Dental care and oral health in Aagenaes syndrome/lymphedema cholestasis syndrome 1.
Identifieur interne : 001434 ( PubMed/Corpus ); précédent : 001433; suivant : 001435Dental care and oral health in Aagenaes syndrome/lymphedema cholestasis syndrome 1.
Auteurs : Mats B Gesund ; Zahra Shafiee ; Monica Drivdal ; Jan Berdén ; Kari StorhaugSource :
- Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry [ 1754-4505 ]
English descriptors
- KwdEn :
- MESH :
- complications : Cholestasis, Lymphedema.
- diagnosis : Mouth Diseases.
- therapy : Mouth Diseases.
- Adult, Dental Care for Chronically Ill, Female, Humans, Male, Middle Aged, Norway, Surveys and Questionnaires, Treatment Outcome.
Abstract
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%).
DOI: 10.1111/scd.12080
PubMed: 25039919
Links to Exploration step
pubmed:25039919Le document en format XML
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<author><name sortKey="B Gesund, Mats" sort="B Gesund, Mats" uniqKey="B Gesund M" first="Mats" last="B Gesund">Mats B Gesund</name>
<affiliation><nlm:affiliation>TAKO'Centre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway; Department of Pediatrics and Department of Clinical and Experimental Medicine, Linköping University, Centre for Orthodontics and Pediatric dentistry, Norrköping, Sweden.</nlm:affiliation>
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<author><name sortKey="Shafiee, Zahra" sort="Shafiee, Zahra" uniqKey="Shafiee Z" first="Zahra" last="Shafiee">Zahra Shafiee</name>
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<author><name sortKey="Drivdal, Monica" sort="Drivdal, Monica" uniqKey="Drivdal M" first="Monica" last="Drivdal">Monica Drivdal</name>
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<author><name sortKey="Berden, Jan" sort="Berden, Jan" uniqKey="Berden J" first="Jan" last="Berdén">Jan Berdén</name>
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<author><name sortKey="Storhaug, Kari" sort="Storhaug, Kari" uniqKey="Storhaug K" first="Kari" last="Storhaug">Kari Storhaug</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Dental care and oral health in Aagenaes syndrome/lymphedema cholestasis syndrome 1.</title>
<author><name sortKey="B Gesund, Mats" sort="B Gesund, Mats" uniqKey="B Gesund M" first="Mats" last="B Gesund">Mats B Gesund</name>
<affiliation><nlm:affiliation>TAKO'Centre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway; Department of Pediatrics and Department of Clinical and Experimental Medicine, Linköping University, Centre for Orthodontics and Pediatric dentistry, Norrköping, Sweden.</nlm:affiliation>
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<author><name sortKey="Shafiee, Zahra" sort="Shafiee, Zahra" uniqKey="Shafiee Z" first="Zahra" last="Shafiee">Zahra Shafiee</name>
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<author><name sortKey="Drivdal, Monica" sort="Drivdal, Monica" uniqKey="Drivdal M" first="Monica" last="Drivdal">Monica Drivdal</name>
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<author><name sortKey="Berden, Jan" sort="Berden, Jan" uniqKey="Berden J" first="Jan" last="Berdén">Jan Berdén</name>
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<author><name sortKey="Storhaug, Kari" sort="Storhaug, Kari" uniqKey="Storhaug K" first="Kari" last="Storhaug">Kari Storhaug</name>
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<series><title level="j">Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry</title>
<idno type="eISSN">1754-4505</idno>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term>
<term>Cholestasis (complications)</term>
<term>Dental Care for Chronically Ill</term>
<term>Female</term>
<term>Humans</term>
<term>Lymphedema (complications)</term>
<term>Male</term>
<term>Middle Aged</term>
<term>Mouth Diseases (diagnosis)</term>
<term>Mouth Diseases (therapy)</term>
<term>Norway</term>
<term>Surveys and Questionnaires</term>
<term>Treatment Outcome</term>
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<keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Cholestasis</term>
<term>Lymphedema</term>
</keywords>
<keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Mouth Diseases</term>
</keywords>
<keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Mouth Diseases</term>
</keywords>
<keywords scheme="MESH" xml:lang="en"><term>Adult</term>
<term>Dental Care for Chronically Ill</term>
<term>Female</term>
<term>Humans</term>
<term>Male</term>
<term>Middle Aged</term>
<term>Norway</term>
<term>Surveys and Questionnaires</term>
<term>Treatment Outcome</term>
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<front><div type="abstract" xml:lang="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%).</div>
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<DateCreated><Year>2015</Year>
<Month>03</Month>
<Day>11</Day>
