Severe tooth wear in Prader-Willi syndrome. A case-control study.
BACKGROUND
Prader-Willi syndrome (PWS) is a rare complex multsystemic genetic disorder characterized by severe neonatal hypotonia, endocrine disturbances, hyperphagia and obesity, mild mental retardation, learning disabilities, facial dysmorphology and oral abnormalities. The purpose of the present study was to explore the prevalence of tooth wear and possible risk factors in individuals with Prader-Willi syndrome.


METHODS
Forty-nine individuals (6-40 years) with PWS and an age- and sex-matched control group were included. Tooth wear was evaluated from dental casts and intraoral photographs and rated by four examiners using the Visual Erosion Dental Examination (VEDE) scoring system and the individual tooth wear index IA. In accordance with the VEDE scoring system, tooth wear was also evaluated clinically. Whole saliva was collected.


RESULTS
Mean VEDE score was 1.70 ± 1.44 in the PWS group and 0.46 ± 0.36 in the control group (p < 0.001). Median IA was 7.50 (2.60-30.70) in the PWS group and 2.60 (0.90-4.70) among controls (p < 0.001). In the PWS group tooth wear correlated significantly with age (VEDE; r = 0.79, p < 0.001, IA; r = 0.82, p < 0.001) and saliva secretion (VEDE; r = 0.46, p = 0.001, IA; r = 0.43, p = 0.002). Tooth grinding was also associated with tooth wear in the PWS group, as indicated by the mean VEDE 2.67 ± 1.62 in grinders and 1.14 ± 0.97 in non-grinders (p = 0.001) and median IA values 25.70 (5.48-68.55) in grinders and 5.70 (1.60-9.10) in non-grinders (p = 0.003). Multivariate linear regression analysis was performed with tooth wear as the dependent variable and PWS (yes/no), age, tooth grinding and saliva secretion as independent variables. PWS (yes/no), age and tooth grinding retained a significant association with tooth wear, VEDE (p < 0.001) and log IA (p < 0.001). The only factor significantly associated with tooth wear in the control group was age.


CONCLUSIONS
Our study provides evidence that tooth wear, in terms of both erosion and attrition, is a severe problem in Prader-Willi syndrome. There is therefore considerable need for prosthodontic rehabilitation in young adults with PWS.
TAKO-centre, Lovisenberg Diakonale Hospital, Lovisenberggt 17, 0440 Oslo, Norway. Ronnaug.Saeves@tako.no
Ronnaug
R
Saeves
Ivar
I
Espelid
Kari
K
Storhaug
Leiv
L
Sandvik
Hilde
H
Nordgarden
D000293:Adolescent; D000328:Adult; D000367:Age Factors; D002012:Bruxism / Q000150:complications; D016022:Case-Control Studies; D002648:Child; D003743:Dental Enamel / Q000473:pathology; D003804:Dentin / Q000473:pathology; D005247:Feeding Behavior; D005260:Female; D005764:Gastroesophageal Reflux / Q000150:complications; D006801:Humans; D008297:Male; D003765:Models, Dental; D023861:Photography, Dental; D011218:Prader-Willi Syndrome / Q000150:complications*; D012307:Risk Factors; D012463:Saliva / Q000737:chemistry / Q000378:metabolism; D012636:Secretory Rate / Q000502:physiology; D019217:Tooth Attrition / Q000209:etiology; D014077:Tooth Erosion / Q000209:etiology; D057085:Tooth Wear / Q000145:classification / Q000209:etiology*; D055815:Young Adult