{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,24]],"date-time":"2026-09-24T13:09:35Z","timestamp":1790255375286,"version":"4.1.0"},"reference-count":32,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2020,12,17]],"date-time":"2020-12-17T00:00:00Z","timestamp":1608163200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2020,12,17]],"date-time":"2020-12-17T00:00:00Z","timestamp":1608163200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Pulmonology"],"published-print":{"date-parts":[[2021,2]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To characterize the clinical presentation of sleep\u2010disordered breathing and respiratory patterns at rest and during a 6\u2010min walk test (6MWT) in children with rapid\u2010onset obesity, hypothalamic dysfunction, hypoventilation, and autonomic dysregulation (ROHHAD) syndrome.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Retrospective study of children with ROHHAD who had a diagnostic baseline polysomnography, daytime cardiorespiratory monitoring at rest and a 6MWT. Polysomnography data were also compared with body mass index\u2010, age\u2010, and sex\u2010matched controls.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Of the eight children with ROHHAD, all eight (100%) had obstructive sleep apnea (OSA) and 2\/8 (25%) had nocturnal hypoventilation (NH) on their baseline polysomnography. Comparing the ROHHAD group to the control group, there were no significant differences in the median (interquartile range [IQR]) obstructive apnea\u2010hypopnea index (11.1 [4.3\u201358.4] vs. 14.4 [10.3\u201323.3] events\/h, respectively;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u2009.78). However, children with ROHHAD showed a significantly higher desaturation index compared to the control group (37.9 [13.7\u201359.8] vs. 14.7 [4.3\u201327.6] events\/h;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u2009.05). While awake at rest, some children with ROHHAD experienced significant desaturations associated with central pauses. During the 6MWT, no significant desaturations were observed, but two children showed moderate functional limitation.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Among children with ROHHAD, respiratory instability may be demonstrated by a significant number and severity of oxygen desaturations during sleep in the presence of OSA, with or without NH, and oxygen desaturations with central pauses at rest during wakefulness. Interestingly, during daily activities that require submaximal effort, children may not experience oxygen desaturations. Early recognition of respiratory abnormalities and targeted therapeutic interventions are important to limit associated morbidity and mortality in ROHHAD.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ppul.25199","type":"journal-article","created":{"date-parts":[[2020,12,3]],"date-time":"2020-12-03T10:27:04Z","timestamp":1606991224000},"page":"479-485","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Sleep\u2010disordered breathing, respiratory patterns during wakefulness and functional capacity in pediatric patients with rapid\u2010onset obesity with hypothalamic dysfunction, hypoventilation and autonomic dysregulation syndrome"],"prefix":"10.1002","volume":"56","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9924-3912","authenticated-orcid":false,"given":"Sarah","family":"Selvadurai","sequence":"first","affiliation":[{"name":"Translational Medicine, Research Institute, Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Benzon","sequence":"additional","affiliation":[{"name":"Translational Medicine, Research Institute, Hospital for Sick Children Toronto Ontario Canada"},{"name":"McMaster University Hamilton Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2197-0738","authenticated-orcid":false,"given":"Giorge","family":"Voutsas","sequence":"additional","affiliation":[{"name":"Translational Medicine, Research Institute, Hospital for Sick Children Toronto Ontario Canada"},{"name":"University of Toronto Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jill","family":"Hamilton","sequence":"additional","affiliation":[{"name":"University of Toronto Toronto Ontario Canada"},{"name":"Division of Endocrinology Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ann","family":"Yeh","sequence":"additional","affiliation":[{"name":"University of Toronto Toronto Ontario Canada"},{"name":"Division of Neurology Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Cifra","sequence":"additional","affiliation":[{"name":"University of Toronto Toronto Ontario Canada"},{"name":"Division of Cardiology Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Indra","family":"Narang","sequence":"additional","affiliation":[{"name":"Translational Medicine, Research Institute, Hospital for Sick Children Toronto Ontario Canada"},{"name":"University of Toronto Toronto Ontario Canada"},{"name":"Division of Respiratory Medicine Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,12,17]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2006-3324"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1155\/2018\/1250721"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1203\/PDR.0b013e318229474d"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1097\/MOP.0000000000000118"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2008-0238"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1965.02090030165011"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-016-0484-1"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.410100104"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1513\/pats.200708-135MG"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1093\/ptj\/80.8.782"},{"key":"e_1_2_7_12_1","unstructured":"Defining childhood obesity2016.https:\/\/www.cdc.gov\/obesity\/childhood\/defining.html"},{"key":"e_1_2_7_13_1","volume-title":"The AASM manual for the scoring of sleep and associated events: Rules, terminology and technical specifications, version 2.0.2 Darien","author":"Berry RB","year":"2013"},{"key":"e_1_2_7_14_1","doi-asserted-by":"crossref","unstructured":"Cardiorespiratory sleep studies in children.Establishment of normative data and polysomnographic predictors of morbidity. 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