For Publishers
DiscoveryMetadataPeer reviewHostingPublishing
For Researchers
JoinPublishReviewCollect
Register
Blog
About
Search
Advanced search
My ScienceOpen
Search
For Publishers
For Researchers
Blog
About
20
views
28
references
 Top references
cited by
30
    
0 reviews
 Review
0
comments
 Comment
0
recommends
+1 Recommend
0 collections
    0
    shares
      375
      similar
       All similar
      • Record: found
      • Abstract: found
      • Article: found
      Is Open Access

      Clinically Important Age-Related Differences in Sleep Related Disordered Breathing in Infants and Children with Prader-Willi Syndrome

      research-article
      Author(s): 1 , 2 , * , 1 , 2 , 2 , 3
      Editor(s):
      Publication date (Electronic):
      Journal: PLoS ONE
      Publisher: Public Library of Science

      Read this article at

      ScienceOpenPublisherPMC
      Bookmark
          There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

          Abstract

          Background

          Sleep related disordered breathing (SDB) in pediatric Prader-Willi Syndrome is gaining increased attention due to the possible association of growth hormone therapy, SDB and sudden death. However data on the patterns of SDB and their management, particularly in infants in this population, is lacking.

          Objective

          The aim of this study was to 1) describe patterns of SDB in growth hormone naive infants with PWS and the management of these disorders in our institution 2) Compare the patterns of sleep disorders between infants and children with PWS.

          Methods and Design

          Polysomnograms of infants and children (0–18 years of age) with Prader-Willi Syndrome were reviewed. Age, sex, anthropometrics, sleep architecture, obstructive and central apnea indices and oxygen saturations were recorded. Data of infants with central sleep apnea treated with oxygen were analyzed to evaluate the efficacy of this treatment. The main outcome measures were obstructive and central apnea indices on a polysomnogram.

          Results

          Data of 44 patients, 23 under 2 years of age and 21 older children were included. Infants when compared with older children were more likely to experience central sleep apnea (43% vs. 5%; p = 0.003). In older children obstructive was significantly more prevalent than central sleep apnea. Supplemental oxygen was used to treat 9/23 infants with central sleep apnea. Oxygen therapy resulted in a significant decrease in the median central apnea index from 14 (5,68) to 1 (0,6; p = 0.008) events/hour and an improvement in the oxygen saturation nadir from 70% (52, 92) to 81% (64, 95; p = 0.080).

          Conclusions

          Central sleep apnea with associated oxygen desaturations is more prevalent in infants compared with older children with Prader-Willi Syndrome. Supplemental oxygen was efficacious in treating central sleep apnea in infants. Routine sleep surveillance for all children with Prader-Willi Syndrome and treatment with oxygen for central sleep apnea should be considered.

          Related collections

          Most cited references28

          • Record: found
          • Abstract: found
          • Article: not found

          Diagnosis and management of childhood obstructive sleep apnea syndrome.

          Carole L Marcus, Lee Jay Brooks, Kari Draper … (2012)
          This technical report describes the procedures involved in developing recommendations on the management of childhood obstructive sleep apnea syndrome (OSAS). The literature from 1999 through 2011 was evaluated. A total of 3166 titles were reviewed, of which 350 provided relevant data. Most articles were level II through IV. The prevalence of OSAS ranged from 0% to 5.7%, with obesity being an independent risk factor. OSAS was associated with cardiovascular, growth, and neurobehavioral abnormalities and possibly inflammation. Most diagnostic screening tests had low sensitivity and specificity. Treatment of OSAS resulted in improvements in behavior and attention and likely improvement in cognitive abilities. Primary treatment is adenotonsillectomy (AT). Data were insufficient to recommend specific surgical techniques; however, children undergoing partial tonsillectomy should be monitored for possible recurrence of OSAS. Although OSAS improved postoperatively, the proportion of patients who had residual OSAS ranged from 13% to 29% in low-risk populations to 73% when obese children were included and stricter polysomnographic criteria were used. Nevertheless, OSAS may improve after AT even in obese children, thus supporting surgery as a reasonable initial treatment. A significant number of obese patients required intubation or continuous positive airway pressure (CPAP) postoperatively, which reinforces the need for inpatient observation. CPAP was effective in the treatment of OSAS, but adherence is a major barrier. For this reason, CPAP is not recommended as first-line therapy for OSAS when AT is an option. Intranasal steroids may ameliorate mild OSAS, but follow-up is needed. Data were insufficient to recommend rapid maxillary expansion.
          Article 0 comments Cited 284 times – based on 0 reviews     Review now
            Bookmark
            • Record: found
            • Abstract: found
            • Article: not found

            Sleep-disordered breathing and school performance in children.

