TY  - JOUR
AB  - Background: The aim of this study was to collect information on oral health-related quality of life (OHRQoL) in people with rare diseases. 

Methods: A questionnaire comprising free text questions and the German version of the standardized Oral Health Impact Profile-14 (OHIP-14) questionnaire on OHRQoL was developed. All participants who indicated oral symptoms in the questionnaire were included in a cluster analysis. Different cluster analyses were performed (Ward's, k-Means) to find symptom profile groups in the data. 

Results: A total of 484 questionnaires with 96 rare diseases were included in the study. The most reported symptoms were anomalies of the tooth formation, dysgnathia, changes in number of the teeth, and malocclusions. The OHIP mean values of the five resulting symptom clusters ranged from 15.1 to 19.9, which is very high compared to the general population in Germany, which has a mean value of 4.09. 

Discussion: All investigated symptoms show a negative association with OHRQoL, but the strongest were for symptoms of the oral mucosa and periodontal diseases. All the symptoms described in this cluster analysis can lead to considerably higher mean values of the OHIP total score among people with rare disease and thus to worse OHRQoL than reported in the general population.
AD  - Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial Manifestations (RDOM), University Hospital Munster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Munster, Germany. s_wiem07@uni-muenster.de.
Institute of German Dentists (IDZ), Research Focus: Sociology of medicine and Health Psychology, Universitatsstrasse 73, D-50931 Koln, Germany. n.frenzel@idz.institute.
Institute of German Dentists (IDZ), Research Focus: Sociology of medicine and Health Psychology, Universitatsstrasse 73, D-50931 Koln, Germany. r.jordan@idz.institute.
Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial Manifestations (RDOM), University Hospital Munster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Munster, Germany. johannes.kleinheinz@ukmuenster.de.
Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial Manifestations (RDOM), University Hospital Munster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Munster, Germany. marcel.hanisch@ukmuenster.de.
AN  - 30011949
AU  - Wiemann, Sabrina
AU  - Frenzel Baudisch, Nicolas
AU  - Jordan, A. Rainer
AU  - Kleinheinz, Johannes
AU  - Hanisch, Marcel
C2  - PMC6068528
DA  - Jul 15
DO  - 10.3390/ijerph15071493
IS  - 7
J2  - Int J Environ Res Public Health
KW  - Ohip-14
OHRQoL
Rare diseases
Oral health
Outcome
Patient reported
Quality of life
L1  - internal-pdf://1033842261/Wiemann-2018-Oral Symptoms and Oral Health-Rel.pdf
N1  - Wiemann, Sabrina
Frenzel Baudisch, Nicolas
Jordan, Rainer A
Kleinheinz, Johannes
Hanisch, Marcel
eng
Switzerland
2018/07/18 06:00
Int J Environ Res Public Health. 2018 Jul 15;15(7). pii: ijerph15071493. doi: 10.3390/ijerph15071493.
PY  - 2018
SN  - 1660-4601 (Electronic)
1660-4601 (Linking)
ST  - Oral Symptoms and Oral Health-Related Quality of Life in People with Rare Diseases in Germany: A Cross-Sectional Study
T2  - International Journal of Environmental Research and Public Health
TI  - Oral Symptoms and Oral Health-Related Quality of Life in People with Rare Diseases in Germany: A Cross-Sectional Study
UR  - https://www.ncbi.nlm.nih.gov/pubmed/30011949
VL  - 15
ID  - 4847
ER  -