title: Orofacial dysfunction in ectodermal dysplasias measured using the Nordic Orofacial Test-Screening protocol.
abstract: OBJECTIVE
  To screen orofacial function in people with various ectodermal dysplasia (ED) syndromes and compare with a healthy reference sample.
  
  
  METHODS
  The ED group comprised 46 individuals (30 M and 16 F; mean age 14.5 years, range 3-55). Thirty-two had hypohidrotic ED, while 14 had other ED syndromes. The reference sample comprised 52 healthy individuals (22 M and 30 F; mean age 24.9 years, range 3-55). Orofacial function was screened using the Nordic Orofacial Test-Screening (NOT-S) protocol containing 12 orofacial function domains (maximum score 12 points).
  
  
  RESULTS
  The total NOT-S score was higher in the ED group than in the healthy group (mean 3.5 vs. 0.4; p<0.001). The dysfunctions most frequently recorded in the subjects with ED occurred in the domains chewing and swallowing (82.6%), dryness of the mouth (45.7%), and speech (43.5%). Those with other ED syndromes scored non-significantly higher than those with hypohidrotic ED (mean 4.6 vs. 3.0; p>0.05).
  
  
  CONCLUSIONS
  Individuals with ED scored higher than a healthy reference sample in all NOT-S domains, especially in the chewing and swallowing, dryness of the mouth, and speech domains. Orofacial function areas and treatment and training outcomes need to be more closely evaluated and monitored.
authors.affiliation: National Oral Disability Centre, Institute for Postgraduate Dental Education, Jönköping, Sweden. birgitta.bergendal@lj.se
authors.forename: Birgitta
authors.initials: B
authors.lastname: Bergendal
authors.forename: Anita
authors.initials: A
authors.lastname: McAllister
authors.forename: Christina
authors.initials: C
authors.lastname: Stecksén-Blicks
mesh_terms: D000293:Adolescent; D000328:Adult; D002648:Child; D002675:Child, Preschool; D003680:Deglutition Disorders / Q000175:diagnosis*; D004476:Ectodermal Dysplasia / Q000150:complications* / Q000503:physiopathology; D005145:Face; D005149:Facial Expression; D005260:Female; D006685:Hoarseness / Q000175:diagnosis; D006801:Humans; D008297:Male; D008334:Mandible / Q000502:physiology; D008403:Mass Screening; D008409:Mastication / Q000502:physiology*; D008410:Masticatory Muscles / Q000502:physiology; D008875:Middle Aged; D009666:Nose / Q000502:physiology; D012119:Respiration; D013064:Speech Disorders / Q000175:diagnosis*; D014831:Voice / Q000502:physiology; D014987:Xerostomia / Q000175:diagnosis*; D055815:Young Adult