0
We're unable to sign you in at this time. Please try again in a few minutes.
Retry
We were able to sign you in, but your subscription(s) could not be found. Please try again in a few minutes.
Retry
There may be a problem with your account. Please contact the AMA Service Center to resolve this issue.
Contact the AMA Service Center:
Telephone: 1 (800) 262-2350 or 1 (312) 670-7827  *   Email: subscriptions@jamanetwork.com
Error Message ......
Original Investigation |

Upper Airway Computed Tomography Measures and Receipt of Tracheotomy in Infants With Robin Sequence

Victoria S. Lee, MD1; Kelly N. Evans, MD2; Francisco A. Perez, MD, PhD3; Assaf P. Oron, PhD4; Jonathan A. Perkins, DO1
[+] Author Affiliations
1Division of Pediatric Otolaryngology, Department of Otolaryngology–Head and Neck Surgery, University of Washington and Seattle Children’s, Seattle
2Department of Pediatrics, Division of Craniofacial Medicine, University of Washington and Seattle Children’s, Seattle
3Department of Radiology, University of Washington and Seattle Children’s, Seattle
4Seattle Children’s Core for Biomedical Statistics, Seattle, Washington
JAMA Otolaryngol Head Neck Surg. 2016;142(8):750-757. doi:10.1001/jamaoto.2016.1010.
Text Size: A A A
Published online

Importance  Airway management in infants with Robin sequence is challenging. Objective upper airway measures associated with severe airway compromise requiring tracheotomy are needed to guide decision making.

Objectives  To define objective upper airway measures in infants with Robin sequence from craniofacial computed tomography (CT) and to identify those measures in Robin sequence associated with tracheotomy.

Design, Setting, and Participants  A cohort study (2003 to 2014, over 1-year follow-up) of 37 infants with Robin sequence evaluated for surgical management and 37 selected age- and sex-matched controls without a craniofacial condition conducted in a pediatric institution’s craniofacial center.

Main Outcomes and Measures  Define and compare CT-generated upper airway measures in these groups: infants with Robin sequence vs controls, and infants with Robin sequence with vs without tracheotomy. A negative difference signifies lower values for the Robin sequence and tracheotomy groups. Clinical data collected included age and height at time of CT scan, sex, tracheotomy presence, associated syndrome, and laboratory indicators of hypoventilation and hypoxemia. To evaluate interrater reliability, 2 raters performed each measurement in the Robin sequence group.

Results  In 74 infants, 17 of 28 measures were different between infants with Robin sequence and those in the control group. Tracheotomy was performed in 14 of 37 (38%) infants with Robin sequence. Infants with tracheotomy more commonly had associated syndromes (12 of 14 [86%] vs 11 of 23 [48%]) and a history of hypoventilation and hypoxemia (13 of 14 [93%] vs 15 of 23 [65%]). Five of the 11 measures associated with tracheotomy were reliable and simpler to measure with the following mean differences (95% CIs) between groups: tongue length, 0.87 (0.26 to 1.48); tongue position relative to palate, 0.83 (0.22 to 1.45); mandibular total length, −0.8 (−1.42 to −0.19); gonial angle, 0.71 (0.08 to 1.34); and inferior pogonial angle, 0.66 (0.02 to 1.29). Using a receiver operating characteristic analysis, a composite score of these 5 measures for predicting tracheotomy risk yielded an area under the curve of 0.83 and achieved 86% sensitivity and 74% specificity.

Conclusions and Relevance  Computed tomography measures quantifying tongue position and mandibular configuration can identify infants with Robin sequence, and importantly, differentiate those who have severe upper airway compromise requiring tracheotomy. Following validation, these measures can be used for objective upper airway assessment and for expediting clinical decision-making in these challenging cases for which no such tools currently exist.

Figures in this Article

Sign in

Purchase Options

• Buy this article
• Subscribe to the journal
• Rent this article ?

Figures

Place holder to copy figure label and caption
Figure 1.
Two- and Three-Dimensional Computed Tomographic Measures

Control patient airway measures (5-month-old): A, airway divisions; B, tongue size and position; C, hyoid position; D, lateral mandible measures; E, inferior pogonial angle; F, bigonial distance.

Graphic Jump Location
Place holder to copy figure label and caption
Figure 2.
Representative Significant Computed Tomographic Measures in an Infant With RS (Left) and Matched Control Patient (Right)

A, Larger tongue area, taller tongue height relative to the vallecula, and more posterior tongue position are shown in RS compared with control consistent with glossoptosis. B, Compressed hyoid position is illustrated in patient with RS compared with control. C and D, A shorter and flatter mandible is demonstrated in a patient with RS compared with control.

Graphic Jump Location
Place holder to copy figure label and caption
Figure 3.
Analysis of Computed Tomographic Composite Scores for Infants With RS With vs Without Tracheotomy

A, Boxplot of 10-measure (FDR <0.2, RPD <10%) and 5-measure simplified CT composite scores for tracheotomy vs no tracheotomy . B, ROC curves for all-measure, 10-measure (FDR <0.2, RPD <10%), and 5-measure simplified CT composite scores, with sensitivity and specificity indicated in parentheses. FDR indicates false discovery rate; ROC, receiver operating characteristic; RPD, relative percent difference.

Graphic Jump Location

Tables

References

Correspondence

CME
Also Meets CME requirements for:
Browse CME for all U.S. States
Accreditation Information
The American Medical Association is accredited by the Accreditation Council for Continuing Medical Education to provide continuing medical education for physicians. The AMA designates this journal-based CME activity for a maximum of 1 AMA PRA Category 1 CreditTM per course. Physicians should claim only the credit commensurate with the extent of their participation in the activity. Physicians who complete the CME course and score at least 80% correct on the quiz are eligible for AMA PRA Category 1 CreditTM.
Note: You must get at least of the answers correct to pass this quiz.
Please click the checkbox indicating that you have read the full article in order to submit your answers.
Your answers have been saved for later.
You have not filled in all the answers to complete this quiz
The following questions were not answered:
Sorry, you have unsuccessfully completed this CME quiz with a score of
The following questions were not answered correctly:
Commitment to Change (optional):
Indicate what change(s) you will implement in your practice, if any, based on this CME course.
Your quiz results:
The filled radio buttons indicate your responses. The preferred responses are highlighted
For CME Course: A Proposed Model for Initial Assessment and Management of Acute Heart Failure Syndromes
Indicate what changes(s) you will implement in your practice, if any, based on this CME course.

Multimedia

Some tools below are only available to our subscribers or users with an online account.

443 Views
0 Citations
×

Sign in

Purchase Options

• Buy this article
• Subscribe to the journal
• Rent this article ?

Related Content

Customize your page view by dragging & repositioning the boxes below.

Articles Related By Topic
Related Collections
Jobs
JAMAevidence.com

The Rational Clinical Examination: Evidence-Based Clinical Diagnosis
Evidence Summary and Review 2

The Rational Clinical Examination: Evidence-Based Clinical Diagnosis
Evidence Summary and Review 2

brightcove.createExperiences();