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Göteborgs universitets publikationer

Orofacial dysfunction in children and adolescents with myotonic dystrophy.

Författare och institution:
Lotta Sjögreen (Institutionen för neurovetenskap och fysiologi, sektionen för klinisk neurovetenskap och rehabilitering); Monica Engvall (Institutionen för odontologi); Anne-Berit Ekström (Institutionen för kliniska vetenskaper, sektionen för kvinnors och barns hälsa); Anette Lohmander (Institutionen för neurovetenskap och fysiologi, sektionen för klinisk neurovetenskap och rehabilitering); Stavros Kiliaridis (Institutionen för odontologi); Mar Tulinius (Institutionen för kliniska vetenskaper, sektionen för kvinnors och barns hälsa)
Publicerad i:
Developmental medicine and child neurology, 49 ( 1 ) s. 18-22
Artikel, refereegranskad vetenskaplig
Sammanfattning (abstract):
Myotonic dystrophy (DM) is a neuromuscular disorder caused by an expansion of a CTG repeat sequence on chromosome 19q13. The aim of the present study was to describe the characteristics and prevalence of oral motor dysfunction in a cohort of children and adolescents with DM and to correlate different aspects of oral motor function with the type of DM and sex. Fifty-six individuals with DM (30 males, 26 females; median age 13y 2mo; range 2y 6mo-21y 5mo) were compared with healthy controls. They were divided into four subgroups: severe congenital DM (n=18); mild congenital DM (n=18); childhood DM (n=18); and classical DM (n=2). A speech-language pathologist assessed different variables of oral motor function, intelligibility, and lip force. The families used a questionnaire to report on eating difficulties and drooling. All individuals with DM had impaired facial expression. Intelligibility was moderately or severely reduced in 30 patients (60%), excluding six patients without speech. Most had a moderate or severe impairment of lip motility (76.0%), tongue motility (52.2%), and lip force (69.2%), causing deviant production of bilabial and dental consonants. The families reported problems with eating (51.9%) and drooling (37.0%). Oral motor dysfunction was most prominent in congenital DM, and males were more affected than females.
Ämne (baseras på Högskoleverkets indelning av forskningsämnen):
Adolescent, Articulation Disorders, diagnosis, epidemiology, physiopathology, Child, Child, Preschool, Cohort Studies, Cross-Sectional Studies, Dyskinesia, Drug-Induced, diagnosis, epidemiology, physiopathology, Female, Humans, Lip, physiopathology, Male, Myotonic Dystrophy, epidemiology, genetics, Observer Variation, Parents, Phonetics, Prevalence, Severity of Illness Index, Sex Distribution
Postens nummer:
Posten skapad:
2008-10-23 14:34
Posten ändrad:
2011-01-20 10:00

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