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Orofacial dysfunction in children and adolescents with myotonic dystrophy.

Artikel i vetenskaplig tidskrift
Författare Lotta Sjögreen
Monica Engvall
Anne-Berit Ekström
Anette Lohmander
Stavros Kiliaridis
Mar Tulinius
Publicerad i Developmental medicine and child neurology
Volym 49
Nummer/häfte 1
Sidor 18-22
ISSN 0012-1622
Publiceringsår 2007
Publicerad vid Institutionen för odontologi
Institutionen för neurovetenskap och fysiologi, sektionen för klinisk neurovetenskap och rehabilitering
Institutionen för kliniska vetenskaper
Sidor 18-22
Språk en
Länkar dx.doi.org/10.1111/j.1469-8749.2007...
Ämnesord 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
Ämneskategorier Medicin och Hälsovetenskap

Sammanfattning

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.

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