دورية أكاديمية
أعرض في EDS

Oral Health-Related Quality of Life in Rare Disorders of Congenital Facial Weakness.

التفاصيل البيبلوغرافية
العنوان: Oral Health-Related Quality of Life in Rare Disorders of Congenital Facial Weakness.
المؤلفون: Liberton DK; Craniofacial Anomalies and Regeneration Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA., Almpani K; Craniofacial Anomalies and Regeneration Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA., Mishra R; Department of Orofacial Sciences, School of Dentistry, University of California, San Francisco, CA 94143, USA., Bassim C; Craniofacial Anomalies and Regeneration Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA., Van Ryzin C; Metabolic Medicine Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892, USA., On Behalf Of The Moebius Syndrome Research Consortium, Webb BD; Department of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA.; Department of Pediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI 53705, USA., Jabs EW; Department of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA.; Department of Clinical Genomics, Mayo Clinic, Rochester, MN 55902, USA., Engle EC; Departments of Neurology and Ophthalmology, Boston Children's Hospital, Boston, MA 02115, USA.; Departments of Neurology and Ophthalmology, Harvard Medical School, Boston, MA 02115, USA.; Howard Hughes Medical Institute, Chevy Chase, MD 20815, USA., Collins FS; Center for Precision Health Research, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892, USA., Manoli I; Metabolic Medicine Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892, USA., Lee JS; Craniofacial Anomalies and Regeneration Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA.
المصدر: International journal of environmental research and public health [Int J Environ Res Public Health] 2024 May 13; Vol. 21 (5). Date of Electronic Publication: 2024 May 13.
نوع المنشور: Journal Article
اللغة: English
بيانات الدورية: Publisher: MDPI Country of Publication: Switzerland NLM ID: 101238455 Publication Model: Electronic Cited Medium: Internet ISSN: 1660-4601 (Electronic) Linking ISSN: 16604601 NLM ISO Abbreviation: Int J Environ Res Public Health Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Basel : MDPI, c2004-
مواضيع طبية MeSH: Quality of Life* , Oral Health*, Humans ; Male ; Female ; Adult ; Young Adult ; Adolescent ; Child ; Middle Aged ; Facial Paralysis/psychology ; Facial Paralysis/physiopathology ; Case-Control Studies ; Rare Diseases/psychology
مستخلص: Congenital facial weakness (CFW) encompasses a heterogenous set of rare disorders presenting with decreased facial movement from birth, secondary to impaired function of the facial musculature. The aim of the present study is to provide an analysis of subject-reported oral health-related quality of life (OHRQoL) in congenital facial weakness (CFW) disorders. Forty-four subjects with CFW and age- and sex- matched controls were enrolled in an Institutional Review Board (IRB)-approved study. Demographic data, medical and surgical history, comprehensive oral examination, and the Oral Health Impact Profile (OHIP-14) were obtained. Compared to unaffected controls, subjects with CFW had higher OHIP-14 scores overall (mean ± SD: 13.11 ± 8.11 vs. 4.46 ± 4.98, p < 0.0001) and within five of seven oral health domains, indicating decreased OHRQoL. Although subjects with Moebius syndrome (MBS) were noted to have higher OHIP-14 scores than those with Hereditary Congenital Facial Paresis (HCFP), there was no significant correlation in OHIP-14 score to age, sex, or specific diagnosis. An increase in OHIP-14 scores in subjects was detected in those who had undergone reanimation surgery. In conclusion, subjects with CFW had poorer OHRQoL compared to controls, and subjects with MBS had poorer OHRQoL than subjects with HCFP. This study provides better understanding of oral health care needs and quality of life in a CFW cohort and suggests that guidelines for dental treatment are required.
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معلومات مُعتمدة: P50 HD105351 United States HD NICHD NIH HHS; U01 HD079068 United States HD NICHD NIH HHS; 1ZIADE000746, PI: Janice Lee; ZIAHG200389, PI: Francis S. Collins; U01HD079068, Intramural PIs: Irini Manoli, Brian Brooks, Carlo Pierpaoli, Extramural PIs: Ethylin Wang Jabs and Elizabeth Engle. Intramural Research Program of the National Institute of Dental and Craniofacial Research; National Human Genome Research Institute; Intramural-extramural collaborative grant
فهرسة مساهمة: Keywords: Carey Fineman Ziter syndrome; OHIP-14; OHRQoL; Oral Health Impact Profile; congenital fibrosis of the extraocular muscles; facial weakness disorders; hereditary congenital facial palsy; moebius syndrome; smile surgery
تواريخ الأحداث: Date Created: 20240525 Date Completed: 20240525 Latest Revision: 20240822
رمز التحديث: 20240822
مُعرف محوري في PubMed: PMC11121611
DOI: 10.3390/ijerph21050615
PMID: 38791829
قاعدة البيانات: MEDLINE
الوصف
تدمد:1660-4601
DOI:10.3390/ijerph21050615