Hurler syndrome: a case report of a 5-year follow-up of dental findings after bone marrow transplantation.
Identifieur interne : 000332 ( Main/Exploration ); précédent : 000331; suivant : 000333Hurler syndrome: a case report of a 5-year follow-up of dental findings after bone marrow transplantation.
Auteurs : Rose O. Wadenya [États-Unis] ; Angela M. Stout ; Avin Gupta ; Janet MongeSource :
- 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 ]
Descripteurs français
- KwdFr :
- Canine (malformations), Conditionnement pour greffe (MeSH), Dent de lait (malformations), Dent incluse (étiologie), Humains (MeSH), Hypoplasie de l'émail dentaire (étiologie), Irradiation corporelle totale (MeSH), Maladies des dents (étiologie), Molaire (malformations), Mucopolysaccharidose de type I (chirurgie), Mucopolysaccharidose de type I (complications), Nourrisson (MeSH), Odontogenèse (physiologie), Racine dentaire (malformations), Transplantation de moelle osseuse (MeSH), Éruption dentaire (MeSH), Éruption dentaire ectopique (étiologie), Études de suivi (MeSH).
- MESH :
- chirurgie : Mucopolysaccharidose de type I.
- malformations : Canine, Dent de lait, Molaire, Mucopolysaccharidose de type I, Racine dentaire.
- physiologie : Odontogenèse.
- étiologie : Dent incluse, Hypoplasie de l'émail dentaire, Maladies des dents, Éruption dentaire ectopique.
- Conditionnement pour greffe, Humains, Irradiation corporelle totale, Nourrisson, Transplantation de moelle osseuse, Éruption dentaire, Études de suivi.
English descriptors
- KwdEn :
- Bone Marrow Transplantation (MeSH), Cuspid (abnormalities), Dental Enamel Hypoplasia (etiology), Follow-Up Studies (MeSH), Humans (MeSH), Infant (MeSH), Molar (abnormalities), Mucopolysaccharidosis I (complications), Mucopolysaccharidosis I (surgery), Odontogenesis (physiology), Tooth Diseases (etiology), Tooth Eruption (MeSH), Tooth Eruption, Ectopic (etiology), Tooth Root (abnormalities), Tooth, Deciduous (abnormalities), Tooth, Unerupted (etiology), Transplantation Conditioning (MeSH), Whole-Body Irradiation (MeSH).
- MESH :
- abnormalities : Cuspid, Molar, Tooth Root, Tooth, Deciduous.
- complications : Mucopolysaccharidosis I.
- etiology : Dental Enamel Hypoplasia, Tooth Diseases, Tooth Eruption, Ectopic, Tooth, Unerupted.
- physiology : Odontogenesis.
- surgery : Mucopolysaccharidosis I.
- Bone Marrow Transplantation, Follow-Up Studies, Humans, Infant, Tooth Eruption, Transplantation Conditioning, Whole-Body Irradiation.
Abstract
Hurler syndrome is a rare autosomal recessive disorder of mucopolysaccharide metabolism. It results from a deficiency in lysosomal enzymes responsible for the breakdown of glycosaminoglycans. Affected individuals may show progressive physical and mental deterioration as glycosaminoglycans are deposited in the organs of the body. Bone marrow transplantation (BMT) is effective in improving some of the clinical manifestations of Hurler syndrome. Death is caused by cardiorespiratory failure and usually occurs before the second decade of life. In this case report, the course of dental development was followed over 5 years, from the primary dentition into the permanent dentition, of a child who was successfully treated with a bone marrow transplant in infancy. The timing of bone marrow therapy has significant and variable effect on the stages of tooth development with implications for the long-term maintenance of the dentition.
