Tongue-lip adhesion (TLA) is commonly used to relieve obstructive sleep apnea (OSA) in infants with Robin sequence (RS), but few studies have evaluated its efficacy with objective measures. The purpose of this study was to measure TLA outcomes using polysomnography. Our hypothesis was that TLA relieves OSA in most infants.
Methods
This is a retrospective study of infants with RS who underwent TLA from 2011 to 2014 and had at least a postoperative polysomnogram. Predictor variables included demographic and birth characteristics, surgeon, syndromic diagnosis, GILLS score, preoperative OSA severity, and clinical course. A successful outcome was defined as minimal OSA (apneahypopnea index score < 5) on postoperative polysomnogram and no need for additional airway intervention. Descriptive, bivariate, and regression statistics were computed, and statistical significance was set at P < .05.
Results
Eighteen subjects who had TLA at a mean age of 28 ± 4.7 days were included. Thirteen (72.2%) had a confirmed or suspected syndrome, and the mean GILLS score was 3 ± 0.3. All parameters trended toward improvement from the preoperative to postoperative polysomnograms, and improvement in OSA severity, oxygen saturation nadir, and arousals per hour was statistically significant (P < .02). This effect was significant across categories of surgeon, syndrome, and GILLS score. Nine subjects (50%) met the criteria for a successful outcome. Bivariate and regression analyses did not demonstrate a significant relationship between success and any predictor variable.
Conclusions
TLA improved airway obstruction in all infants with RS but resolved OSA in only nine patients, and success was unpredictable.
AbramowiczS., BacicJ.D., MullikenJ.B., RogersG.F.Validation of the GILLS score for tongue-lip adhesion in Robin sequence patients.J Craniofac Surg.2012; 23: 382–386.
2.
ArgamasoR.V.Glossopexy for upper airway obstruction in Robin sequence.Cleft Palate Craniofac J.1992; 29: 232–238.
3.
BerryR.B., BudhirajaR., GottliebD.J., GozalD., IberC., KapurV.K., MarcusC.L., MehraR., ParthasarathyS., QuanS.F.. Rules for scoring respiratory events in sleep: update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events. Deliberations of the Sleep Apnea Definitions Task Force of the American Academy of Sleep Medicine.J Clin Sleep Med.2012; 8: 597–619.
4.
Caouette-LabergeL., BayetB., LarocqueY.The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities.Plast Reconstr Surg.1994; 93: 934–942.
5.
CarrM.M., PojeC.P., KingstonL., KielmaD., HeardC.Complications in pediatric tracheostomies.Laryngoscope.2001; 111: 1925–1928.
6.
DemkeJ., BassimM., PatelM.R., DeanS., RahbarR., van AalstJ.A., DrakeA.Parental perceptions and morbidity: tracheostomy and Pierre Robin sequence.Int J Pediatr Otorhinolaryngol.2008; 72: 1509–1516.
7.
DennyA.D., AmmC.A., SchaeferR.B.Outcomes of tongue-lip adhesion for neonatal respiratory distress caused by Pierre Robin sequence.J Craniofac Surg.2004; 15: 819–823.
8.
DennyA.D., TalismanR., HansonP.R., RecinosR.F.Mandibular distraction osteogenesis in very young patients to correct airway obstruction.Plast Reconstr Surg.2001; 108: 302–311.
9.
EvansA.K., RahbarR., RogersG.F., MullikenJ.B., VolkM.S.Robin sequence: a retrospective review of 115 patients.Int J Pediatr Otorhinolaryngol.2006; 70: 973–980.
10.
HuangF., LoL.J., ChenY.R., YangJ.C., NiuC.K., ChungM.Y.Tonguelip adhesion in the management of Pierre Robin sequence with airway obstruction: technique and outcome.Chang Gung Med J.2005; 28: 90–96.
11.
HuntC.E., CorwinM.J., ListerG., Weese-MayerD.E., NeumanM.R., TinsleyL., BairdT.M., KeensT.G., CabralH.J.Longitudinal assessment of hemoglobin oxygen saturation in healthy infants during the first 6 months of age. Collaborative Home Infant Monitoring Evaluation (CHIME) Study Group.J Pediatr.1999; 135: 580–586.
12.
IsonoS., TanakaA., IshikawaT., NishinoT.Developmental changes in collapsibility of the passive pharynx during infancy.Am J Respir Crit Care Med.2000; 162: 832–836.
13.
KatoI., FrancoP., GroswasserJ., KelmansonI., TogariH., KahnA.Frequency of obstructive and mixed sleep apneas in 1,023 infants.Sleep.2000; 23: 487–492.
14.
KatzE.S., MitchellR.B., D'AmbrosioC.M.Obstructive sleep apnea in infants.Am J Respir Crit Care Med.2012; 185: 805–816.
