Positioning as a conservative treatment option in infants with micrognathia and/or cleft

  • Karen A. Waters
    Sleep Medicine, The Children's Hospital at Westmead, Westmead, NSW, 2145, Australia.
    The Children's Hospital at Westmead, Sydney, 2145, NSW, Australia

    Department of Child and Adolescent Health, Faculty of Medicine, University of Sydney, NSW, 2050, Australia
    Search for articles by this author
Published:September 17, 2021DOI:https://doi.org/10.1016/j.siny.2021.101282


      • Polysomnography (PSG) provides detailed measures of sleep and breathing
      • PSG data indicate that not all infants have improved sleep and breathing when prone compared to supine
      • There is no current consensus on the parameters used to define acceptable limits for PSG parameters when infants are prone
      • Additional home monitoring is recommended when prone positioning is advised to manage an infant's airway obstruction


      Evaluation and management of airway obstruction in prone position were reviewed from studies in infants with micrognathia and/or cleft palate, using polysomnography (PSG) or similar measures, and comparing prone against other positions. Most studies identified were case series from specialist referral centres. Airway obstruction appears more severe on PSG than clinical assessment, but there is no consensus for PSG definitions of mild, moderate or severe airway obstruction. Infants show individual variability in responses to positioning; sleep quality tends to improve when prone, but 22–25% have better respiratory outcomes when supine. Most centres recommend home monitoring if advising that an infant be placed prone to manage their airway obstruction. In conclusion, in case series, success rates for managing infant airway obstruction by prone positioning vary from 12 to 76%. PSG studies comparing prone with other sleep positions can help differentiate which infants show improved airway obstruction and/or sleep quality when positioned prone.


      To read this article in full you will need to make a payment


      Subscribe to Seminars in Fetal and Neonatal Medicine
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect


