Velopharyngeal insufficiency after cleft palate repair in patients with isolated Robin sequence versus isolated cleft palate: A systematic review

Abstract

Background: Robin sequence (RS) is characterized by micrognathia, glossoptosis, and upper airway obstruction, and is often combined with a cleft palate. It is unclear whether RS negatively impacts the development of velopharyngeal incompetence (VPI) and attainable speech outcomes. This study systematically reviewed speech outcomes in patients with cleft and isolated RS (IRS) compared with only isolated cleft palate (ICP).Methods: A literature search following the preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines was performed using the PubMed and EMBASE databases. Articles reporting speech outcomes following primary palatoplasty in patients with IRS only or IRS versus ICP were identified. Study characteristics and methods, primary- and VPI palatoplasty, speech measurements, and post-operative complications were collected. Primary outcomes included VPI and need for speech correcting surgery (SCS). Methodological quality was appraised using the methodological index for non-randomized studies (MINORS) criteria (range: 0-16 and 0-24).Results: Nineteen studies reported VPI event rates that varied between 14% and 88% for IRS and 0% and 62% for ICP. Five out of 8 studies (67%) comparing VPI event rates between IRS and ICP found no significant difference. SCS rates varied between 0% and 48% for IRS and 0% and 24% for ICP. Six out of 9 studies (67%) comparing SCS rates between IRS and ICP, found no significant difference. Combined VPI event rates were 36.1% for the IRS group and 26% for the ICP group, for SCS rates this was 20% for IRS and 13% for ICP.Conclusion: Most articles found no significant difference between the VPI and SCS rates indicating that speech outcomes might be similar in patients with IRS and ICP. To better compare these groups a standardized international protocol is needed.