Laryngoscopy Grade Improvement and Difficult Airway Resolution in Infants With Robin Sequence Undergoing Mandibular Distraction Osteogenesis: A Multi-Institutional Study
Restricted accessResearch articleFirst published online July, 2021
Laryngoscopy Grade Improvement and Difficult Airway Resolution in Infants With Robin Sequence Undergoing Mandibular Distraction Osteogenesis: A Multi-Institutional Study
Mandibular distraction osteogenesis (MDO) aims to relieve tongue-based airway obstruction in Robin Sequence (RS). We investigated direct laryngoscopy grade (DLG) improvement and difficult airway (DA) resolution following MDO.
Design:
Retrospective cohort analysis.
Setting:
Three tertiary care institutions.
Patients:
Sixty-four infants with RS who underwent a single MDO procedure in their first year of life were identified from January 2010 to January 2019.
Main Outcome Measures:
The primary outcome was DLG pre- and post-MDO. Secondary outcomes included DA designation, pre- and post-MDO polysomnographic assessment for obstructive sleep apnea (OSA), length of stay, need for gastrostomy, and major/minor adverse events.
Results:
Median DLG improved from II pre-MDO to I at the time of distractor removal (n = 43, P < .001). No significant change was seen in patients with a third recorded time point (eg, palatoplasty; n = 78, P = .52). Twenty-six (47%) of 55 patients were designated as DA pre-MDO, and 10 (18%) of 55 patients retained the label post-MDO (P < .01). Five (50%) of these 10 patients appeared to be inappropriately retained. Median obstructive apnea–hypopnea index improved from 38.6 (range 31.2-62.8) pre-MDO to 2.9 (range 1-3.9) post-MDO (n = 12; P = .002).
Conclusion:
Mandibular distraction osteogenesis allowed for DLG improvement that was stably maintained as well as functional improvement in OSA, with minimal morbidity. Difficult airway designation persisted in the electronic record of some infants despite clinical resolution.
AlmajedAViezel-MathieuAGilardinoMSFloresRLTholpadySSCôtA. Outcome following surgical interventions for micrognathia in infants with Pierre Robin sequence: a systematic review of the literature. Cleft Palate-Craniofac J. 2017;54(1):32.
2.
Al-SamkariHTKaneAAMolterDWVachharajaniA.Neonatal outcomes of Pierre Robin sequence: an institutional experience. Clin Pediatr (Phila). 2010;49(12):1117–1122.
3.
AlvesRSResendeMBSkomroRPSouzaFJReedUC. Sleep and neuromuscular disorders in children. Sleep Med Rev. 2009;13(2):133.
4.
ApfelbaumJLHagbergCACaplanRA, et al.Practice guidelines for management of the difficult airway: an updated report by the American society of anesthesiologists task force on management of the difficult airway. Anesthesiology. 2013;118(2):251.
5.
BangiyevJNTraboulsiHAbdulhamidIRozzelleAThottamPJ. Sleep architecture in Pierre-Robin sequence: the effect of mandibular distraction osteogenesis. Int J Pediatr Otorhinolaryngol. 2016;89:72.
6.
BekiszJMWangMMRickertSMRodriguezAJFloresRL. Sleep-disordered breathing and airway assessment using polysomnography in pediatric patients with craniofacial disorders. J Craniofac Surg. 2020;31(3):720–726.
7.
BreikOTiveyDUmapathysivamKAndersonP. Mandibular distraction osteogenesis for the management of upper airway obstruction in children with micrognathia: a systematic review. Int J Oral Maxillofac Surg. 2016;45(6):769.
8.
BreikOUmapathysivamKTiveyDAndersonP. Feeding and reflux in children after mandibular distraction osteogenesis for micrognathia: a systematic review. Int J Pediatr Otorhinolaryngol. 2016;85:128–135.
9.
BrookerGECooperMGBrookerGECooperMG. Airway management for infants with severe micrognathia having mandibular distraction osteogenesis. Anaesthesia & Intensive Care. 2010;38(1):43.
10.
CormackRSLehaneJ. Difficult tracheal intubation in obstetrics. Anaesthesia. 1984;39(11):1105.
11.
CostaMATuMMMurageKPTholpadySSEngleWAFloresRL. Robin sequence: mortality, causes of death, and clinical outcomes. Plast Reconstr Surg. 2014;134(4):738.
12.
