Publications by authors named "Ignacio E Tapia"

59 Publications

Continuous positive airway pressure care for pediatric obstructive sleep apnea: A long-term quality improvement initiative.

Pediatr Pulmonol 2022 Jul 13. Epub 2022 Jul 13.

Sleep Center, Division of Pulmonary and Sleep Medicine, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

Successful treatment of pediatric obstructive sleep apnea syndrome (OSAS) with continuous positive airway pressure (CPAP) is challenging due to behavioral, technical, medical, and systems factors. We undertook a quality improvement (QI) initiative involving physicians, nurses, psychologists, and respiratory therapists to improve CPAP outpatient care and processes. We aimed to: (1) increase the proportion of patients with a follow-up visit within 4 months of initiation of CPAP, (2) reduce the median time to first follow-up visit to under 4 months, and (3) increase the proportion of patients obtaining a post-initiation polysomnogram within 1 year to >50%. We also explored healthcare utilization (HCU) in a subsample of patients. Interventions focused on developing a tracking system and standardizing interdisciplinary clinical care. The proportion of patients returning to clinic within 4 months improved from 38.2% to 65.5% and median time to first follow-up visit improved from 133 to 56 days. The percentage of patients who returned for a post-initiation polysomnogram within 1 year was 71.1%. Subsample analyses showed significant reductions in the length of stay for emergency department visits from pre-CPAP initiation (Mdn = 3.00 h; interquartile range [IQR] = 7.00) to post-initiation (Mdn = 2.00 h, IQR = 5.00). The length of hospitalizations was also significantly shorter from pre (Mdn = 48.00 h, IQR = 243.00) to post-CPAP initiation (Mdn = 0.00 h, IQR = 73.00). A standardized, tracked approach to interdisciplinary outpatient CPAP care can improve follow-up care and potentially HCU.
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http://dx.doi.org/10.1002/ppul.26075DOI Listing
July 2022

A Trial of Intranasal Corticosteroids to Treat Childhood OSA Syndrome.

Chest 2022 Jun 30. Epub 2022 Jun 30.

Department of Pediatrics, Division of Developmental and Behavioral Pediatrics, Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine, Philadelphia, PA.

Background: Intranasal corticosteroids (INCS) are frequently used to treat OSA syndrome (OSAS) in children. However, their efficacy has not been rigorously tested.

Research Question: Do INCS result in improved OSAS symptoms, polysomnography findings, behavior, and quality of life compared with placebo?

Study Design And Methods: In this randomized, double-blind, placebo-controlled trial, children with OSAS aged 5 to 12 years (N = 134) were randomized 2:1 to receive 3 months of INCS or placebo. Children in the INCS arm were then re-randomized to receive 9 months of INCS or placebo. Polysomnography, symptoms, and neurobehavioral findings were measured at baseline, 3 months, and 12 months. The primary outcome was change in obstructive apnea hypopnea index (OAHI) at 3 months, available for 122 children. The secondary outcome was OAHI change at 12 months, available for 70 children.

Results: Median (interquartile range) age and OAHI at baseline for the entire group were 7.9 (6.3 to 9.9) years and 5.8 (3.6 to 9.7) events per hour. OAHI changes at 3 months (-1.72 [-3.91 to 1.92] events per hour) and 12 months (-1.2 [-4.22 to 1.71] events per hour) were not different between the two groups (P = not significant). OSAS symptoms and neurobehavioral results did not differ between the INCS and placebo groups at 3 and 12 months. The 38 children who received INCS for 12 months reported a significant OAHI decrease from 7.2 (3.62 to 9.88) events per hour to 3.7 (1.56 to 6.4) events per hour (P = .039).

Interpretation: In children with OSAS, treatment with INCS did not result in significant polysomnography, neurobehavioral, or symptom changes at 3 and 12 months of treatment. Twelve months of INCS treatment resulted in a statistically significant but not clinically relevant OAHI reduction.

Clinical Trial Registration: ClinicalTrials.gov; No.: NCT02180672; URL: www.

Clinicaltrials: gov.
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http://dx.doi.org/10.1016/j.chest.2022.06.026DOI Listing
June 2022

Pediatric pulmonary year in review 2021: Sleep medicine.

Pediatr Pulmonol 2022 Jul 2. Epub 2022 Jul 2.

Sleep Center, Division of Pulmonary and Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

Pediatric pulmonology publishes original research, review articles, and case reports on a wide variety of pediatric respiratory disorders. In this article, we summarized the past year's publications in sleep medicine and reviewed selected literature from other journals in this field. We focused on original research articles exploring aspects of sleep-disordered breathing in patients with underlying conditions such as cystic fibrosis, asthma, and sickle cell disease. We also explored sleep-disordered breathing risk factors, monitoring, diagnosis, and treatment; and included recent recommendations for drug-induced sleep endoscopy and ways to monitor and improve PAP adherence remotely.
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http://dx.doi.org/10.1002/ppul.26047DOI Listing
July 2022

Neuroendocrine cell hyperplasia of infancy: Feasibility of objective evaluation with quantitative CT.

Clin Imaging 2022 Jun 8;89:43-48. Epub 2022 Jun 8.

Department of Radiology, Children's Hospital of Philadelphia, USA; Perelman School of Medicine, University of Pennsylvania, USA.