            D Gozal (1998)
            To assess the impact of sleep-associated gas exchange abnormalities (SAGEA) on school academic performance in children. Prospective study. Urban public elementary schools. Two hundred ninety-seven first-grade children whose school performance was in the lowest 10th percentile of their class ranking. Children were screened for obstructive sleep apnea syndrome at home using a detailed parental questionnaire and a single night recording of pulse oximetry and transcutaneous partial pressure of carbon dioxide. If SAGEA was diagnosed, parents were encouraged to seek medical intervention for SAGEA. School grades of all participating children for the school year preceding and after the overnight study were obtained. SAGEA was identified in 54 children (18.1%). Of these, 24 underwent surgical tonsillectomy and adenoidectomy (TR), whereas in the remaining 30 children, parents elected not to seek any therapeutic intervention (NT). Overall mean grades during the second grade increased from 2.43 +/- 0.17 (SEM) to 2.87 +/- 0.19 in TR, although no significant changes occurred in NT (2.44 +/- 0.13 to 2.46 +/- 0.15). Similarly, no academic improvements occurred in children without SAGEA. SAGEA is frequently present in poorly performing first-grade students in whom it adversely affects learning performance. The data suggest that a subset of children with behavioral and learning disabilities could have SAGEA and may benefit from prospective medical evaluation and treatment.
            Article 0 comments Cited 140 times – based on 0 reviews     Review now
              Bookmark
              • Record: found
              • Abstract: found
              • Article: not found

              Normal polysomnographic values for children and adolescents.

              D J Basinki, S. Ward, K Omlin … (1992)
              Although polysomnography is routinely performed to evaluate children and adolescents with sleep-disordered breathing, normal polysomnographic values for the pediatric age group have not yet been established. We therefore performed overnight polysomnography in 50 normal children and adolescents (mean age 9.7 +/- 4.6 SD yr, range 1.1 to 17.4 yr). Of the children 56% were male. Chest wall motion, ECG, oronasal airflow, end-tidal PCO2 (PETCO2), arterial oxygen saturation (SaO2), and electrooculogram were monitored. Children had 0.1 +/- 0.5 (range 0 to 3.1) obstructive apneas per hour of total sleep time, with only 18% of children having any obstructive apneas. No child had obstructive apneas > 10 s in duration. Of the children 30% had central apneas > or = 10 s in duration, and one child had a central apnea associated with SaO2 45 mm Hg) occurred for 7 +/- 19% total sleep time (range 0 to 91%). The SaO2 nadir was 96 +/- 2% (range 89 to 98%), with only one child desaturating below 90% in association with a central apnea. We conclude that polysomnographic results in the pediatric age group differ from those in adults. Recommendations for normal polysomnographic criteria are given.
              Article 0 comments Cited 65 times – based on 0 reviews     Review now
                Bookmark
                All references

                Author and article information

                Contributors
                Mathias Baumert: Role: Editor
                Journal
                Journal ID (nlm-ta): PLoS One
                Journal ID (iso-abbrev): PLoS ONE
                Journal ID (publisher-id): plos
                Journal ID (pmc): plosone
                Title: PLoS ONE
                Publisher: Public Library of Science (San Francisco, USA )
                ISSN (Electronic): 1932-6203
                Publication date Collection: 2014
                Publication date (Electronic): 30 June 2014
                Volume: 9
                Issue: 6
                Electronic Location Identifier: e101012
                Affiliations
                [1 ]Division of Endocrinology, The Hospital for Sick Children, Toronto, Ontario, Canada
                [2 ]The University of Toronto, Toronto, Ontario, Canada
                [3 ]Division of Respiratory Medicine, The Hospital for Sick Children, Toronto, Ontario, Canada
                University of Adelaide, Australia
                Author notes

                Competing Interests: The authors have declared that no competing interests exist.

                Conceived and designed the experiments: MC IN. Wrote the paper: MC. Collected, analyzed, and interpreted data: MC. Contributed to study design: JH. Assisted with interpretation of data: JH. Edited the manuscript: JH. Involved in data analysis and interpretation: IN. Revised and edited the manuscript: IN.

                Article
                Publisher ID: PONE-D-13-50273
                DOI: 10.1371/journal.pone.0101012
                PMC ID: 4076199
                PubMed ID: 24979549
                SO-VID: e2d44bac-a927-47d9-9a27-cbf5b0510fe8
                Copyright statement: Copyright @ 2014
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                Date received : 30 November 2013
                Date accepted : 2 June 2014
                Page count
                Pages: 6
                Funding
                These authors have no support or funding to report.
                Categories
                Subject: Research Article
                Subject: Biology and Life Sciences
                Subject: Physiology
                Subject: Physiological Processes
                Subject: Sleep
                Subject: Medicine and Health Sciences
                Subject: Endocrinology
                Subject: Pediatric Endocrinology
                Subject: Epidemiology
                Subject: Pediatric Epidemiology
                Subject: Neurology
                Subject: Sleep Disorders
                Subject: Pediatrics
                Subject: Pulmonology
                Subject: Research and Analysis Methods
                Subject: Research Design
                Subject: Clinical Research Design

                ScienceOpen disciplines: Uncategorized
                Data availability:
                ScienceOpen disciplines: Uncategorized

                Comments

                Comment on this article

                scite_

                Similar content375

                See all similar

                Cited by29

                See all cited by

                Most referenced authors350

                See all reference authors