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

Autor:	Dunworth K; Department of Surgery, Division of Plastic, Maxillofacial, and Oral Surgery, Duke University School of Medicine, Durham, NC, USA., Sharif-Askary B; Department of Surgery, Division of Plastic, Maxillofacial, and Oral Surgery, Duke University School of Medicine, Durham, NC, USA., Grames L; Cleft Palate and Craniofacial Institute, St. Louis Children's Hospital, St. Louis, USA., Jones C; Duke Cleft & Craniofacial Center, Duke Children's Hospital, Durham, NC, USA.; Division of Plastic, Maxillofacial, and Oral Surgery, Department of Surgery, Duke University Health System, Durham, NC, USA., Kern J; Duke Cleft & Craniofacial Center, Duke Children's Hospital, Durham, NC, USA.; Department of Speech Pathology & Audiology, Duke University Hospital, Durham, NC, USA., Nyswonger-Sugg J; Duke Cleft & Craniofacial Center, Duke Children's Hospital, Durham, NC, USA.; Department of Speech Pathology & Audiology, Duke University Hospital, Durham, NC, USA., Suárez A; Duke Cleft & Craniofacial Center, Duke Children's Hospital, Durham, NC, USA.; Department of Speech Pathology & Audiology, Duke University Hospital, Durham, NC, USA., Thompson K; Cleft Lip and Palate Program/Craniofacial Program, Boston Children's Hospital, Boston, MA, USA.; Department of Otolaryngology and Communication Enhancement, Boston Children's Hospital, Boston, MA, USA., Ching J; University of Florida Craniofacial Center, University of Florida, Gainesville, FL, USA.; Division of Plastic and Reconstructive Surgery, Department of Surgery, University of Florida, Gainesville, FL, USA., Golden B; Pediatric Cleft Lip and Palate Surgery Program, Orlando Health Arnold Palmer Hospital for Children, Orlando, FL, USA., Merrill C; Division of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine in St. Louis, St. Louis, USA., Nguyen P; Division of Plastic Surgery, Department of Surgery, University of Texas Health Science Center at Houston, Houston, TX, USA., Patel K; Cleft Palate and Craniofacial Institute, St. Louis Children's Hospital, St. Louis, USA.; Division of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine in St. Louis, St. Louis, USA., Rogers-Vizena CR; Cleft Lip and Palate Program/Craniofacial Program, Boston Children's Hospital, Boston, MA, USA.; Department of Surgery, Harvard Medical School, Boston, MA, USA.; Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, MA, USA., Rottgers SA; Cleft and Craniofacial Center, Johns Hopkins Children's Center, Baltimore, MD, USA.; Division of Plastic and Reconstructive Surgery, Department of Surgery, Johns Hopkins Medicine, Baltimore, MD, USA., Skolnick GB; Division of Plastic and Reconstructive Surgery, Department of Surgery, Washington University School of Medicine in St. Louis, St. Louis, USA., Allori AC; Department of Surgery, Division of Plastic, Maxillofacial, and Oral Surgery, Duke University School of Medicine, Durham, NC, USA.; Duke Cleft & Craniofacial Center, Duke Children's Hospital, Durham, NC, USA.; Division of Plastic, Maxillofacial, and Oral Surgery, Department of Surgery, Duke University Health System, Durham, NC, USA.
Jazyk:	angličtina
Zdroj:	The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association [Cleft Palate Craniofac J] 2023 Oct 16, pp. 10556656231207469. Date of Electronic Publication: 2023 Oct 16.
DOI:	10.1177/10556656231207469
Abstrakt:	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. Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Databáze:	MEDLINE
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