Airway Morphological Changes in Pierre Robin Sequence: A Retrospective Study

Abstract

Objective:

To investigate airway morphology changes in patients with Pierre Robin sequence (PRS) pre–/post–mandibular distraction osteogenesis (MDO) and to compare morphologic changes to age-matched controls.

Design:

Retrospective case–control study.

Setting:

Urban, academic, tertiary medical center.

Patients, Participants:

Fifteen patients with PRS after MDO to relieve upper airway obstruction (UAO) (2008-2018); age-matched controls for post-MDO patients.

Interventions:

Mandibular distraction osteogenesis, curvilinear internal mandibular distractors.

Main Outcome Measures:

(1) Physiologic improvement after MDO (apnea–hypopnea index; minimum oxygen saturation); (2) airway size (volume, surface area, length, mean/minimum cross-sectional area), shape (lateral:anterior–posterior ratio, cross-sectional area ratios, uniformity, sphericity), and changes with MDO; and (3) post-MDO airway size, shape versus age-matched controls.

Results:

Airway size increased after MDO (volume, P = .01; surface area, P = .02; length, P = .01), as did cross-sectional area (mean, P = .02; minimum, P = .02; minimum retropalatal, P = .05, mid-retroglossal, P = .02). Post-MDO PRS airways were larger than controls (volume, P < .01; surface area, P < .01; length, P < .01, cross-sectional area, P = .03). Airway shape remained nonuniform and flat post-MDO; control airways were round. Two syndromic patients required repeat MDO and had subphysiologic post-MDO airway cross-sectional area. Post-MDO PRS patients with supraphysiologic cross-sectional area along the entire airway had no UAO recurrence.

Conclusions:

In this small, heterogenous patient sample, MDO increases airway size, may preferentially affect the retropalatal airway, and often results in supraphysiologic airway dimensions. These retropalatal changes may be important in relieving severe UAO in patients with PRS. Generalizability of our results is limited by small cohort size and patient heterogeneity.

Keywords

airway morphology cleft palate mandibular distraction osteogenesis Pierre Robin sequence tongue-based upper airway obstruction

Get full access to this article

View all access options for this article.

References

Abramson

Susarla

Troulis

Kaban

. Age-related changes of the upper airway assessed by 3-dimensional computed tomography. J Craniofac Surg. 2009;20(suppl 1):657–663.

Berry

Budhiraja

Gottlieb

Gozal

Iber

Kapur

Marcus

Mehra

Parthasarathy

Quan

, et al. Rules for scoring respiratory events in sleep: update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events. JCSM. 2012;8(5):597–619.

Breik

Tivey

Umapathysivam

Anderson

. 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):769–782.

Evans

Sie

Hopper

Glass

Hing

Cunningham

. Robin sequence: from diagnosis to development of an effective management plan. Pediatrics. 2011;127(5):936–948.

Figueroa

Glupker

Fitz

BeGole

. Mandible, tongue, and airway in Pierre Robin sequence: a longitudinal cephalometric study. Cleft Palate Craniofac J. 1991;28(4):425–434.

Flores

Greathouse

Costa

Tahiri

Soleimani

Tholpady

. Defining failure and its predictors in mandibular distraction for Robin sequence. J Craniomaxillofac Surg. 2015;43(8):1614–1619.

Hong

Kearns

. Airway characteristics of infants with Pierre Robin sequence who undergo mandibular distraction osteogenesis. Ear Nose Throat J. 2015;94(8):E25–E29.

Khansa

Hall

Madhoun

Splaingard

Baylis

Kirschner

Pearson

. 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–983e.

Lee

Bradley

. Surgical considerations in Pierre Robin sequence. Clin Plast Surg. 2014;41(2):211–217.

10.

Marques

De Sousa

Carneiro

Barbieri

Bettiol

Gutierrez

. Clinical experience with infants with Robin sequence: a prospective study. Cleft Palate Craniofac J. 2001;38(2):171–178.

11.

Mhlaba

Chen

Bandla

Baroody

Reid

. Predictive soft tissue airway volume analysis in mandibular distraction. J Cran Surg. 2016;27(1):181–184.

12.

Perlyn

Schmelzer

Sutera

Kane

Govier

Marsh

. Effect of distraction osteogenesis of the mandible on upper airway volume and resistance in children with micrognathia. Plast Reconstr Surg. 2002;109(6):1809–1818.

13.

Pfaff

Metzler

Kim

Steinbacher

. Mandibular volumetric increase following distraction osteogenesis. British J Plast Surg. 2014;67(9):1209–1214.

14.

Robin

A fall of the base of the tongue considered as a new cause of nasopharyngeal respiratory impairment: Pierre Robin sequence, a translation. 1923. Plast Reconstr Surg. 1994;93(6):1301–1303.

15.

Runyan

Uribe-Rivera

Karlea

Meinzen-Derr

Rothchild

Saal

Hopkin

Gordon

. Cost analysis of mandibular distraction versus tracheostomy in neonates with Pierre Robin sequence. Otolaryngol Head Neck Surg. 2014;151(5):811–818.

16.

Sher

Shprintzen

Thorpy

. Endoscopic observations of obstructive sleep apnea in children with anomalous upper airways: predictive and therapeutic value. Int J Ped Otorhinol. 1986;11(2):135–146.

17.

Strohl

Butler

Malhotra

. Mechanical properties of the upper airway. Compr Physiol. 2012;2(3):1853–1872.

18.

Zellner

Mhlaba

Reid

Steinbacher

. Does mandibular distraction vector influence airway volumes and outcome? J Oral Maxill Surg. 2017;75(1):167–177.

19.

Zimmerman

Humphries

Roy

Reid

. Mandibular distraction for micrognathia in neonates. NeoReviews. 2018;19:1–16.

Supplementary Material

Please find the following supplemental material available below.

For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.

For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.

0.00 MB