Objective: To describe quantitative CT parameters of children with a typical pattern for NEHI and compare them to controls.

Materials And Methods: Eleven patients (7 boys) with NEHI and an available chest CT concordant NEHI were identified. Eleven age-, sex-, height-matched, with CT technique-matching were identified for comparison. An open-source software was used to segment the lung parenchyma into lobes using the fissures. Quantitative parameters such as low attenuation areas, mean lung density, kurtosis, skewness, ventilation heterogeneity, lung mass, and volume were calculated for both controls and cases.

Results: Analysis of the lung parenchyma showed that patients with NEHI had a lower mean lung density (-615 HU vs -556 HU, p = 0.03) with higher ventilation heterogeneity (0.23 vs 0.19, p = 0.04), lung mass (232 g vs 146 g, p = 0.01) and volume (595 mL vs 339 mL, p = 0.008) compared to controls. Most lobes followed this trend, except the middle lobe that showed only a higher lung mass (32.9 g vs 19.6 g, p = 0.02) and volume (77.4 vs 46.9, p = 0.005) in patients with NEHI compared to controls.

Conclusion: Quantitative CT is a feasible technique in children with a typical pattern for NEHI and is associated with differences in attenuation, ventilation heterogeneity, and lung volume.
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http://dx.doi.org/10.1016/j.clinimag.2022.06.004DOI Listing
June 2022

Sleep Deficiency in Adolescents: The School Start Time Debate.

Clin Chest Med 2022 06;43(2):239-247

Division of Pulmonary and Sleep Medicine, Children's Hospital of Philadelphia, 3401 Civic Center Boulevard, Philadelphia, PA 19104, USA.

Adolescence is commonly accepted as a challenging time for sleep, with multiple factors contributing to sleep deficiency in adolescents. These include physiologic changes with shifts in their circadian rhythm; medical sleep disorders; and social, cultural, and environmental factors. Early school start times negatively affect sleep in adolescents as well, with poorer outcomes in their overall health, wellbeing, and performance. This article highlights the different contributing factors for sleep deficiency in adolescents and the consequences of sleep deficiency. In addition, the authors discuss the impact of delayed school start times in improving adolescents' sleep and overall function.
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http://dx.doi.org/10.1016/j.ccm.2022.02.008DOI Listing
June 2022

Neurobehavioral morbidity of pediatric mild sleep-disordered breathing and obstructive sleep apnea.

Sleep 2022 05 12;45(5). Epub 2022 Feb 12.

Brigham and Women's Hospital, Division of Sleep and Circadian Disorders, Boston, MA, USA.

Study Objectives: 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 < 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.
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http://dx.doi.org/10.1093/sleep/zsac035DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9113015PMC
May 2022

Conducting clinical trials in persons with Down syndrome: summary from the NIH INCLUDE Down syndrome clinical trials readiness working group.

J Neurodev Disord 2022 03 23;14(1):22. Epub 2022 Mar 23.

Alzheimer's Therapeutic Research Institute, Keck School of Medicine, University of Southern California, Los Angeles, USA.

The recent National Institute of Health (NIH) INCLUDE (INvestigation of Co-occurring conditions across the Lifespan to Understand Down syndromE) initiative has bolstered capacity for the current increase in clinical trials involving individuals with Down syndrome (DS). This new NIH funding mechanism offers new opportunities to expand and develop novel approaches in engaging and effectively enrolling a broader representation of clinical trials participants addressing current medical issues faced by individuals with DS. To address this opportunity, the NIH assembled leading clinicians, scientists, and representatives of advocacy groups to review existing methods and to identify those areas where new approaches are needed to engage and prepare DS populations for participation in clinical trial research. This paper summarizes the results of the Clinical Trial Readiness Working Group that was part of the INCLUDE Project Workshop: Planning a Virtual Down Syndrome Cohort Across the Lifespan Workshop held virtually September 23 and 24, 2019.
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http://dx.doi.org/10.1186/s11689-022-09435-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8942061PMC
March 2022

Health disparities in pediatric sleep-disordered breathing.

Paediatr Respir Rev 2022 Jan 28. Epub 2022 Jan 28.

Sleep Center, Division of Pulmonary and Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA; Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA. Electronic address:

Sleep-disordered breathing reflects a continuum of overnight breathing difficulties, ranging from mild snoring to obstructive sleep apnea syndrome. Sleep-disordered breathing in childhood is associated with significant adverse outcomes in multiple domains of functioning. This review summarizes the evidence of well-described ethnic, racial, and socioeconomic disparities in pediatric sleep-disordered breathing, from its prevalence to its treatment-related outcomes. Research on potential socio-ecological contributors to these disparities is also reviewed. Critical future research directions include the development of interventions that address the modifiable social and environmental determinants of these health disparities.
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http://dx.doi.org/10.1016/j.prrv.2022.01.005DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9329494PMC
January 2022

Comparison of Caregiver- and Child-Reported Quality of Life in Children With Sleep-Disordered Breathing.

Otolaryngol Head Neck Surg 2022 Mar 8:1945998221083288. Epub 2022 Mar 8.

Department of Otolaryngology, Eastern Virginia Medical School; Department of Pediatric Sleep Medicine, Children's Hospital of The King's Daughters, Norfolk, Virginia, USA.

