Predictors of Respiratory Dysfunction at Diagnosis of Robin Sequence

Laryngoscope. 2021 Dec;131(12):2811-2816. doi: 10.1002/lary.29685. Epub 2021 Jun 12.

Abstract

Objectives/hypothesis: Robin sequence (RS) consists of associated micrognathia, glossoptosis, and respiratory dysfunction, with or without cleft palate. Studies on how different patient characteristics impact the severity of respiratory dysfunction are scarce and contradictory; this study investigates how different features affect respiratory obstruction severity at diagnosis of RS in controlled analysis.

Study design: Retrospective cohort study that enrolled 71 RS patients under 90 days old who received care in our institution from 2009 to 2020.

Methods: The primary outcome, respiratory dysfunction, was categorized into four severity groups and analyzed using a multinomial logistic regression model that considered age, sex, mandible length, cleft palate, syndromic diagnosis, other airway anomalies, and degree of glossoptosis.

Results: Mandible length, syndromic diagnosis, and Yellon grade 3 glossoptosis were related to poorer respiratory outcomes (need for respiratory support). In univariate analysis, for each additional 1 mm of mandible length at diagnosis, a mean reduction of 28% in the risk of needing respiratory support was observed (OR = 0.72; 0.58-0.89); syndromic diagnosis and grade 3 glossoptosis also raised the risk (OR = 6.50; 1.59-26.51 and OR = 12.75; 1.03-157.14, respectively). In multivariate analysis, only mandible length significantly maintained its effects (OR = 0.73; 0.56-0.96), a 27% reduction.

Conclusions: Mandible length was an independent predictor for more severe respiratory dysfunction in RS patients, with larger mandibles showing protective effects. Syndromic diagnosis and Yellon grade 3 glossoptosis are also likely to be associated with poorer respiratory outcomes, although this was not demonstrated in multivariate analysis.

Level of evidence: 4 Laryngoscope, 131:2811-2816, 2021.

Keywords: Robin sequence; mandible; obstructive sleep apnea; respiratory dysfunction.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Female
  • Glossoptosis / complications*
  • Glossoptosis / diagnosis
  • Glossoptosis / pathology
  • Humans
  • Imaging, Three-Dimensional
  • Infant
  • Infant, Newborn
  • Male
  • Mandible / diagnostic imaging
  • Mandible / pathology
  • Organ Size
  • Pierre Robin Syndrome / complications*
  • Pierre Robin Syndrome / diagnosis
  • Pierre Robin Syndrome / pathology
  • Prognosis
  • Protective Factors
  • Respiration Disorders / diagnosis
  • Respiration Disorders / epidemiology*
  • Respiration Disorders / etiology
  • Retrospective Studies
  • Risk Assessment / methods
  • Risk Assessment / statistics & numerical data
  • Risk Factors
  • Severity of Illness Index
  • Tomography, X-Ray Computed