CuSRSidmanJD. Rates and risks of gastrostomy tubes in infants with cleft palate. Arch Otolaryngol Head Neck Surg. 2011;137(3):275–281.
DalesioNMDiaz-RodriguezNKokaR, et al.Development of a multidisciplinary pediatric airway program: an institutional experience. Hosp Pediatr. 2019;9(6):468–475.
DeMauroSBPatelPRMedoff-CooperBPosenchegMAbbasiS. Postdischarge feeding patterns in early- and late-preterm infants. Clin Pediatr (Phila). 2011;50(10):957–962.
17.
DennyAAmmC. New technique for airway correction in neonates with severe Pierre Robin sequence. J Pediatr. 2005;147(1):97.
18.
DennyAD. Distraction osteogenesis in Pierre Robin neonates with airway obstruction. Clin Plast Surg. 2004;31(2):221.
19.
DiepGKEisemannBSFloresRL. Neonatal mandibular distraction osteogenesis in infants with Pierre Robin sequence. J Craniofac Surg. 2020; 31(4):1137–1141.
20.
EvansKNSieKCHopperRAGlassRPHingAVCunninghamML. Robin sequence: from diagnosis to development of an effective management plan. Pediatrics. 2011;127(5):936.
21.
FloresRLGreathouseSTCostaMTahiriYSoleimaniTTholpadySS. Defining failure and its predictors in mandibular distraction for robin sequence. J Craniomaxillofac Surg. 2015;43(8):1614.
22.
GoldsteinJAChungCPaligaJTCieloCMarcusCLLioyJBartlettSPTaylorJA. Mandibular distraction osteogenesis for the treatment of neonatal tongue-based airway obstruction. J Craniofac Surg. 2015;26(3):634–641.
23.
HeffernanCBCalabreseCEResnickCM. Does mandibular distraction change the laryngoscopy grade in infants with Robin sequence?J Oral Maxillofac Surg. 2019;77(2):371.
24.
HongPKearnsD. Airway characteristics of infants with Pierre Robin sequence who undergo mandibular distraction osteogenesis. Ear Nose Throat J. 2015;94(8):E25.
25.
HornsteinLBorchersD. Stroke in an infant prior to the development of manifestations of neurofibromatosis. Neurofibromatosis. 1989;2(2):116.
26.
JadcherlaSRWangMVijayapalASLeuthnerSR. Impact of prematurity and co-morbidities on feeding milestones in neonates: a retrospective study. J Perinatol. 2010;30(3):201–208.
27.
KarempelisPHagenMMorrellNRobyBB. Associated syndromes in patients with Pierre Robin sequence. Int J Pediatr Otorhinolaryngol. 2020;131:109842.
28.
KatzESMitchellRBD’AmbrosioCM. Obstructive sleep apnea in infants. Am J Respir Crit Care Med. 2012;185(8):805–816.
29.
KhansaIHallCMadhounLLSplaingardMBaylisAKirschnerREPearsonGD. Airway and feeding outcomes of mandibular distraction, tongue-lip adhesion, and conservative management in Pierre Robin sequence: a prospective study. Plast Reconstr Surg. 2017;139(4):975e.
30.
KohLKKongCEIp-YamPC. The modified Cormack-Lehane score for the grading of direct laryngoscopy: evaluation in the Asian population. Anaesth Intensive Care. 2002;30(1):48.
31.
LamDJTabanginMEShikaryTA, et al.Outcomes of mandibular distraction osteogenesis in the treatment of severe micrognathia. JAMA Otolaryngol Head Neck Surg. 2014;140(4):338.
32.
LessenBS. Effect of the premature infant oral motor intervention on feeding progression and length of stay in preterm infants. Adv Neonatal Care. 2011;11(2):129–139.
33.
LidskyMELanderTASidmanJD. Resolving feeding difficulties with early airway intervention in Pierre Robin sequence. Laryngoscope. 2008;118(1):120–123.
34.
MandellDLYellonRFBradleyJPIzadiKGordonCB. Mandibular distraction for micrognathia and severe upper airway obstruction. Arch Otolaryngol Head Neck. 2004;130(3):344.
35.
MarkLJHerzerKRCoverR, et al.Difficult airway response team: a novel quality improvement program for managing hospital-wide airway emergencies. Anesth Analg. 2015;121(1):127.
36.
