Awake Flexible Fiberoptic Laryngoscopy to diagnose glossoptosis in Robin Sequence patients

Clin Otolaryngol. 2016 Oct;41(5):467-71. doi: 10.1111/coa.12552. Epub 2016 Feb 9.

Abstract

Background: Robin Sequence (RS) is usually defined as the combination of micrognathia, glossoptosis and upper airway obstruction. No objective criteria to diagnose RS exist. To compare management strategy results, a single RS definition using objective criteria is needed. The most frequently used primary diagnostic tool for glossoptosis is awake Flexible Fiberoptic Laryngoscopy (aFFL).

Objectives: To determine the reliability of the aFFL videos as an independent diagnostic tool itself, rather than on the complete evaluation of a patient.

Design, setting, participants: All RS individuals from an existing cohort with an available aFFL video were included retrospectively. Thirty age-matched patients without pathologic findings on aFFL were used as controls. aFFL videos were scored by six otolaryngologists as: a. Marked glossoptosis, b. Mild glossoptosis, c. Severity unknown, d. No glossoptosis, e. Insufficient video quality. Videos were anonymised and rated twice, in altered sequences, after a washout period of minimally 2 weeks.

Main outcome measures: Inter-rater and intrarater agreement.

Results: Twenty-six videos of 16 RS patients and 30 videos of controls were included. Inter-rater agreement was fair in the whole group (κ: 0.320) and RS group (κ: 0.226), and fair to moderate in determining presence of glossoptosis (total group κ: 0.430; RS κ: 0.302; controls κ: 0.212). The intrarater agreement for the presence of glossoptosis in RS was moderate (κ: 0.541).

Conclusions: aFFL offers fair to moderate inter-rater agreement, with moderate intrarater agreement, in evaluating glossoptosis in RS. Using aFFL as the single tool in choosing management strategies in RS seems insufficient. There is need for a more reliable, patient friendly diagnostic tool or an internationally accepted aFFL scoring system, to diagnose glossoptosis in RS.

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • Female
  • Fiber Optic Technology
  • Glossoptosis / diagnosis*
  • Glossoptosis / etiology
  • Humans
  • Infant
  • Infant, Newborn
  • Laryngoscopy / methods*
  • Male
  • Pierre Robin Syndrome / complications*
  • Reproducibility of Results
  • Retrospective Studies
  • Video Recording