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• W2922289051 abstract "Free AccessInfants - Polysomnography - CommentaryUnderstanding the Spectrum of Treatment Options for Infants With Pierre Robin Sequence and Airway Obstruction Joanna E. MacLean, MD, PhD, FRCPC Joanna E. MacLean, MD, PhD, FRCPC Address correspondence to: Joanna E. MacLean, BSc(Hon), MD, PhD, FRCPC, Division of Respiratory Medicine, Department of Pediatrics, University of Alberta, 4-590 Edmonton Clinic Health Academy (ECHA), 11405 87 Avenue, Edmonton AB T6G 1C9(780) 248-5584(888) 353-1323 E-mail Address: [email protected] Department of Pediatrics and Women and Children's Health Research Institute, Faculty of Medicine and Dentistry, University of Alberta, Alberta, Canada; Stollery Children's Hospital, Edmonton, Alberta, Canada Search for more papers by this author Published Online:March 15, 2019https://doi.org/10.5664/jcsm.7654Cited by:3SectionsPDF ShareShare onFacebookTwitterLinkedInRedditEmail ToolsAdd to favoritesDownload CitationsTrack Citations AboutINTRODUCTIONPierre Robin sequence (PRS), also referred to as Robin sequence, is defined by micrognathia, glossoptosis, and upper airway obstruction and results in varying degrees of airway compromise after birth.1,2 Both evaluation and treatment of airway obstruction in infants with PRS varies between centers with considerable controversy around the role of polysomnography for the diagnosis of sleep-related breathing disorders and assessment of treatment response.3,4 Disagreements about the role of polysomnography are undoubtedly influenced by access to polysomnographic testing as this is limited or unavailable in many areas.5,6 On the treatment side, there are differences between centers with respect to available treatment options and how treatment success is evaluated. While there has been considerably more study of the surgical options compared to nonsurgical options, the field overall is hampered by a relative paucity of high quality evidence.7There is a relatively long list of options for management of airway obstruction in infants with PRS. Nonsurgical options include prone sleep position,8 supplemental oxygen, use of an oral appliance with a velar extension,9 oropharyngeal or nasopharyngeal tube, noninvasive ventilation (typically continuous positive airway pressure), and placement of an endotracheal tube.3 Surgical options include tongue-lip adhesion (or glossopexy), mandibular distraction osteogenesis, subperiosteal release of the floor of the mouth, and tracheostomy—with additional surgical options used more selectively.3 There are multiple algorithms from different centers to support treatment decisions but none incorporate all treatment options and all use different strategies for decision making.3,10,11 With few studies comparing treatment modalities and even fewer randomized trials, there is little information on the characteristics of infants that will best respond to any given therapy.7,11 The study by Ehsan and colleagues in this issue of the Journal of Clinical Sleep Medicine12 adds another option, watchful waiting, to this list.The study reports on a cohort on infants with PRS who were treated with conservative (ie, nonsurgical) management.12 The study design was a 14-year retrospective cohort study that included infants with PRS who underwent polysomnography under 3 months of age, had a repeat polysomnography before 12 months of age, and who were managed conservatively at a single center. The study highlights important challenges to advancing our understanding of treatment options for airway obstruction in infants with PRS. The study was conducted in a center where a multi-disciplinary team that included pediatric pulmonology and sleep medicine experts assessed all infants with PRS, and all infants underwent overnight polysomnography, bedside nasopharyngoscopy, and computed tomography of the maxillofacial skeleton. From their retrospective, 14-year review, the authors excluded 33% of infants from the study group because polysomnography was not completed in infancy. The majority of infants with PRS were managed surgically with the minority (36%) receiving conservative management; this reflects a growing trend for surgical management despite reports that prone sleep position resolves airway obstruction in 40% to 70% of infants with PRS.3 Conservative management for this cohort included supplemental oxygen and watchful waiting. Follow-up showed that by 1 month of age, the majority of infants achieved full oral feeds and both obstructive and total respiratory events decreased on follow-up polysomnography. They observed changes in sleep parameters that reflected expected changes in healthy infants. This shows that even in the absence of surgical intervention to augment the airway, both feeding and airway obstruction improve. Some of the improvements related to surgical interventions may be attributable to normal developmental improvements in sleep and breathing during the time between a baseline polysomnography and a post-surgical follow-up study.The study highlights the importance of considering multiple outcomes, including feeding and growth, in addition to the results of polysomnography. Improvement in growth, leading to growth of the airway, is likely an important component of improving airway obstruction in infants with PRS and explains why more infants than older children will see improvement in their obstructive sleep apnea to the point that treatment can be stopped.13 Airway obstruction compromises breathing and oral feeding, so progression to oral feeding is another marker of improvement in airway obstruction. Feeding difficulties