MarquesILde SousaTVCarneiroAFPeresSPBarbieriMA, Bettiol H. [Robin sequence: a single treatment protocol]. J Pediatr. 2005;81(1):14.
37.
McGinleyBMSchwartzARSchneiderHKirknessJPSmithPLPatilaSP. Upper airway neuromuscular compensation during sleep is defective in obstructive sleep apnea. J Appl Physiol. 2008;105(1):197.
38.
MillerCK. Feeding issues and interventions in infants and children with clefts and craniofacial syndromes. Semin Speech Lang. 2011;32(2):115–126.
39.
MizunoKUedaA. The maturation and coordination of sucking, swallowing, and respiration in preterm infants. J Pediatr. 2003;142(1):36–40.
40.
MuddPAPerkinsJNHarwoodJEValdezSAllenGC. Early intervention: distraction osteogenesis of the mandible for severe airway obstruction. Otolaryngol Head Neck Surg. 2012;146(3):467–472.
41.
MurageKPCostaMAFrielMTHavlikRJTholpadySSFloresRL. Complications associated with neonatal mandibular distraction osteogenesis in the treatment of Robin sequence. J Craniofac Surg. 2014;25(2):383.
42.
MurageKPTholpadySSFrielMHavlikRJFloresRL. Outcomes analysis of mandibular distraction osteogenesis for the treatment of Pierre Robin sequence. Plast Reconstr Surg. 2013;132(2):419.
43.
PapoffPGuelfiGCicchettiR, et al.Outcomes after tongue-lip adhesion or mandibular distraction osteogenesis in infants with Pierre Robin sequence and severe airway obstruction. Int J Oral Maxillofac Surg. 2013;42(11):1418.
44.
PremaratneIDBrownstoneNLotfiPImahiyeroboTA. Incidence of gastroesophageal reflux disease in children with cleft lip and palate and an evaluation of its impact on weight gain. Ann Plast Surg. 2020;85(S1 Suppl 1):S141–S142.
45.
ScottARMaderNS. Regional variations in the presentation and surgical management of Pierre Robin sequence. Laryngoscope. 2014;124(12):2818.
46.
SetabutrDPerezMRTruongMTSendersCWRubinsteinBK. Neurofibromatosis of the larynx causing stridor and sleep apnea. Am J Otolaryngol. 2014;35(5):631.
47.
SusarlaSMEvansKNKapadiaHVasilakouNEgbertMAHopperRA. Distraction osteogenesis normalizes mandibular body-symphysis morphology in infants with Robin sequence. J Oral Maxillofac Surg (02782391). 2018;76(1):169.
48.
SusarlaSMMundingerGSChangCCSwansonEWLoughDRottgersSARedettRJKumarAR. Gastrostomy placement rates in infants with Pierre Robin sequence: a comparison of tongue-lip adhesion and mandibular distraction osteogenesis. Plast Reconstr Surg. 2017;139(1):149.
49.
VatlachSMaasCPoetsCF. Birth prevalence and initial treatment of Robin sequence in Germany: a prospective epidemiologic study. Orphanet J Rare Dis. 2014;9(1):1.
50.
VerlindenCRvan de VijfeijkenSETuinzingDBJansmaEPBeckingAGSwennenGR. Complications of mandibular distraction osteogenesis for developmental deformities: a systematic review of the literature. Int J Oral Maxillofac Surg. 2015;44(1):44.
51.
VolkASDavisMJNarawaneAM, et al.Quantification of mandibular morphology in Pierre Robin sequence to optimize mandibular distraction osteogenesis. Cleft Palate Craniofac J. 2020;57(8):1032–1040. doi:10.1177/1055665620913780
52.
WadieGMLobeTE. Gastroesophageal reflux disease in neurologically impaired children: the role of the gastrostomy tube. Semin Laparosc Surg. 2002;9(3):180–189.
53.
YentisSMLeeDJ. Evaluation of an improved scoring system for the grading of direct laryngoscopy. Anaesthesia. 1998;53(11):1041.
54.
ZellnerEGMhlabaJMReidRRSteinbacherDM. Does mandibular distraction vector influence airway volumes and outcome?J Oral Maxillofac Surg (02782391). 2017;75(1):167.
55.
ZhangRSHoppeICTaylorJABartlettSP. Surgical management and outcomes of Pierre Robin sequence: a comparison of mandibular distraction osteogenesis and tongue-lip adhesion. Plast Reconstr Surg. 2018;142(2):480.
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