Skip to main content

Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network.

Publication ,  Journal Article
Dunworth, K; Sharif-Askary, B; Grames, L; Jones, C; Kern, J; Nyswonger-Sugg, J; Suárez, A; Thompson, K; Ching, J; Golden, B; Merrill, C ...
Published in: Cleft Palate Craniofac J
March 2025

ObjectiveTo assess the ability of a cleft-specific multi-site learning health network registry to describe variations in cleft outcomes by cleft phenotypes, ages, and treatment centers. Observed variations were assessed for coherence with prior study findings.DesignCross-sectional analysis of prospectively collected data from 2019-2022.SettingSix cleft treatment centers collected data systematically during routine clinic appointments according to a standardized protocol.Participants714 English-speaking children and adolescents with non-syndromic cleft lip/palate.InterventionRoutine multidisciplinary care and systematic outcomes measurement by cleft teams.Outcome MeasuresSpeech outcomes included articulatory accuracy measured by Percent Consonants Correct (PCC), velopharyngeal function measured by Velopharyngeal Competence (VPC) Rating Scale (VPC-R), intelligibility measured by caregiver-reported Intelligibility in Context Scale (ICS), and two CLEFT-Q™ surveys, in which patients rate their own speech function and level of speech distress.Results12year-olds exhibited high median PCC scores (91-100%), high frequency of velopharyngeal competency (62.50-100%), and high median Speech Function (80-91) relative to younger peers parsed by phenotype. Patients with bilateral cleft lip, alveolus, and palate reported low PCC scores (51-91%) relative to peers at some ages and low frequency of velopharyngeal competency (26.67%) at 5 years. ICS scores ranged from 3.93-5.0 for all ages and phenotypes. Speech Function and Speech Distress were similar across phenotypes.ConclusionsThis exploration of speech outcomes demonstrates the current ability of the cleft-specific registry to support cleft research efforts as a source of "real-world" data. Further work is focused on developing robust methodology for hypothesis-driven research and causal inference.

Duke Scholars

Published In

Cleft Palate Craniofac J

DOI

EISSN

1545-1569

Publication Date

March 2025

Volume

62

Issue

3

Start / End Page

445 / 459

Location

United States

Related Subject Headings

  • Velopharyngeal Insufficiency
  • United States
  • Speech Intelligibility
  • Speech Disorders
  • Registries
  • Prospective Studies
  • Male
  • Humans
  • Female
  • Dentistry
 

Citation

APA
Chicago
ICMJE
MLA
NLM
Dunworth, K., Sharif-Askary, B., Grames, L., Jones, C., Kern, J., Nyswonger-Sugg, J., … Allori, A. C. (2025). Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network. Cleft Palate Craniofac J, 62(3), 445–459. https://doi.org/10.1177/10556656231207469
Dunworth, Kristina, Banafsheh Sharif-Askary, Lynn Grames, Carlee Jones, Jennifer Kern, Jillian Nyswonger-Sugg, Arthur Suárez, et al. “Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network.Cleft Palate Craniofac J 62, no. 3 (March 2025): 445–59. https://doi.org/10.1177/10556656231207469.
Dunworth K, Sharif-Askary B, Grames L, Jones C, Kern J, Nyswonger-Sugg J, et al. Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network. Cleft Palate Craniofac J. 2025 Mar;62(3):445–59.
Dunworth, Kristina, et al. “Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network.Cleft Palate Craniofac J, vol. 62, no. 3, Mar. 2025, pp. 445–59. Pubmed, doi:10.1177/10556656231207469.
Dunworth K, Sharif-Askary B, Grames L, Jones C, Kern J, Nyswonger-Sugg J, Suárez A, Thompson K, Ching J, Golden B, Merrill C, Nguyen P, Patel K, Rogers-Vizena CR, Rottgers SA, Skolnick GB, Allori AC. Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network. Cleft Palate Craniofac J. 2025 Mar;62(3):445–459.

Published In

Cleft Palate Craniofac J

DOI

EISSN

1545-1569

Publication Date

March 2025

Volume

62

Issue

3

Start / End Page

445 / 459

Location

United States

Related Subject Headings

  • Velopharyngeal Insufficiency
  • United States
  • Speech Intelligibility
  • Speech Disorders
  • Registries
  • Prospective Studies
  • Male
  • Humans
  • Female
  • Dentistry