Patient Factors Influencing Speech Outcomes in Velopharyngeal Function Following Initial Cleft Palate Repair: A Systematic Review and Meta-Analysis

David C G Sainsbury*, Caroline C Williams, Sophie Butterworth, Catherine de Blacam, Matthew Fell, Joanne Mullen, William Breakey, Colm Murphy, Peter Hodgkinson, Yvonne E Wren

*Corresponding author for this work

Research output: Contribution to journalArticle (Academic Journal)peer-review

2 Citations (Scopus)
141 Downloads (Pure)

Abstract

Objective
Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair.
Design
A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed.
Setting
Systematic review and meta-analysis of relevant literature.
Patients/Participants
Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants.
Interventions
Individuals undergoing initial palatoplasty.
Main Outcome Measures
Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded.
Results
Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up.
Conclusions
Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech.
Systematic Review Registration
Registered with PROSPERO CRD42017051624.
Original languageEnglish
JournalCleft Palate-Craniofacial Journal
Early online date2 Aug 2023
DOIs
Publication statusE-pub ahead of print - 2 Aug 2023

Bibliographical note

Funding Information:
This manuscript is respectfully dedicated to the memory of Professor James Law (Newcastle University, UK), the foremost researcher in child language development, who provided invaluable advice. The authors are extremely grateful to Rachel Playforth (Assistant Librarian, Royal Sussex County Hospital) for her help with the search strategy and reviewing the protocol and to Claire Leng and Helen Charlton (Senior Library Assistants, Newcastle upon Tyne Hospitals Foundation Trust) for help in retrieving papers. The Cleft and Craniofacial Conditions Clinical Studies Group and the Cleft and Craniofacial Conditions Early Career Researcher Group supported the activities of the Cleft Multidisciplinary Collaborative. Ambika Chadha was involved in preliminary discussions regarding the methodology for this review. Evidence synthesis support was provided by Bronia Arnott, Suzanne McDonald and Fiona Beyer (Newcastle University). This work contained in this manuscript was presented at the 14th International Cleft and Craniofacial Congress, 13 July 2022, Edinburgh, UK.

Funding Information:
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the Craniofacial Society of Great Britain and Ireland.

Publisher Copyright:
© 2023, American Cleft Palate Craniofacial Association.

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