Objective: 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 ( < .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.
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http://dx.doi.org/10.1177/01945998221083288DOI Listing
March 2022

Clinical utility of repeated positive airway pressure titrations in children with obstructive sleep apnea syndrome.

J Clin Sleep Med 2022 04;18(4):1021-1026

Division of Pulmonary & Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

Study Objectives: Positive airway pressure (PAP) is the second line of treatment for obstructive sleep apnea syndrome in children. It is common practice following initiation of PAP to perform repeat titration polysomnography to re-evaluate the patient's therapeutic pressure; however, data supporting this practice are lacking. We hypothesized that repeat PAP titration would result in significant setting changes in children with obstructive sleep apnea syndrome.

Methods: We retrospectively analyzed demographic, polysomnographic, and PAP data of children with obstructive sleep apnea syndrome aged 0-18 years who were initiated on PAP and underwent 2 titration studies over a 2-year period. PAP mode and recommended pressure differences between the 2 titrations were compared.

Results: 64 children met inclusion criteria. The median (interquartile range) baseline obstructive apnea-hypopnea index and SpO nadir were 14.8 (8.7-32.7) events/h and 88.5% (85-92%), respectively. The mean differences in obstructive apnea-hypopnea index, SpO nadir, and % total sleep time with SpO < 90% between both titrations were negligible, including children with obesity, adenotonsillar hypertrophy, and trisomy 21. Additionally, there was no significant difference in mean PAP pressure between 2 separate titration studies for those on continuous PAP or bilevel PAP.

Conclusions: Overall, repeat PAP titration in children with obstructive sleep apnea syndrome within the timeframe here described did not result in significant changes in PAP mode, continuous PAP pressure, or obstructive apnea-hypopnea index. Based on these data, repeat PAP titration within 2 years of an initial titration does not appear to be necessary.

Citation: Yendur O, Feld L, Miranda-Schaeubinger M, et al. Clinical utility of repeated positive airway pressure titrations in children with obstructive sleep apnea syndrome. . 2022;18(4):1021-1026.
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http://dx.doi.org/10.5664/jcsm.9790DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8974391PMC
April 2022

More than just a wheeze: bronchiolitis and obstructive sleep apnea in children.

Sleep 2021 12;44(12)

Division of Pulmonary & Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

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http://dx.doi.org/10.1093/sleep/zsab227DOI Listing
December 2021

Opportunities, barriers, and recommendations in down syndrome research.

Transl Sci Rare Dis 2021 15;5(3-4):99-129. Epub 2021 Apr 15.

Waisman Center, University of Wisconsin-Madison, Madison, WI, USA.

Background: Recent advances in medical care have increased life expectancy and improved the quality of life for people with Down syndrome (DS). These advances are the result of both pre-clinical and clinical research but much about DS is still poorly understood. In 2020, the NIH announced their plan to update their DS research plan and requested input from the scientific and advocacy community.

Objective: The National Down Syndrome Society (NDSS) and the LuMind IDSC Foundation worked together with scientific and medical experts to develop recommendations for the NIH research plan.

Methods: NDSS and LuMind IDSC assembled over 50 experts across multiple disciplines and organized them in eleven working groups focused on specific issues for people with DS.

Results: This review article summarizes the research gaps and recommendations that have the potential to improve the health and quality of life for people with DS within the next decade.

Conclusions: This review highlights many of the scientific gaps that exist in DS research. Based on these gaps, a multidisciplinary group of DS experts has made recommendations to advance DS research. This paper may also aid policymakers and the DS community to build a comprehensive national DS research strategy.
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http://dx.doi.org/10.3233/trd-200090DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8279178PMC
April 2021

Neck fat and obstructive sleep apnea in obese adolescents.

Sleep 2021 11;44(11)

University of Pennsylvania, Philadelphia, PA.

Study Objectives: Increased neck circumference, a surrogate for the neck fat that can narrow the upper airway in obese individuals, is a risk factor for obstructive sleep apnea syndrome (OSAS) in adults, but the association between neck fat and OSAS in adolescent males and females is unknown. We hypothesized that obese adolescents with OSAS have more neck fat than controls, females more neck fat than males, and that neck fat correlates with obesity and OSAS severity.

Methods: Obese adolescents with OSAS and obese and normal-weight controls underwent upper airway magnetic resonance imaging, polysomnography, and anthropometrics, including neck circumference measurement. Intra-neck and subcutaneous neck fat measurements were manually segmented and compared among the three groups using ANOVA and between males and females using t-tests. The relationship between polysomnographic parameters and neck fat measurements was assessed in adolescents with OSAS using Pearson correlations.

Results: One-hundred nineteen adolescents (38 females) were studied: 39 obese with OSAS, 34 obese controls, and 46 normal-weight controls. Neck fat was not greater in adolescents with OSAS compared to obese controls (p=0.35), and neck fat volume was not related to OSAS severity (p = 0.36). However, obese adolescents had more neck fat than normal-weight controls (p < 0.001), and neck fat volume correlated with neck circumference (r = 0.53, p < 0.001). Females had significantly greater cross-sectional neck fat than males (p < 0.001).

