Presentation and management of type 1 laryngeal clefts: A systematic review and meta-analysis

Int J Pediatr Otorhinolaryngol. 2020 Nov:138:110370. doi: 10.1016/j.ijporl.2020.110370. Epub 2020 Sep 9.

Abstract

Objective: To evaluate outcomes of injection laryngoplasty (IL) and endoscopic surgical repair for the treatment of type 1 laryngeal clefts (LC1) and to determine the most common presentations of LC1.

Methods: Primary studies were searched for in PubMed, Scopus, OVID, and Cochrane Library using search terms laryngeal cleft and laryngotracheoesophageal cleft for articles published from database inception through August 2019. The search identified all articles pertaining to the symptomatology and management of LC1. Meta-analysis was performed on presenting symptoms and improvement of clinical symptoms and swallow function.

Results: Twenty-four studies, comprising 713 patients with a mean age of 33.7 months (range 0-168 months), were included. The most common presenting symptoms were aspiration (80%, CI [55%-96%]), dysphagia (61%, CI [47%-74%]), and choking (57%, CI [37%-76%]). Gastroesophageal reflux disease (GERD) was the most common comorbidity (68%, CI [51%-83%]) and premature birth comprised 27% [21%-32%] of the patients. Overall, 38% of patients received IL as a primary therapy. Prior to injection, 91% [87%-94%] of patients aspirated on swallow evaluations, and 62% [55%-68%] aspirated post-injection. At an average follow-up time of 6.8 months, 90% [70%-100%] of parents reported symptom improvement. An additional 54% of patients underwent endoscopic surgical repair as primary treatment. The rate of aspiration decreased from 73% [43%-94%] to 28% [13%-46%] after repair. At a mean follow-up of 14.2 months, 80% [67%-91%] reported symptom improvement.

Conclusion: A high level of clinical suspicion is necessary to detect LC1 since its most common presenting symptoms overlap with other diseases commonly found in this patient population. Both IL and formal surgical repair were effective in managing LC1.

Keywords: Aspiration; Injection; Laryngeal cleft; Laryngoplasty; Swallow evaluation.

Publication types

  • Meta-Analysis
  • Review
  • Systematic Review

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Congenital Abnormalities* / surgery
  • Deglutition Disorders* / etiology
  • Deglutition Disorders* / surgery
  • Humans
  • Infant
  • Infant, Newborn
  • Laryngoplasty*
  • Larynx* / abnormalities
  • Larynx* / surgery
  • Retrospective Studies

Supplementary concepts

  • Laryngeal cleft