Langerhans cell histiocytosis with oral manifestations: a rare and unusual case report
Identifieur interne : 001E11 ( Pmc/Checkpoint ); précédent : 001E10; suivant : 001E12Langerhans cell histiocytosis with oral manifestations: a rare and unusual case report
Auteurs : Bk. Yashoda-Devi ; N. Rakesh ; Manjushree AgarwalSource :
- Journal of Clinical and Experimental Dentistry [ 1989-5488 ] ; 2012.
Abstract
Langerhans cell histiocytosis (LCH), is a rare, proliferative disorder in which the accumulation of pathologic Langerhans cells leads to local tissue infiltration and destruction. We present a case of a 32 years old, completely edentulous female patient who presented with erythema of hard palate, maxillary alveolar mucosa and mucosa over the distobuccal part of mandibular alveolar ridge with foci of ulcerations. Histopathologic features were suggestive of LCH which was confirmed by immunohistochemistry which was CD1a positive, confirmatory for LCH. Bone scan revealed multiple bone involvement. At this stage, disease had already progressed to multisystem involvement with endocrinal abnormalities (primary hypothyroidism and hyperprolactinemia), requiring aggressive treatment. Therefore, this case is a reminder of the possibility of occurrence of this rare disease in the oral cavity which might manifest itself in multiple presentations thus easily leading to the misdiagnosis and therefore, it could be easily overlooked by dentists.
Url:
DOI: 10.4317/jced.50728
PubMed: 24558565
PubMed Central: 3917634
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Langerhans cell histiocytosis with oral manifestations:
a rare and unusual case report</title><author><name sortKey="Yashoda Devi, Bk" sort="Yashoda Devi, Bk" uniqKey="Yashoda Devi B" first="Bk." last="Yashoda-Devi">Bk. Yashoda-Devi</name><affiliation><nlm:aff id="A1">MDS, Professor and HOD, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author><author><name sortKey="Rakesh, N" sort="Rakesh, N" uniqKey="Rakesh N" first="N." last="Rakesh">N. Rakesh</name><affiliation><nlm:aff id="A2">MDS, Associate Professor, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author><author><name sortKey="Agarwal, Manjushree" sort="Agarwal, Manjushree" uniqKey="Agarwal M" first="Manjushree" last="Agarwal">Manjushree Agarwal</name><affiliation><nlm:aff id="A3">PG student (MDS), Department of oral medicine and radiology, M.S. Ramaiah Dental College and hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">24558565</idno><idno type="pmc">3917634</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3917634</idno><idno type="RBID">PMC:3917634</idno><idno type="doi">10.4317/jced.50728</idno><date when="2012">2012</date><idno type="wicri:Area/Pmc/Corpus">001D67</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">001D67</idno><idno type="wicri:Area/Pmc/Curation">001D67</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">001D67</idno><idno type="wicri:Area/Pmc/Checkpoint">001E11</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">001E11</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Langerhans cell histiocytosis with oral manifestations:
a rare and unusual case report</title><author><name sortKey="Yashoda Devi, Bk" sort="Yashoda Devi, Bk" uniqKey="Yashoda Devi B" first="Bk." last="Yashoda-Devi">Bk. Yashoda-Devi</name><affiliation><nlm:aff id="A1">MDS, Professor and HOD, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author><author><name sortKey="Rakesh, N" sort="Rakesh, N" uniqKey="Rakesh N" first="N." last="Rakesh">N. Rakesh</name><affiliation><nlm:aff id="A2">MDS, Associate Professor, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author><author><name sortKey="Agarwal, Manjushree" sort="Agarwal, Manjushree" uniqKey="Agarwal M" first="Manjushree" last="Agarwal">Manjushree Agarwal</name><affiliation><nlm:aff id="A3">PG student (MDS), Department of oral medicine and radiology, M.S. Ramaiah Dental College and hospital, Bangaluru</nlm:aff><wicri:noCountry code="subfield">Bangaluru</wicri:noCountry></affiliation></author></analytic><series><title level="j">Journal of Clinical and Experimental Dentistry</title><idno type="eISSN">1989-5488</idno><imprint><date when="2012">2012</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Langerhans cell histiocytosis (LCH), is a rare, proliferative disorder in which the accumulation of pathologic Langerhans cells leads to local tissue infiltration and destruction. We present a case of a 32 years old, completely edentulous female patient who presented with erythema of hard palate, maxillary alveolar mucosa and mucosa over the distobuccal part of mandibular alveolar ridge with foci of ulcerations. Histopathologic features were suggestive of LCH which was confirmed by immunohistochemistry which was CD1a positive, confirmatory for LCH. Bone scan revealed multiple bone involvement. At this stage, disease had already progressed to multisystem involvement with endocrinal abnormalities (primary hypothyroidism and hyperprolactinemia), requiring aggressive treatment. Therefore, this case is a reminder of the possibility of occurrence of this rare disease in the oral cavity which might manifest itself in multiple presentations thus easily leading to the misdiagnosis and therefore, it could be easily overlooked by dentists.