result in longer length of stay, so early assessment of feeding is important.14 Assessment of feeding, along with polysomnography, is an important part of a complete initial airway assessment of infants with PRS.3,15,16How to choose the best intervention for an individual infant with PRS is an unanswered question. While randomized controlled trials would provide the highest level of evidence, their application in this setting is challenging given, for example, differences in the expertise with each interventions between centers that might impact treatment outcomes. Multi-center registries of infants with PRS would provide important information if initial and follow-up assessments could be standardized and include objective measures where possible. Standardizing assessment tools, again with a focus on objective measures where possible, as well as including comparison groups would strengthen the quality of evidence derived from single center observational studies and facilitate combining data across studies. Understanding the outcomes that are important to children with PRS and their parents and caregivers will ensure that study results are relevant to clinical care. With considerable heterogeneity between infants with PRS, information from a large number of infants will be necessary to identify consistent characteristics that predict treatment response and favorable health outcomes.DISCLOSURE STATEMENTDr. MacLean reports no conflicts of interest.CITATIONMacLean JE. Understanding the spectrum of treatment options for infants with Pierre Robin sequence and airway obstruction. J Clin Sleep Med. 2019;15(3):373–374.REFERENCES1 van Nunen DPF, van den Boogaard MH, Breugem CCRobin Sequence: continuing heterogeneity in nomenclature and diagnosisJ Craniofac Surg2018294985987 CrossrefGoogle Scholar2 MacLean JE, Fitzsimons D, Fitzgerald DA, Waters KAThe spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palateArch Dis Child2012971210581063 CrossrefGoogle Scholar3 Resnick CM, LeVine J, Calabrese CE, Padwa BL, Hansen A, Katwa UEarly management of infants with Robin Sequence: an international survey and algorithmJ Oral Maxillofac Surg2019771136156 CrossrefGoogle Scholar4 MacLean JESleep frequently asked questions: question 1: what abnormalities do babies with cleft lip and/or palate have on polysomnography?Paediatr Respir Rev2018274447 CrossrefGoogle Scholar5 Kaditis AG, Alonso Alvarez ML, Boudewyns A, et al.Obstructive sleep disordered breathing in 2- to 18-year-old children: diagnosis and managementEur Respir J20164716994 CrossrefGoogle Scholar6 Katz SL, Witmans M, Barrowman N, et al.Paediatric sleep resources in Canada: the scope of the problemPaediatr Child Health2014197367372 CrossrefGoogle Scholar7 Bekisz JM, Fryml E, Flores RLA review of randomized controlled trials in cleft and craniofacial surgeryJ Craniofac Surg2018292293301 CrossrefGoogle Scholar8 Coutier L, Guyon A, Reix P, Franco PImpact of prone positioning in infants with Pierre Robin sequence: a polysomnography studySleep Med201954257261 CrossrefGoogle Scholar9 Poets CF, Maas C, Buchenau W, et al.Multicenter study on the effectiveness of the pre-epiglottic baton plate for airway obstruction and feeding problems in Robin sequenceOrphanet J Rare Dis201712146 CrossrefGoogle Scholar10 Hicks KE, Billings KR, Purnell CA, et al.An algorithm for airway management in patients with Pierre Robin sequenceJ Craniofac Surg201829511871192 CrossrefGoogle Scholar11 Gómez OJ, Barón OI, Peñarredonda MLPierre Robin sequence: an evidence-based treatment proposalJ Craniofac Surg2018292332338 CrossrefGoogle Scholar12 Ehsan Z, Kurian C, Weaver KN, et al.Longitudinal sleep outcomes in neonates with Pierre Robin sequence treated conservativelyJ Clin Sleep Med2019153477482 LinkGoogle Scholar13 Bedi PK, Castro-Codesal M, DeHaan K, MacLean JEUse and outcomes of long-term noninvasive ventilation for infantsCanadian Journal of Respiratory, Critical Care, and Sleep Medicine201824205212 CrossrefGoogle Scholar14 Dorise B, Trivedi A, Galea C, Walker K, Mehta BFeeding practices and growth of infants with Pierre Robin sequenceInt J Pediatr Otorhinolaryngol20191181114 CrossrefGoogle Scholar15 Reddy VSEvaluation of upper airway obstruction in infants with Pierre Robin sequence and the role of polysomnography--review of current evidencePaediatr Respir Rev2016178087 Google Scholar16 Rathé M, Rayyan M, Schoenaers J, et al.Pierre Robin sequence: management of respiratory and feeding complications during the first year of life in a tertiary referral centreInt J Pediatr Otorhinolaryngol201579812061212 CrossrefGoogle Scholar Previous article Next article FiguresReferencesRelatedDetailsCited by Yang R, Swanson J and Cielo C Craniofacial Syndromes Pediatric Sleep Medicine, 10.1007/978-3-030-65574-7_54, (655-665), . Paradigm Shift in Airway Management for Pierre Robin Sequence: Tongue Base SuspensionSafri S, Wickemeyer J and Valika T Journal of Craniofacial Surgery, 10.1097/SCS.0000000000007198, Vol. 32, No. 3, (e288-e290), Online publication date: 1-May-2021. The Comparison of Pierre Robin Sequence and Non-Syndromic Cleft PalateKocaaslan F, Sendur S, Koçak I and Çelebiler Ö Journal of Craniofacial Surgery, 10.1097/SCS.0000000000005961, Vol. 31, No. 1, (226-229), . Volume 15 • Issue 03 • March 15, 2019ISSN (print): 1550-9389ISSN (online): 1550-9397Frequency: Monthly Metrics History Submitted for publicationFebruary 26, 2019Submitted in final revised formFebruary 26, 2019Accepted for publicationFebruary 27, 2019Published onlineMarch 15, 2019 Information© 2019 American Academy of Sleep MedicinePDF download" @default.
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