Conclusions: While neck fat is associated with obesity and neck circumference in adolescents and is greater in females versus males, it does not appear to correlate with presence and severity of OSAS.
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http://dx.doi.org/10.1093/sleep/zsab158DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8598172PMC
November 2021

The impact of obstructive sleep apnea on bronchiolitis severity in children with Down syndrome.

Sleep Med 2021 07 16;83:188-195. Epub 2021 Apr 16.

Sleep Center, Children's Hospital of Philadelphia, Philadelphia, PA, USA; Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA. Electronic address:

Objectives: Acute bronchiolitis commonly causes respiratory failure in children ≤2 years, and is particularly severe in those with Down syndrome (DS). Obstructive sleep apnea (OSA), common in DS, is also associated with respiratory complications. However, it is unknown whether OSA is associated with worse outcomes in children with and without DS, hospitalized with bronchiolitis. We hypothesized that in children with bronchiolitis, OSA is associated with worse outcomes in those with DS, independent of DS-related comorbidities.

Methods: Hospital discharge records of children with bronchiolitis aged ≤2 years were obtained for 1997-2012 from the Kid's Inpatient Database. The primary outcome was invasive mechanical ventilation (IMV), and secondary outcomes were non-invasive mechanical ventilation (NIMV), length of hospital stay, and inflation-adjusted cost of hospitalization (IACH). Multivariable regression was conducted to ascertain the associations between OSA and primary and secondary outcomes accounting for DS-associated comorbidities.

Results: There were 928,961 hospitalizations for bronchiolitis. The DS group with bronchiolitis (n = 8697) was more likely to have OSA [241 (2.77%) vs 1293 (0.14%), p < 0.001] compared to the non-DS group (n = 920,264). Multivariable logistic regression showed that OSA was associated with IMV (adjusted odds ratio [OR], 3.32 [95% CI 2.54-4.35], p < 0.0001) in all children with bronchiolitis; and in those with DS, it was associated with IMV (adjusted OR, 2.34 [95% CI 1.38-3.97], p = 0.002), NIMV (adjusted OR, 8.21 [95% CI 4.48-15.04], p < 0.0001) and IACH (adjusted β, 0.18 [95% CI 0.02-0.34], p = 0.031).

Conclusions: OSA is independently associated with assisted ventilation in all children hospitalized with bronchiolitis, regardless of DS-associated comorbidities in those with DS. The severity of bronchiolitis in children with DS may be driven by the high prevalence of OSA.
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http://dx.doi.org/10.1016/j.sleep.2021.04.004DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8205980PMC
July 2021

Polysomnographic predictors of abnormal brainstem imaging in children.

J Clin Sleep Med 2021 07;17(7):1411-1421

Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania.

Study Objectives: Evaluation of elevated central apnea-hypopnea index (CAHI) or prolonged central apneas in pediatric patients typically includes neuroimaging with a focus on brainstem pathology. There is little evidence guiding thresholds of polysomnographic variables that accurately predict abnormal neuroimaging. We sought to evaluate whether additional polysomnographic variables may help predict brainstem pathology.

Methods: A 10-year retrospective review of patients ages 1-18 years who received a brain magnetic resonance imaging (MRI) for an indication of central sleep apnea diagnosed via polysomnography was performed. Demographics, medical history, polysomnogram variables, and MRI results were compared.

Results: This study included 65 patients (69.2% male). The median age was 5.8 years (interquartile range, 3.0-8.3). Most patients had negative (normal or nonsignificant) MRIs (n = 45, 69.2%); 20 (30.8%) had abnormal MRIs. Of the patients with abnormal MRIs, 13 (20.0%) had abnormalities unrelated to the brainstem. Seven patients (10.8%) were found to have brainstem pathology and had a median CAHI of 10.8 events/h (interquartile range, 6.5-21.9), and three of seven (42.9%) had hypoventilation and were more likely to have developmental delay, abnormal neurological examinations, and reflux. Other patients (n = 58) had a median CAHI of 5.6 events/h (interquartile range, 3.1-9.1), and seven (12.1%) had hypoventilation. Area under the curve and receiver operating characteristic curves showed a CAHI ≥ 9.5 events/h and ≥ 6.4% of total sleep time with end-tidal CO₂ ≥ 50 mm Hg predicted abnormal brainstem imaging. Prolonged central apneas did not predict abnormal brainstem imaging.

Conclusions: Most patients with central sleep apnea do not have MRIs implicating structurally abnormal brainstems. Utilizing a cutoff of CAHI of ≥ 9.5 events/h, ≥ 6.4% total sleep time with end-tidal CO₂ ≥ 50 mm Hg and/or frank hypoventilation, and additional clinical history may optimize MRI utilization in patients with central sleep apnea.
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http://dx.doi.org/10.5664/jcsm.9210DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8314628PMC
July 2021

Positive airway pressure for the treatment of the childhood obstructive sleep apnea syndrome.

Pediatr Pulmonol 2022 08 1;57(8):1897-1903. Epub 2021 Mar 1.