</p><p><bold> Key words:</bold>Langerhans cell histiocytosis, immunohistochemistry, bone scan.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="D Ngio, G" uniqKey="D Ngio G">G D’Angio</name></author><author><name sortKey="Favara, Be" uniqKey="Favara B">BE Favara</name></author><author><name sortKey="Ladisch, S" uniqKey="Ladisch S">S Ladisch</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hernandez Juyol, M" uniqKey="Hernandez Juyol M">M Hernandez-Juyol</name></author><author><name sortKey="Boj Quesada, Jr" uniqKey="Boj Quesada J">JR Boj-Quesada</name></author><author><name sortKey="Gallego Melcon, S" uniqKey="Gallego Melcon S">S Gallego-Melcon</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Glotzbecker, Mp" uniqKey="Glotzbecker M">MP Glotzbecker</name></author><author><name sortKey="Carpentieri, Df" uniqKey="Carpentieri D">DF Carpentieri</name></author><author><name sortKey="Dormans, Jp" uniqKey="Dormans J">JP Dormans</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Writing Group Of The Histiocyte, Society" uniqKey="Writing Group Of The Histiocyte S">Society Writing Group of the Histiocyte</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Brenner, M" uniqKey="Brenner M">M Brenner</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Nezelof, C" uniqKey="Nezelof C">C Nezelof</name></author><author><name sortKey="Basset, F" uniqKey="Basset F">F Basset</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Buchmann, L" uniqKey="Buchmann L">L Buchmann</name></author><author><name sortKey="Emami, A" uniqKey="Emami A">A Emami</name></author><author><name sortKey="Wei, Jl" uniqKey="Wei J">JL Wei</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Eckardt, A" uniqKey="Eckardt A">A Eckardt</name></author><author><name sortKey="Schultze, A" uniqKey="Schultze A">A Schultze</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dziegiel, P" uniqKey="Dziegiel P">P Dziegiel</name></author><author><name sortKey="Dolilnska Krajewska, B" uniqKey="Dolilnska Krajewska B">B Dolilnska-Krajewska</name></author><author><name sortKey="Dumanska, M" uniqKey="Dumanska M">M Dumanska</name></author><author><name sortKey="Weclawek, J" uniqKey="Weclawek J">J Weclawek</name></author><author><name sortKey="Jelen, M" uniqKey="Jelen M">M Jelen</name></author><author><name sortKey="Podhorska Okol W, M" uniqKey="Podhorska Okol W M">M Podhorska-Okolów</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Loducca, Sv" uniqKey="Loducca S">SV Loducca</name></author><author><name sortKey="Mantesso, A" uniqKey="Mantesso A">A Mantesso</name></author><author><name sortKey="Araujo, Ns" uniqKey="Araujo N">NS Araujo</name></author><author><name sortKey="Magalhaes, Mh" uniqKey="Magalhaes M">MH Magalhaes</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Malpas, Js" uniqKey="Malpas J">JS Malpas</name></author><author><name sortKey="Norton, Aj" uniqKey="Norton A">AJ Norton</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Lieberman, Ph" uniqKey="Lieberman P">PH Lieberman</name></author><author><name sortKey="Jones, Cr" uniqKey="Jones C">CR Jones</name></author><author><name sortKey="Steinman, Rm" uniqKey="Steinman R">RM Steinman</name></author><author><name sortKey="Eriandson, Ra" uniqKey="Eriandson R">RA Eriandson</name></author><author><name sortKey="Smith, J" uniqKey="Smith J">J Smith</name></author><author><name sortKey="Gee T Et, Al" uniqKey="Gee T Et A">al Gee T et</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">J Clin Exp Dent</journal-id><journal-id journal-id-type="iso-abbrev">J Clin Exp Dent</journal-id><journal-id journal-id-type="publisher-id">Medicina Oral S.L.</journal-id><journal-title-group><journal-title>Journal of Clinical and Experimental Dentistry</journal-title></journal-title-group><issn pub-type="epub">1989-5488</issn><publisher><publisher-name>Medicina Oral S.L.</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">24558565</article-id><article-id pub-id-type="pmc">3917634</article-id><article-id pub-id-type="publisher-id">50728</article-id><article-id pub-id-type="doi">10.4317/jced.50728</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject><subj-group><subject>Oral Medicine and Pathology</subject></subj-group></subj-group></article-categories><title-group><article-title>Langerhans cell histiocytosis with oral manifestations:
