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

Kristina Dunworth, Banafsheh Sharif-Askary, Lynn Grames, Carlee Jones, Jennifer Kern, Jillian Nyswonger-Sugg, Arthur Suárez, Karen Thompson, Jessica Ching, Brent Golden, Corinne Merrill, Phuong Nguyen, Kamlesh Patel, Carolyn R. Rogers-Vizena, S. Alex Rottgers, Gary B. Skolnick, Alexander C. Allori

Research output: Contribution to journalArticlepeer-review


Objective: To 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. Design: Cross-sectional analysis of prospectively collected data from 2019–2022. Setting: Six cleft treatment centers collected data systematically during routine clinic appointments according to a standardized protocol. Participants: 714 English-speaking children and adolescents with non-syndromic cleft lip/palate. Intervention: Routine multidisciplinary care and systematic outcomes measurement by cleft teams. Outcome Measures: Speech 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. Results: 12year-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. Conclusions: This 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.

Original languageEnglish
JournalCleft Palate-Craniofacial Journal
StateAccepted/In press - 2023


  • articulation
  • cleft lip
  • cleft lip and palate
  • cleft palate
  • nasality
  • nonsyndromic clefting
  • outcomes
  • pediatrics
  • registry
  • resonance
  • speech assessment
  • speech development
  • speech disorders
  • velopharyngeal dysfunction
  • velopharyngeal function


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