Division of Pulmonary and Sleep Medicine, Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

In this review, we have summarized the benefits of treatment with positive airway pressure for the childhood obstructive sleep apnea syndrome and presented a socio-ecological framework to enhance our understanding of positive airway pressure adherence predictors and important targets of comprehensive positive airway pressure treatment models across different pediatric populations. Although positive airway pressure is clearly a beneficial treatment for pediatric obstructive sleep apnea syndrome, additional research is needed to evaluate how socio-ecological factors may interact to predict positive airway pressure adherence, with more attention to the impact of the broader healthcare setting and on treatment approaches and outcomes in special pediatric populations.
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http://dx.doi.org/10.1002/ppul.25318DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8408267PMC
August 2022

Normal age-related quantitative CT values in the pediatric lung: from the first breath to adulthood.

Clin Imaging 2021 Jul 4;75:111-118. Epub 2021 Jan 4.

Department of Radiology, Children's Hospital of Philadelphia, USA.

Objective: To characterize the normal progression of quantitative CT parameters in normal children from birth to adulthood.

Materials And Methods: Patients aged 0-18 years with non-contrast-enhanced chest CT and evidence of normal lung parenchyma were included. Patients with respiratory symptoms, incomplete anthropometric measurements, or sub-optimal imaging technique were excluded. Segmentation was performed using an open-source software with an automated threshold segmentation. The following parameters were obtained: mean lung density, kurtosis, skewness, lung volume, and mass. Linear and exponential regression models were calculated with age and height as independent variables. A p-value of <0.05 was considered significant.

Results: 220 patients (111 females, 109 males) were included. Mean age was 9.6 ± 5.9 years and mean height was 133.9 ± 35.1 cm. Simple linear regression showed a significant relationship between mean lung density with age (R 2 = 0.70) and height (R 2 = 0.73). Kurtosis displayed a significant exponential correlation with age (R 2 = 0.70) and height (R 2 = 0.71). Skewness showed a significant exponential correlation with age (R 2 = 0.71) and height (R 2 = 0.73). Lung mass showed a correlation with age (R 2 = 0.93) and height (R 2 = 0.92). Exponential regression showed a significant relationship between lung volume with age (R 2 = 0.88) and height (R 2 = 0.93).

Conclusion: Quantitative CT parameters of the lung parenchyma demonstrate changes from birth to adulthood. As children grow, the mean lung density decreases, and the lung parenchyma becomes more homogenous.
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http://dx.doi.org/10.1016/j.clinimag.2020.12.021DOI Listing
July 2021

Impact of obstructive sleep apnea on assisted ventilation in children with asthma exacerbation.

Pediatr Pulmonol 2021 05 11;56(5):1103-1113. Epub 2021 Jan 11.

Sleep Center, Division of Pulmonary and Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

Objective: To determine the impact of obstructive sleep apnea (OSA) on asthma exacerbation severity in children hospitalized for asthma exacerbation.

Hypothesis: OSA is associated with greater use of invasive mechanical ventilation (IMV) and noninvasive mechanical ventilation (NIMV) in children hospitalized for asthma exacerbation.

Study Design: A retrospective cohort study.

Patient-subject Selection: Hospitalization records of children aged 2-18 years admitted for acute asthma exacerbation were obtained for 2000, 2003, 2006, 2009, and 2012 from the Kids' Inpatient Database.

Methodology: The primary exposure was OSA, the primary outcome was IMV, and secondary outcomes were NIMV, length of hospital stay (LOS), and inflation-adjusted cost of hospitalization. Multivariable logistic regression, negative binomial, and linear regression were conducted to ascertain the impact of OSA on primary and secondary outcomes. Exploratory analyses investigated the impact of obesity on primary and secondary outcomes.

Results: Among 564,467 hospitalizations for acute asthma exacerbation, 4209 (0.75%) had OSA. Multivariable regression indicated that OSA was associated with IMV (adjusted odds ratio [OR], 5.33 [95% confidence interval, CI: 4.35-6.54], p < .0001), NIMV (adjusted OR, 8.30 [95% CI: 6.56-10.51], p < .0001), longer LOS (adjusted incidence rate ratio, 1.34 [95% CI 1.28-1.43], p < .0001), and greater inflation-adjusted cost of hospitalization (adjusted β, 0.38 [95% CI: 0.33-0.43], p < .0001). Obesity was also significantly associated IMV, NIMV, longer LOS, and greater inflation-adjusted cost of hospitalization. There was no interaction between OSA and obesity.

Conclusion: OSA is an independent risk factor for IMV, NIMV, longer LOS, and elevated inflation-adjusted costs of hospitalization in children hospitalized for asthma exacerbation.
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http://dx.doi.org/10.1002/ppul.25247DOI Listing
May 2021

Disparities in Sleep Health and Potential Intervention Models: A Focused Review.

Chest 2021 03 30;159(3):1232-1240. Epub 2020 Sep 30.

Division of Pulmonary, Critical Care, and Sleep Medicine, West Virginia University, WV. Electronic address:

Disparities in sleep health are important but underrecognized contributors to health disparities. Understanding the factors contributing to sleep heath disparities and developing effective interventions are critical to improving all aspects of heath. Sleep heath disparities are impacted by socioeconomic status, racism, discrimination, neighborhood segregation, geography, social patterns, and access to health care as well as by cultural beliefs, necessitating a cultural appropriateness component in any intervention devised for reducing sleep health disparities. Pediatric sleep disparities require innovative and urgent intervention to establish a foundation of lifelong healthy sleep. Tapping the vast potential of technology in improving sleep health access may be an underutilized tool to reduce sleep heath disparities. Identifying, implementing, replicating, and disseminating successful interventions to address sleep disparities have the potential to reduce overall disparities in health and quality of life.
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http://dx.doi.org/10.1016/j.chest.2020.09.249DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7525655PMC
March 2021

The burden of obstructive sleep apnea in pediatric sickle cell disease: a Kids' inpatient database study.