a rare and unusual case report</article-title></title-group><contrib-group><contrib contrib-type="author" corresp="yes"><name><surname>Yashoda-Devi</surname><given-names>BK.</given-names></name><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Rakesh</surname><given-names>N.</given-names></name><xref ref-type="aff" rid="A2">2</xref></contrib><contrib contrib-type="author"><name><surname>Agarwal</surname><given-names>Manjushree</given-names></name><xref ref-type="aff" rid="A3">3</xref></contrib></contrib-group><aff id="A1"><label>1</label>MDS, Professor and HOD, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</aff><aff id="A2"><label>2</label>MDS, Associate Professor, Department of Oral Medicine and Radiology, M S Ramaiah Dental College and Hospital, Bangaluru</aff><aff id="A3"><label>3</label>PG student (MDS), Department of oral medicine and radiology, M.S. Ramaiah Dental College and hospital, Bangaluru</aff><author-notes><corresp> Department of Oral Medicine and Radiology
M S Ramaiah Dental College and Hospital
New BEL road, M.S.R.I.T Post Bangaluru-54
, E-mail: <email>dryashodadevi@yahoo.com</email></corresp></author-notes><pub-date pub-type="epub"><day>1</day><month>10</month><year>2012</year></pub-date><pub-date pub-type="collection"><month>10</month><year>2012</year></pub-date><volume>4</volume><issue>4</issue><fpage>e252</fpage><lpage>e255</lpage><history><date date-type="accepted"><day>8</day><month>3</month><year>2012</year></date><date date-type="received"><day>29</day><month>10</month><year>2011</year></date></history><permissions><copyright-statement>Copyright: © 2012 Medicina Oral S.L.</copyright-statement><copyright-year>2012</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/2.5/"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Langerhans cell histiocytosis (LCH), is a rare, proliferative disorder in which the accumulation of pathologic Langerhans cells leads to local tissue infiltration and destruction. We present a case of a 32 years old, completely edentulous female patient who presented with erythema of hard palate, maxillary alveolar mucosa and mucosa over the distobuccal part of mandibular alveolar ridge with foci of ulcerations. Histopathologic features were suggestive of LCH which was confirmed by immunohistochemistry which was CD1a positive, confirmatory for LCH. Bone scan revealed multiple bone involvement. At this stage, disease had already progressed to multisystem involvement with endocrinal abnormalities (primary hypothyroidism and hyperprolactinemia), requiring aggressive treatment. Therefore, this case is a reminder of the possibility of occurrence of this rare disease in the oral cavity which might manifest itself in multiple presentations thus easily leading to the misdiagnosis and therefore, it could be easily overlooked by dentists.
</p><p><bold> Key words:</bold>Langerhans cell histiocytosis, immunohistochemistry, bone scan.</p></abstract></article-meta></front></pmc><affiliations><list></list><tree><noCountry><name sortKey="Agarwal, Manjushree" sort="Agarwal, Manjushree" uniqKey="Agarwal M" first="Manjushree" last="Agarwal">Manjushree Agarwal</name><name sortKey="Rakesh, N" sort="Rakesh, N" uniqKey="Rakesh N" first="N." last="Rakesh">N. Rakesh</name><name sortKey="Yashoda Devi, Bk" sort="Yashoda Devi, Bk" uniqKey="Yashoda Devi B" first="Bk." last="Yashoda-Devi">Bk. Yashoda-Devi</name></noCountry></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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