15.
KirschnerR.E., LowD.W., RandallP., BartlettS.P., McDonald-McGinnD.M., SchultzP.J., ZackaiE.H., LaRossaD.Surgical airway management in Pierre Robin sequence: is there a role for tongue-lip adhesion?Cleft Palate Craniofac J.2003; 40: 13–18.
16.
KolstadC.K., SendersC.W., RubinsteinB.K., TollefsonT.T.Mandibular distraction osteogenesis: at what age to proceed.Int J Pediatr Otorhinolaryngol.2011; 75: 1380–1384.
17.
MagalangU.J., DmochowskiJ., VeeramachaneniS., DrawA., MadorM.J., El-SolhA., GrantB.J.Prediction of the apnea-hypopnea index from overnight pulse oximetry.Chest.2003; 124: 169–1701.
18.
MandellD.L., YellonR.F., BradleyJ.P., IzadiK., GordonC.B.Mandibular distraction for micrognathia and severe upper airway obstruction.Arch Otolaryngol Head Neck Surg.2004; 130: 344–348.
19.
MarquesI.L., de SousaT.V., CarneiroA.F., BarbieriM.A., BettiolH., GutierrezM.R.Clinical experience with infants with Robin sequence: a prospective study.Cleft Palate Craniofac J.2001; 38: 171–178.
MeyerA.C., LidskyM.E., SampsonD.E., LanderT.A., LiuM., SidmanJ.D.Airway interventions in children with Pierre Robin sequence.Otolaryngol Head Neck Surg.2008; 138: 782–787.
22.
Montgomery-DownsH.E., GozalD.Snore-associated sleep fragmentation in infancy: mental development effects and contribution of secondhand cigarette smoke exposure.Pediatrics.2006; 117: e496–e502.
23.
MurageK.P., TholpadyS.S., FrielM., HavlikR.J., FloresR.L.Outcomes analysis of mandibular distraction osteogenesis for the treatment of Pierre Robin sequence.Plast Reconstr Surg.2013; 132: 419–421.
24.
OwA.T., CheungL.K.Meta-analysis of mandibular distraction osteogenesis: clinical applications and functional outcomes.Plast Reconstr Surg.2008; 121: 54e–69e.
25.
RogersG.F., MurthyA.S., LaBrieR.A., MullikenJ.B.The GILLS score: part I. Patient selection for tongue-lip adhesion in Robin sequence.Plast Reconstr Surg.2011; 128: 243–251.
26.
SchaeferR.B., StadlerJ.A.III, GosainA.K.To distract or not to distract: an algorithm for airway management in isolated Pierre Robin sequence.Plast Reconstr Surg.2004; 113: 1113–1125.
27.
ScottA.R., TibesarR.J., LanderT.A., SampsonD.E., SidmanJ.D.Mandibular distraction osteogenesis in infants younger than 3 months.Arch Facial Plast Surg.2011; 13: 173–179.
28.
SedaghatA.R., AndersonI.C., McGinleyB.M., RossbergM.I., RedettR.J., IshmanS.L.Characterization of obstructive sleep apnea before and after tongue-lip adhesion in children with micrognathia.Cleft Palate Craniofac J.2012; 49: 21–26.
29.
SherA.E., ShprintzenR.J., ThorpyM.J.Endoscopic observations of obstructive sleep apnea in children with anomalous upper airways: predictive and therapeutic value.Int J Pediatr Otorhinolaryngol.1986; 11: 135–146.
30.
SpicuzzaL., LeonardiS., La RosaM.Pediatric sleep apnea: early onset of the ‘syndrome'?Sleep Med Rev.2009; 13: 111–122.
31.
SpringM.A., MountD.L.Pediatric feeding disorder and growth decline following mandibular distraction osteogenesis.Plast Reconstr Surg.2006; 118: 476–482.
32.
TahiriY., Viezel-MathieuA., AldekhayelS., LeeJ., GilardinoM.The effectiveness of mandibular distraction in improving airway obstruction in the pediatric population.Plast Reconstr Surg.2014; 133: 352e–359e.
33.
TroulisM.J., KabanL.B.Complications of mandibular distraction osteogenesis.Oral Maxillofac Surg Clin North Am.2003; 15: 251–264.
34.
WetmoreR.F., HandlerS.D., PotsicW.P.Pediatric tracheostomy. Experience during the past decade.Ann Otol Rhinol Laryngol.1982; 91: 628–632.
35.
WittenbornW., PanchalJ., MarshJ.L., SekarK.C., GurleyJ.Neonatal distraction surgery for micrognathia reduces obstructive apnea and the need for tracheotomy.J Craniofac Surg.2004; 15: 623–630.