Sleep 2021 02;44(2)

Sleep Center, Children's Hospital of Philadelphia, Philadelphia, PA.

Study Objectives: Obstructive sleep apnea (OSA) is associated with cardiovascular and cerebrovascular morbidity. Patients with sickle cell disease (SCD) are at increased risk for both neurologic complications (NC) and OSA. However, the relationship between OSA and SCD complications is unclear. We hypothesized that there would be an association between OSA diagnosis and SCD complications.

Methods: Hospital discharge records of patients with SCD aged < 19 years were obtained for the years 1997, 2000, 2003, 2006, 2009, and 2012 from the Kid's Inpatient Database. The primary outcome, NC, a composite of stroke, transient ischemic attack, and seizures. Secondary outcomes included acute chest syndrome (ACS), vaso-occlusive crisis, length of hospital stay, and inflation-adjusted cost of hospitalization. Multivariable regression was conducted to ascertain the association of OSA with primary and secondary outcomes. Analyses were adjusted for the use of noninvasive mechanical ventilation (NIMV) to determine its role as NC risk modifier.

Results: There were 203,705 SCD discharges included in the analysis, of which 2,820 (1.4%) and 4,447 (2.2%) also included OSA and NC diagnoses. Multivariable logistic regression indicated that OSA was associated with NC (adjusted odds ratio [OR], 1.50 [95% CI 1.02-2.21], p = 0.039) and ACS (OR, 1.34 [95% CI 1.08-1.67], p = 0.009) in children with SCD. In the multivariable analysis adjusted for NIMV, the significant association between OSA and NC was no longer observed (OR, 1.39 [95% CI 0.94-2.05], p = 0.100).

Conclusions: OSA is associated with a 50% increase of odds of NC in children with SCD in this nationwide dataset. The use of NIMV to treat OSA may modify the risk of OSA-associated NC.
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http://dx.doi.org/10.1093/sleep/zsaa157DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7879414PMC
February 2021

Positive Airway Pressure for the Treatment of OSA in Infants.

Chest 2021 02 15;159(2):810-817. Epub 2020 Aug 15.

Division of Pulmonary & Sleep Medicine, Children's Hospital of Philadelphia, Philadelphia, PA; Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA.

Background: Positive airway pressure (PAP) is a standard therapy for the treatment of OSA in children, but objective data on the effectiveness of PAP in infants are sparse. The aim of this study was to compare the effectiveness of PAP in infants younger than 6 months of age with that in school-aged children.

Research Question: Compared with PAP in school-aged children, can PAP be titrated as successfully in infants, and is adherence to PAP similar in both age groups?

Study Design And Methods: Single-center retrospective study. For consecutive infants younger than 6 months of age and school-aged children 5 to 10 years of age with OSA treated with PAP, baseline and titration polysomnography data, PAP adherence data, and parent-reported barriers to adherence were compared between groups.

Results: Forty-one infants and 109 school-aged children were included. Median obstructive apnea hypopnea index (OAHI) in infants was 25.7/h (interquartile range [IQR], 17.8-35.9/h) and was greater than that in school-aged children (12.1/hr; IQR, 7.6-21.5/h; P < .0001). After PAP titration, OAHI was reduced by a median of 92.1% in infants, similar to the median 93.4% reduction in school-aged children (P = .67). PAP was used in infants on 94.7% of nights, which was more than the 83% in school-aged children (P = .003). No differences were found in barriers to adherence between infants and school-aged children, with behavioral barriers being most common in both groups.

Interpretation: Objective data demonstrate that PAP is both highly effective at treating OSA and well-tolerated in infants. Like older patients, PAP should be considered along with other therapies for the treatment of OSA in even the youngest children.
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http://dx.doi.org/10.1016/j.chest.2020.08.020DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7856529PMC
February 2021

Auto-titrating CPAP for the treatment of obstructive sleep apnea in children: a good beginning.

J Clin Sleep Med 2020 10;16(10):1825-1826

Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

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http://dx.doi.org/10.5664/jcsm.8726DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7954011PMC
October 2020

Pediatric pulmonology year in review 2019: Sleep medicine.

Pediatr Pulmonol 2020 08 11;55(8):1885-1891. Epub 2020 Jun 11.

Division of Pulmonary Medicine, Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

Pediatric Pulmonology publishes original research, review articles as well as case reports on a wide variety of pediatric respiratory disorders. In this article, we summarize the past year's publications in sleep medicine and we review selected literature from other journals within this field. Articles highlighted are topics on risk factors of sleep-disordered breathing, diagnosis, and treatment of obstructive sleep apnea as well as the utility of polysomnography in various complex conditions.
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http://dx.doi.org/10.1002/ppul.24865DOI Listing
August 2020

Pediatric Adenotonsillectomy Trial for Snoring (PATS): protocol for a randomised controlled trial to evaluate the effect of adenotonsillectomy in treating mild obstructive sleep-disordered breathing.

