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Dental care and oral health in Aagenaes syndrome/lymphedema cholestasis syndrome 1
Linköping University, Department of Clinical and Experimental Medicine, Division of Clinical Sciences. Region Östergötland, Public Dental Health Care. Linköping University, Faculty of Medicine and Health Sciences. TakoCentre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway.
TakoCentre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway.
Centre for Rare Disorders, Oslo University Hospital, Oslo, Norway.
TakoCentre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway.
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2015 (English)In: Special Care in Dentistry, ISSN 0275-1879, Vol. 35, no 2, 83-89 p.Article in journal (Refereed) Published
Abstract [en]

Aagenaes syndrome/lymphedema cholestasis syndrome 1 (LCS1) is a rare genetic disorder characterized by neonatal cholestasis and lymphedema. The aim was to assess dental care and oral health in adults with LCS1. Fifteen (9M, 6F) individuals diagnosed with LCS1, aged 19-59 years participated. The study evaluated salivary secretion rate, dental radiographs, intraoral photos and included a questionnaire. Eight (53%) had regular dental checkups. Three had received subsidized dental care. Seven (47%) had two or more subjective symptoms of xerostomia. Three (20%) had a decreased stimulated salivary secretion rate below 0.7 mL/minute. Seven (47%) had dentin caries. Marginal periodontitis was found in all six patients above 35 years of age, but not before that age. Thirteen (87%) had tooth discoloration, which was extensive in three (20%). Conclusion. Several patients with LCS1 have problems with periodontitis and tooth discoloration. Frequent dental checkups are therefore recommended. © 2014 Special Care Dentistry Association and Wiley Periodicals, Inc.

Place, publisher, year, edition, pages
Wiley: 12 months , 2015. Vol. 35, no 2, 83-89 p.
Keyword [en]
dental caries; government subsidy; lymphedema cholestasis syndrome; periodontitis; tooth discoloration
National Category
Dentistry
Identifiers
URN: urn:nbn:se:liu:diva-119300DOI: 10.1111/scd.12080PubMedID: 25039919ScopusID: 2-s2.0-84924492179OAI: oai:DiVA.org:liu-119300DiVA: diva2:820732
Available from: 2015-06-12 Created: 2015-06-12 Last updated: 2016-04-11

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Bågesund, Mats
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Division of Clinical SciencesPublic Dental Health CareFaculty of Medicine and Health Sciences
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