model. Because clustering units at the time level (i.e., time points) are ordered and hence unexchangeable, we utilize a multivariate Dirichlet process mixture model by specifying a Dirichlet process prior at the patient level whose base measure employs change points at the time level to achieve the desired joint clustering. We consider structured covariance between consecutive time points and assess model performance through simulation studies. We apply the model to data on 24‐hr ambulatory blood pressure monitoring and examine the relationship between diastolic blood pressure and pediatric obstructive sleep apnoea.
    Type of Medium: Online Resource
    ISSN: 2049-1573 , 2049-1573
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2020
    detail.hit.zdb_id: 2687133-6
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  • 8
    In: Learning Health Systems, Wiley, Vol. 7, No. 3 ( 2023-07)
    Abstract: A learning health network is a type of learning health system in which stakeholders use network organization to improve health and health care. Building on existing resources in the cystic fibrosis (CF) community, the Cystic Fibrosis Learning Network (CFLN) was designed to improve medical outcomes and quality of life through an intentional focus on achieving reliable evidence‐based chronic care delivery and creating a system for data‐driven collaborative learning. Methods We describe the development and growth of the CFLN considering six domains of a Network Maturity Grid: system leadership; governance and policy management; quality improvement (QI); engagement and community building; data and analytics; and research. We illustrate the impact of the CFLN experience on chronic care processes and indicators of collaborative infrastructure. Results The CFLN represents 36 accredited care centers in the CF Foundation Care Center Network caring for over 6300 patients. Of 6779 patient clinical care visits/quarter, 77% are entered into the CF Foundation Patient Registry within 30 days, providing timely means to track outcomes. Collaborative visit planning is occurring in 93% of clinical care visits to share agenda setting with patients and families. Almost all CFLN teams (94%, n = 34) have a patient/family partner (PFP), and 74% of PFPs indicate they are actively participating, taking ownership of, or leading QI initiatives with the interdisciplinary care team. In 2022, 97% of centers reported completing 1–13 improvement cycles per month, and 82% contributed to monthly QI progress reports to share learning. Conclusion The CFLN is a maturing, collaborative infrastructure. CFLN centers practice at an advanced level of coproduction. The CFLN fosters interdisciplinary and PFP leadership and the performance of consistent data‐driven improvement cycles. CFLN centers are positioned to respond to rapid changes in evidence‐based care and advance the practice of QI and implementation science on a broader scale.
    Type of Medium: Online Resource
    ISSN: 2379-6146 , 2379-6146
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2868158-7
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  • 9
    In: Learning Health Systems, Wiley, Vol. 7, No. 3 ( 2023-07)
    Abstract: The Cystic Fibrosis (CF) Foundation sponsored the design, pilot testing, and implementation of the CF Learning Network (CFLN) to explore how the Foundation's Care Center Network (CCN) could become a learning health system. Six years after the design, the Foundation commissioned a formative mixed methods evaluation of the CFLN to assess: CFLN participants' understanding of program goals, attributes, and perceptions of current and future impact. Methods We performed semi‐structured interviews with CFLN participants to identify perceived goals, attributes, and impact of the network. Following thematic analyses, we developed and distributed a survey to CFLN members and a matched sample of CCN programs to understand whether the themes were unique to the CFLN. Results Interviews with 24 CFLN participants were conducted. Interviewees identified the primary CFLN goal as improving outcomes for people living with CF, with secondary goals of providing training in quality improvement (QI), creating a learning community, engaging all stakeholders in improvement, and spreading best practices to the CCN. Project management, use of data, common QI methods, and the learning community were seen as critical to success. Survey responses were collected from 103 CFLN members and 25 CCN members. The data revealed that CFLN respondents were more likely than CCN respondents to connect with other CF programs, routinely use data for QI, and engage patient and family partners in QI. Conclusions Our study suggests that the CFLN provides value beyond that achieved by the CCN. Key questions remain about whether spread of the CFLN could improve outcomes for more people living with CF.
    Type of Medium: Online Resource
    ISSN: 2379-6146 , 2379-6146
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2868158-7
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  • 10
    In: BMJ Open Quality, BMJ, Vol. 11, No. 2 ( 2022-05), p. e001844-
    Abstract: The Cystic Fibrosis Foundation chronic care guidelines recommend monitoring clinical status of a patient with cystic fibrosis (CF) through quarterly interdisciplinary visits. At the beginning of the COVID-19 pandemic, the Cystic Fibrosis Learning Network (CFLN) designed and initiated a telehealth (TH) innovation lab (TH ILab) to support transition from the classic CF care model of quarterly in-person office visits to a care model that included TH. Aim The specific aims of the TH ILab were to increase the percentage of virtual visits with interdisciplinary care (IDC) from 60% to 85% and increase the percentage of virtual visits in which patients and families participated in shared agenda setting (AS) from 52% to 85% by 31 December 2020. Methods The model for improvement methodology was used to determine the ILab aims, theory, interventions and measures. In the testing phase of the ILab, data related to process and outcome measures as well as learnings from plan–do–study–act cycles were collected, analysed and shared weekly with the TH ILab teams. Participating centres created processes for IDC and AS for TH visits and developed and shared quality improvement tools specific to their local context with other centres during the ILab weekly meetings and via a secure CFLN-maintained platform. Results Both specific aims were achieved ahead of the expected target date. By August 2020, 85% of the TH ILab visits provided IDC and 92% of patients were seen for CF care by teams from the TH ILab that participated in AS. Conclusion Shared learning through a collaborative, data-driven process in the CFLN TH ILab rapidly led to standardised TH IDC and AS, which achieved reliable and sustainable processes which could be reproduced by other networks.
    Type of Medium: Online Resource
    ISSN: 2399-6641
    Language: English
    Publisher: BMJ
    Publication Date: 2022
    detail.hit.zdb_id: 2952859-8
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