BMJ Open 2020 03 15;10(3):e033889. Epub 2020 Mar 15.

Division of Sleep Medicine and Circadian Disorders, Brigham and Women's Hospital and Harvard Medical School, Boston, Massachusetts, USA.

Introduction: Mild obstructive sleep-disordered breathing (oSDB), characterised by habitual snoring without frequent apnoeas and hypopnoeas on polysomnography, is prevalent in children and commonly treated with adenotonsillectomy (AT). However, the absence of high-level evidence addressing the role of AT in improving health and behavioural outcomes has contributed to significant geographical variations in care and potential for surgery to be both overused and underused.

Methods And Analysis: The Pediatric Adenotonsillectomy Trial for Snoring (PATS) is a single-blinded, multicentre randomised controlled trial designed to evaluate the effect of AT in treating mild oSDB. Four hundred sixty eligible children, aged 3.0-12.9 years old, will be randomised to either early adenotonsillectomy or to watchful waiting with supportive care (WWSC) with a 1:1 ratio. The study's coprimary endpoints are (1) change from baseline in executive behaviour relating to self-regulation and organisation skills as measured by the Behavioural Rating Inventory of Executive Function (BRIEF) Global Composite Score (GEC); and (2) change from baseline in vigilance as measured on the Go-No-Go (GNG) signal detection parameter (d-prime). A mixed effects model will be used to compare changes in the BRIEF GEC score and GNG score at 6 and 12 months from baseline between the AT arm and the WWSC arm.

Ethics And Dissemination: The study protocol was approved by the institutional review board (IRB) at Children's Hospital of Philadelphia (CHOP) on 3 October 2014 (14-0 11 214). The approval of CHOP as the central IRB of record was granted on 29 February 2016. The results will be published in peer-reviewed journals and presented at academic conferences. The data collected from the PATS study will be deposited in a repository (National Sleep Research Resource, sleepdata.org) after completion of the study to maximise use by the scientific community.

Trial Registration Number: NCT02562040; Pre-results.
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http://dx.doi.org/10.1136/bmjopen-2019-033889DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073822PMC
March 2020

Auto-titrating CPAP for the treatment of obstructive sleep apnea in children.

J Clin Sleep Med 2020 06;16(6):871-878

Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

Study Objectives: In-laboratory titration polysomnography (PSG) is standard to determine optimal therapeutic continuous positive airway pressure (CPAP) in children with obstructive sleep apnea (OSA). The use of auto-titrating CPAP devices (autoCPAP) as an alternative to CPAP titration has not been well studied in children. We hypothesized that autoCPAP-derived pressures (P, P, P) would be similar to titration PSG pressure (P).

Methods: This is a retrospective study of children with OSAS initiated on autoCPAP between 2007 and 2017, who used autoCPAP for at least 2 h/night and who had adequate titration PSG were included in the analysis. AutoCPAP-derived pressures were obtained from use downloads and compared with P. P predictive factors were analyzed by median regression. Nonparametric methods were used for analysis.

Results: Of 110 children initiated on autoCPAP, 44 satisfied the inclusion criteria. Age (median (interquartile range)) was 13.01 (9.98-16.72) years, and 63.6% were obese. P median (interquartile range) was 8 (7-11) cmH₂O, mean autoCPAP-derived pressure (P) was 6.2 (5.6-7.6) cmH₂O, peak mean pressure (P) was 9.4 (7.7-11.1) cmH₂O, and average device pressure ≤ 90% of the time (P) was 8.1 (7.2-9.7) cmH₂O. AutoCPAP-derived pressures correlated with P (P < .05). P was lower than the other 3 pressures (P < .0002). Median regression analysis demonstrated that after adjusting for patient characteristics such as age, sex, and obesity status, autoCPAP-derived pressures remained significant predictors of P (P < .05). There were no significant interactions between these patient characteristics and autoCPAP-derived pressures.

Conclusions: This study demonstrates that autoCPAP-derived pressures correlate with the titration PSG-derived pressures. These results indicate that autoCPAP can be used in the pediatric population and can determine pressures that are close to the titration pressures.
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http://dx.doi.org/10.5664/jcsm.8348DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7849672PMC
June 2020

Secondhand Smoke exposure and risk of Obstructive Sleep Apnea in Children.

Int J Pediatr Otorhinolaryngol 2020 Mar 29;130:109807. Epub 2019 Nov 29.

Department of Psychiatry, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.

Objectives: Obstructive sleep apnea (OSA) has a prevalence of 4% in children. Few studies have explored the role of secondhand smoke (SHS) on OSA severity and have shown contradicting results. Most studies have focused on the effect of SHS on snoring. This study explored the association of SHS exposure and OSA severity in children aged 3-18 years.

Methods: This is a retrospective single center IRB-approved study. Electronic Medical Records (EMR) were queried between 1/24/2015 and 1/24/2018 to obtain data on SHS exposure with standard questionnaires from perioperative database. SHS was analyzed as a binary variable and OSA was measured using obstructive apnea hypopnea index (OAHI) from polysomnography (PSG) as a continuous variable. Analyses were done on all children and in those with severe OSA (OAHI≥10/h) as a subgroup.