</DateCreated>
<DateCompleted><Year>2016</Year>
<Month>12</Month>
<Day>13</Day>
</DateCompleted>
<DateRevised><Year>2016</Year>
<Month>12</Month>
<Day>30</Day>
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<Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1754-4505</ISSN>
<JournalIssue CitedMedium="Internet"><Volume>35</Volume>
<Issue>2</Issue>
<PubDate><MedlineDate>2015 Mar-Apr</MedlineDate>
</PubDate>
</JournalIssue>
<Title>Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry</Title>
<ISOAbbreviation>Spec Care Dentist</ISOAbbreviation>
</Journal>
<ArticleTitle>Dental care and oral health in Aagenaes syndrome/lymphedema cholestasis syndrome 1.</ArticleTitle>
<Pagination><MedlinePgn>83-9</MedlinePgn>
</Pagination>
<ELocationID EIdType="doi" ValidYN="Y">10.1111/scd.12080</ELocationID>
<Abstract><AbstractText Label="UNLABELLED">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%).</AbstractText>
<AbstractText Label="CONCLUSION" NlmCategory="CONCLUSIONS">Several patients with LCS1 have problems with periodontitis and tooth discoloration. Frequent dental checkups are therefore recommended.</AbstractText>
<CopyrightInformation>© 2014 Special Care Dentistry Association and Wiley Periodicals, Inc.</CopyrightInformation>
</Abstract>
<AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Bågesund</LastName>
<ForeName>Mats</ForeName>
<Initials>M</Initials>
<AffiliationInfo><Affiliation>TAKO'Centre, National Resource Centre for Oral Health in Rare Medical Conditions, Lovisenberg Diakonale Hospital, Oslo, Norway; Department of Pediatrics and Department of Clinical and Experimental Medicine, Linköping University, Centre for Orthodontics and Pediatric dentistry, Norrköping, Sweden.</Affiliation>
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<Author ValidYN="Y"><LastName>Shafiee</LastName>
<ForeName>Zahra</ForeName>
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<Author ValidYN="Y"><LastName>Drivdal</LastName>
<ForeName>Monica</ForeName>
<Initials>M</Initials>
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<Author ValidYN="Y"><LastName>Berdén</LastName>
<ForeName>Jan</ForeName>
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<Author ValidYN="Y"><LastName>Storhaug</LastName>
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<Language>eng</Language>
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<ArticleDate DateType="Electronic"><Year>2014</Year>
<Month>07</Month>
<Day>18</Day>
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<MedlineJournalInfo><Country>United States</Country>
<MedlineTA>Spec Care Dentist</MedlineTA>
<NlmUniqueID>8103755</NlmUniqueID>
<ISSNLinking>0275-1879</ISSNLinking>
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<SupplMeshList><SupplMeshName Type="Disease" UI="C535330">Aagenaes syndrome</SupplMeshName>
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<MeshHeadingList><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName>
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<MeshHeading><DescriptorName UI="D002779" MajorTopicYN="N">Cholestasis</DescriptorName>
<QualifierName UI="Q000150" MajorTopicYN="Y">complications</QualifierName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D017742" MajorTopicYN="Y">Dental Care for Chronically Ill</DescriptorName>
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<MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName>
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<MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D008209" MajorTopicYN="N">Lymphedema</DescriptorName>
<QualifierName UI="Q000150" MajorTopicYN="Y">complications</QualifierName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D008875" MajorTopicYN="N">Middle Aged</DescriptorName>
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<MeshHeading><DescriptorName UI="D009059" MajorTopicYN="N">Mouth Diseases</DescriptorName>
<QualifierName UI="Q000175" MajorTopicYN="Y">diagnosis</QualifierName>
<QualifierName UI="Q000628" MajorTopicYN="Y">therapy</QualifierName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D009664" MajorTopicYN="N">Norway</DescriptorName>
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<MeshHeading><DescriptorName UI="D011795" MajorTopicYN="N">Surveys and Questionnaires</DescriptorName>
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<MeshHeading><DescriptorName UI="D016896" MajorTopicYN="N">Treatment Outcome</DescriptorName>
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<KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">dental caries</Keyword>
<Keyword MajorTopicYN="N">government subsidy</Keyword>
<Keyword MajorTopicYN="N">lymphedema cholestasis syndrome</Keyword>
<Keyword MajorTopicYN="N">periodontitis</Keyword>
<Keyword MajorTopicYN="N">tooth discoloration</Keyword>
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