DOI: 10.1111/j.1754-4505.2009.00115.x
PubMed: 20051069
Affiliations:
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Le document en format XML
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Wadenya</name><affiliation wicri:level="2"><nlm:affiliation>Department of Preventive and Restorative Sciences, Division of Pediatric and Community Oral Health, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania, USA. wadenya@dental.upenn.edu</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Preventive and Restorative Sciences, Division of Pediatric and Community Oral Health, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania</wicri:regionArea><placeName><region type="state">Pennsylvanie</region></placeName></affiliation></author><author><name sortKey="Stout, Angela M" sort="Stout, Angela M" uniqKey="Stout A" first="Angela M" last="Stout">Angela M. 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It results from a deficiency in lysosomal enzymes responsible for the breakdown of glycosaminoglycans. Affected individuals may show progressive physical and mental deterioration as glycosaminoglycans are deposited in the organs of the body. Bone marrow transplantation (BMT) is effective in improving some of the clinical manifestations of Hurler syndrome. Death is caused by cardiorespiratory failure and usually occurs before the second decade of life. In this case report, the course of dental development was followed over 5 years, from the primary dentition into the permanent dentition, of a child who was successfully treated with a bone marrow transplant in infancy. The timing of bone marrow therapy has significant and variable effect on the stages of tooth development with implications for the long-term maintenance of the dentition.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">20051069</PMID><DateCompleted><Year>2010</Year><Month>03</Month><Day>25</Day></DateCompleted><DateRevised><Year>2010</Year><Month>01</Month><Day>06</Day></DateRevised><Article PubModel="Print"><Journal><ISSN IssnType="Electronic">1754-4505</ISSN><JournalIssue CitedMedium="Internet"><Volume>30</Volume><Issue>1</Issue><PubDate><MedlineDate>2010 Jan-Feb</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>Hurler syndrome: a case report of a 5-year follow-up of dental findings after bone marrow transplantation.</ArticleTitle><Pagination><MedlinePgn>14-7</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1111/j.1754-4505.2009.00115.x</ELocationID><Abstract><AbstractText>Hurler syndrome is a rare autosomal recessive disorder of mucopolysaccharide metabolism. It results from a deficiency in lysosomal enzymes responsible for the breakdown of glycosaminoglycans. Affected individuals may show progressive physical and mental deterioration as glycosaminoglycans are deposited in the organs of the body. Bone marrow transplantation (BMT) is effective in improving some of the clinical manifestations of Hurler syndrome. Death is caused by cardiorespiratory failure and usually occurs before the second decade of life. In this case report, the course of dental development was followed over 5 years, from the primary dentition into the permanent dentition, of a child who was successfully treated with a bone marrow transplant in infancy. The timing of bone marrow therapy has significant and variable effect on the stages of tooth development with implications for the long-term maintenance of the dentition.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Wadenya</LastName><ForeName>Rose O</ForeName><Initials>RO</Initials><AffiliationInfo><Affiliation>Department of Preventive and Restorative Sciences, Division of Pediatric and Community Oral Health, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania, USA. wadenya@dental.upenn.edu</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Stout</LastName><ForeName>Angela M</ForeName><Initials>AM</Initials></Author><Author ValidYN="Y"><LastName>Gupta</LastName><ForeName>Avin</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>Monge</LastName><ForeName>Janet</ForeName><Initials>J</Initials></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Spec Care Dentist</MedlineTA><NlmUniqueID>8103755</NlmUniqueID><ISSNLinking>0275-1879</ISSNLinking></MedlineJournalInfo><CitationSubset>D</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D016026" MajorTopicYN="Y">Bone Marrow Transplantation</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D003481" MajorTopicYN="N">Cuspid</DescriptorName><QualifierName UI="Q000002" MajorTopicYN="N">abnormalities</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D003744" MajorTopicYN="N">Dental Enamel Hypoplasia</DescriptorName><QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D005500" MajorTopicYN="N">Follow-Up Studies</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D007223" MajorTopicYN="N">Infant</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008963" MajorTopicYN="N">Molar</DescriptorName><QualifierName UI="Q000002" MajorTopicYN="N">abnormalities</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008059" MajorTopicYN="N">Mucopolysaccharidosis I</DescriptorName><QualifierName UI="Q000150" MajorTopicYN="N">complications</QualifierName><QualifierName UI="Q000601" MajorTopicYN="Y">surgery</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D009805" MajorTopicYN="N">Odontogenesis</DescriptorName><QualifierName UI="Q000502" MajorTopicYN="Y">physiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014076" MajorTopicYN="N">Tooth Diseases</DescriptorName><QualifierName UI="Q000209" MajorTopicYN="Y">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014078" MajorTopicYN="N">Tooth Eruption</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D014079" MajorTopicYN="N">Tooth Eruption, Ectopic</DescriptorName><QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014092" MajorTopicYN="N">Tooth Root</DescriptorName><QualifierName UI="Q000002" MajorTopicYN="N">abnormalities</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014094" MajorTopicYN="N">Tooth, Deciduous</DescriptorName><QualifierName UI="Q000002" MajorTopicYN="N">abnormalities</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014097" MajorTopicYN="N">Tooth, Unerupted</DescriptorName><QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D019172" MajorTopicYN="N">Transplantation Conditioning</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D014916" MajorTopicYN="N">Whole-Body Irradiation</DescriptorName></MeshHeading></MeshHeadingList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="entrez"><Year>2010</Year><Month>1</Month><Day>7</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2010</Year><Month>1</Month><Day>7</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2010</Year><Month>3</Month><Day>26</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">20051069</ArticleId><ArticleId IdType="pii">SCD115</ArticleId><ArticleId IdType="doi">10.1111/j.1754-4505.2009.00115.x</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>États-Unis</li></country><region><li>Pennsylvanie</li></region></list><tree><noCountry><name sortKey="Gupta, Avin" sort="Gupta, Avin" uniqKey="Gupta A" first="Avin" last="Gupta">Avin Gupta</name><name sortKey="Monge, Janet" sort="Monge, Janet" uniqKey="Monge J" first="Janet" last="Monge">Janet Monge</name><name sortKey="Stout, Angela M" sort="Stout, Angela M" uniqKey="Stout A" first="Angela M" last="Stout">Angela M. Stout</name></noCountry><country name="États-Unis"><region name="Pennsylvanie"><name sortKey="Wadenya, Rose O" sort="Wadenya, Rose O" uniqKey="Wadenya R" first="Rose O" last="Wadenya">Rose O. Wadenya</name></region></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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