Results: EMR query yielded 101,884 children of whom 3776 had PSG. Limiting baseline PSG in 3-18-year-old and reliable information on SHS yielded 167 analyzable children of whom 70 had severe OSA. Children exposed to SHS had significantly more public insurance than non-exposed (p < 0.0001). Among children with severe OSA, median OAHI was significantly higher in SHS exposed compared to non-exposed (29.0vs.19.5,p = 0.04), but not across all children. In multivariable analysis SHS exposure increased OAHI by 48% in severe OSA subgroup (95%CI: 8%-102%; p = 0.01) when adjusted for race, body mass index, and adjusted household income.

Conclusion: Children aged 3-18 years with severe OSA who were exposed to SHS were found to have 1.48 increase in odds of OAHI than those without SHS exposure. Results could be limited by retrospective nature of study and EMR tools.
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http://dx.doi.org/10.1016/j.ijporl.2019.109807DOI Listing
March 2020

Diving Deeper: Rethinking AHI as the Primary Measure of OSA Severity.

J Clin Sleep Med 2019 08;15(8):1075-1076

Division of Pulmonary Medicine/Sleep Center, Children's Hospital of Philadelphia, Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania.

Citation: Cielo CM, Tapia IE. Diving deeper: rethinking AHI as the primary measure of OSA severity. J Clin Sleep Med. 2019;15(8):1075-1076.
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http://dx.doi.org/10.5664/jcsm.7856DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6707045PMC
August 2019

Pediatric pulmonology year in review 2018: Sleep medicine.

Pediatr Pulmonol 2019 10 13;54(10):1501-1507. Epub 2019 Jun 13.

Division of Pulmonary Medicine, Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

Pediatric Pulmonology publishes original research, case reports, and review articles on topics related to a wide range of children's respiratory disorders. In this article (Part 4 of a five-part series), we summarize the past year's publications in sleep medicine, in the context of selected literature in this area from other journals. Articles are highlighted on topics including infant sleep, diagnosis, and treatment of obstructive sleep apnea, and sleep disorders in chronic disease.
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http://dx.doi.org/10.1002/ppul.24415DOI Listing
October 2019

Adherence to Positive Airway Pressure for the Treatment of Obstructive Sleep Apnea in Children With Developmental Disabilities.

J Clin Sleep Med 2019 06 15;15(6):915-921. Epub 2019 Jun 15.

Sleep Center, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

Study Objectives: To determine whether adherence to positive airway pressure (PAP) differs in children with developmental disabilities (DD) compared to typically developing (TD) children.

Methods: PAP adherence of 240 children initiated on PAP for obstructive sleep apnea (OSA) was retrospectively analyzed. Adherence between groups, expressed as percentage of nights used and hours of usage on nights used at 3 and 6 months, was compared. Predictive factors of adherence were studied using a median regression model.

Results: A total of 103 children with DD (median [interquartile range] age = 7.9 [3.2-13.1] years) and 137 TD (11.0 [5.5-16.1], = .005) children were included. Percentage of nights used was significantly higher in children with DD at 3 (DD = 86.7 [33.9-97.9], TD = 62.9 [30.8-87.8] = .01) and 6 months (DD = 90.0 [53.3-100], TD = 70.7 [29.2-90.8], = .003). Hours of usage on nights used at 3 and 6 months were similar between groups (DD = 5.0 [1.4-7.9], TD = 4.6 [1.9-7.2], = .715; DD = 6.4 [1.8-8.3], TD = 5.7 [2.5-7.3], = .345, respectively). This adherence measure improved over time in both groups (DD, = .007; TD, = .005). At 6 months, higher median neighborhood income and titration at or before 6 months were significantly predictive for percentage of nights used; higher PAP pressure was significantly predictive for hours of usage in both groups.

Conclusions: Children with DD had better PAP adherence expressed as percentage of nights used than TD children. Hours of usage on nights used at 3 and 6 months were similar between groups and improved over time. Higher income and titration at or before 6 months were predictive of adherence in all children. These findings indicate that children with DD can successfully wear PAP.
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http://dx.doi.org/10.5664/jcsm.7850DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6557645PMC
June 2019

Upper airway visualization in pediatric obstructive sleep apnea.

Paediatr Respir Rev 2019 Nov 4;32:48-54. Epub 2019 Apr 4.

Sleep Center, Division of Pulmonary Medicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA. Electronic address:

Pediatric Obstructive Sleep Apnea (OSA) is a condition that may lead to a variety of comorbidities in adolescence and adulthood. The gold standard of diagnosing OSA is polysomnography (PSG). Over the past fifteen years numerous publications have explored how to better visualize the upper airway to further assess OSA in the pediatric population, and eventually institute personalized treatment. Lateral neck radiograph, cephalometry, computed axial tomography, and magnetic resonance imaging are all unique imaging techniques that are used in the diagnosis of OSA. Drug Induced Sleep Endoscopy is a direct visualization technique that is gathering momentum in pediatrics. Each approach has respective benefits and weaknesses. However, none of them at this time can replace PSG. They are a helpful supplement in those patients with complicated upper airway anatomy and in those with residual OSA.
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http://dx.doi.org/10.1016/j.prrv.2019.03.007DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6776720PMC
November 2019
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