        • Breugem C.C.
        • Evans K.N.
        • Poets C.F.
        • Suri S.
        • Picard A.
        • Filip C.
        • et al.
        Best practices for the diagnosis and evaluation of infants with Robin sequence: a clinical consensus report.
        JAMA Pediatr. 2016; 170: 894-902
        • Li W.Y.
        • Poon A.
        • Courtemanche D.
        • Verchere C.
        • Robertson S.
        • Bucevska M.
        • et al.
        Airway management in pierre Robin sequence: the vancouver classification.
        Plast Surg (Oakv). 2017; 25: 14-20
        • Caouette-Laberge L.
        • Bayet B.
        • Larocque Y.
        The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities.
        Plast Reconstr Surg. 1994; 93: 934-942
        • Daniel M.
        • Bailey S.
        • Walker K.
        • Hensley R.
        • Kol-Castro C.
        • Badawi N.
        • et al.
        Airway, feeding and growth in infants with Robin sequence and sleep apnoea.
        Int J Pediatr Otorhinolaryngol. 2013; 77: 499-503
        • Stubenitsky B.M.
        • Taylor H.O.
        • Peters D.
        • Kelly C.
        • Harkness M.
        • Forrest C.
        Predictive value of weight gain and airway obstruction in isolated Robin sequence.
        Cleft Palate Craniofac J. 2010; 47: 378-381
        • Mackay D.R.
        Controversies in the diagnosis and management of the Robin sequence.
        J Craniofac Surg. 2011; 22: 415-420
        • Cole A.
        • Lynch P.
        • Slator R.
        A new grading of Pierre Robin sequence.
        Cleft Palate Craniofac J. 2008; 45: 603-606
        • Manica D.
        • Schweiger C.
        • Sekine L.
        • Fagondes S.C.
        • Kuhl G.
        • Collares M.V.
        • et al.
        Association of polysomnographic parameters with clinical symptoms severity grading in Robin sequence patients: a cohort nested cross-sectional study.
        Sleep Med. 2018; 43: 96-99
        • Runyan C.M.
        • Uribe-Rivera A.
        • Tork S.
        • Shikary T.A.
        • Ehsan Z.
        • Weaver K.N.
        • et al.
        Management of airway obstruction in infants with pierre Robin sequence.
        Plast Reconstr Surg Glob Open. 2018; 6: e1688
        • Cote A.
        • Fanous A.
        • Almajed A.
        • Lacroix Y.
        Pierre Robin sequence: review of diagnostic and treatment challenges.
        Int J Pediatr Otorhinolaryngol. 2015; 79: 451-464
        • Giudice A.
        • Barone S.
        • Belhous K.
        • Morice A.
        • Soupre V.
        • Bennardo F.
        • et al.
        Pierre Robin sequence: a comprehensive narrative review of the literature over time.
        J Stomatol Oral Maxillofac Surg. 2018; 119: 419-428
        • Karempelis P.
        • Hagen M.
        • Morrell N.
        • Roby B.B.
        Associated syndromes in patients with pierre Robin sequence.
        Int J Pediatr Otorhinolaryngol. 2020; 131: 109842
        • Evans A.K.
        • Rahbar R.
        • Rogers G.F.
        • Mulliken J.B.
        • Volk M.S.
        Robin sequence: a retrospective review of 115 patients.
        Int J Pediatr Otorhinolaryngol. 2006; 70: 973-980
        • Anderson I.C.
        • Sedaghat A.R.
        • McGinley B.M.
        • Redett R.J.
        • Boss E.F.
        • Ishman S.L.
        Prevalence and severity of obstructive sleep apnea and snoring in infants with Pierre Robin sequence.
        Cleft Palate Craniofac J. 2011; 48: 614-618
        • van Lieshout M.J.
        • Joosten K.F.
        • Hoeve H.L.
        • Mathijssen I.M.
        • Koudstaal M.J.
        • Wolvius E.B.
        Unravelling Robin sequence: considerations of diagnosis and treatment.
        Laryngoscope. 2014; 124: E203-E209
        • Goudy S.
        • Jiramongkolchai P.
        • Chinnadurai S.
        Logistic regression analysis of Pierre Robin sequence patients requiring surgical intervention.
        Laryngoscope. 2017; 127: 945-949
        • Kimple A.J.
        • Baldassari C.M.
        • Cohen A.P.
        • Landry A.
        • Ishman S.L.
        Polysomnographic results of prone versus supine positioning in micrognathia.
        Int J Pediatr Otorhinolaryngol. 2014; 78: 2056-2059
        • Coutier L.
        • Guyon A.
        • Reix P.
        • Franco P.
        Impact of prone positioning in infants with Pierre Robin sequence: a polysomnography study.
        Sleep Med. 2019; 54: 257-261
        • MacLean J.E.
        • Fitzsimons D.
        • Fitzgerald D.A.
        • Waters K.A.
        The spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palate.
        Arch Dis Child. 2012; 97: 1058-1063
        • Hong H.
        • Wee C.P.
        • Haynes K.
        • Urata M.
        • Hammoudeh J.
        • Ward S.L.D.
        Evaluation of obstructive sleep apnea in prone versus nonprone body positioning with polysomnography in infants with Robin sequence.
        Cleft Palate Craniofac J. 2020; 57: 141-147
        • Kukkola H.-L.
        • Vuola P.
        • Seppä-Moilanen M.
        • Salminen P.
        • Kirjavainen T.
        Pierre Robin sequence causes position-dependent obstructive sleep apnea in infants.
        Arch Dis Child. 2021; (in press)
        • Carpenter R.G.
        • Irgens L.M.
        • Blair P.S.
        • England P.D.
        • Fleming P.
        • Huber J.
        • et al.
        Sudden unexplained infant death in 20 regions in Europe: case control study.
        Lancet. 2004; 363: 185-191
        • van Lieshout M.J.
        • Joosten K.F.
        • Mathijssen I.M.
        • Koudstaal M.J.
        • Hoeve H.L.
        • van der Schroeff M.P.
        • et al.
        Robin sequence: a European survey on current practice patterns.
        J Cranio-Maxillo-Fac Surg. 2015; 43: 1626-1631
        • Costa M.A.
        • Tu M.M.
        • Murage K.P.
        • Tholpady S.S.
        • Engle W.A.
        • Flores R.L.
        Robin sequence: mortality, causes of death, and clinical outcomes.
        Plast Reconstr Surg. 2014; 134: 738-745
        • Logjes R.J.H.
        • Haasnoot M.
        • Lemmers P.M.A.
        • Nicolaije M.F.A.
        • van den Boogaard M.H.
        • Mink van der Molen A.B.
        • et al.
        Mortality in Robin sequence: identification of risk factors.
        Eur J Pediatr. 2018; 177: 781-789
        • Rathe M.
        • Rayyan M.
        • Schoenaers J.
        • Dormaar J.T.
        • Breuls M.
        • Verdonck A.
        • et al.
        Pierre Robin sequence: management of respiratory and feeding complications during the first year of life in a tertiary referral centre.
        Int J Pediatr Otorhinolaryngol. 2015; 79: 1206-1212
        • Freed G.E.
        • Martinez F.
        The history of home cardiorespiratory monitoring.
        Pediatr Ann. 2017; 46: e303-e308
        • Committee on F, Newborn
        American Academy of P. Apnea, sudden infant death syndrome, and home monitoring.
        Pediatrics. 2003; 111: 914-917
        • Fish L.A.
        • Jones E.J.H.
        A survey on the attitudes of parents with young children on in-home monitoring technologies and study designs for infant research.
        PLoS One. 2021; 16e0245793
        • ZhuParris A.
        • Kruizinga M.D.
        • van Gent M.
        • Dessing E.
        • Exadaktylos V.
        • Doll R.J.
        • et al.
        Development and technical validation of a smartphone-based cry detection algorithm.
        Front Pediatr. 2021; 9: 651356
        • Dangerfield M.I.
        • Ward K.
        • Davidson L.
        • Adamian M.
        Initial experience and usage patterns with the owlet smart sock monitor in 47,495 newborns.
        Glob Pediatr Health. 2017; 4 (2333794X17742751)
      1. Owlet. 2021
        • Anjewierden S.
        • Humpherys J.
        • LaPage M.J.
        • Asaki S.Y.
        • Aziz P.F.
        Detection of tachyarrhythmias in a large cohort of infants using direct-to-consumer heart rate monitoring.
        J Pediatr. 2021; 232: 147-153 e1