Solitary langerhans cell histiocytosis in an adult: case report and literature review
Identifieur interne : 000504 ( Main/Exploration ); précédent : 000503; suivant : 000505Solitary langerhans cell histiocytosis in an adult: case report and literature review
Auteurs : Cíntia Ferreira Gonçalves ; Marília Oliveira Morais ; Rita De Cássia Gonçalves Alencar ; Aline Carvalho Batista ; Elismauro Francisco MendonçaSource :
- BMC Research Notes [ 1756-0500 ] ; 2016.
Abstract
Langerhans cell histiocytosis (LCH) is a disease that often affects children, but can also occur in adults and smokers. Oral manifestations are unusual and are characterized by bone pain, tooth mobility, necrotic ulcers and local edema. The aim of this paper is to describe a clinical case of LCH in an oral cavity that mimicked oral squamous cell carcinoma.
A male, 63 years old, complaining about a “wound in the mouth” for 6 months, without any pain or spontaneous bleeding. His medical history was free of disease. The patient was a smoker for 33 years. Intraoral examination revealed a destructive ulcerative lesion around the upper left first and second molars that resembled an oral squamous cell carcinoma. Biopsy of the ulcerative lesion was performed and the microscopic features showed an inflammatory infiltrate rich in plasma cells. Based on this microscopical finding, the final diagnosis was periodontal disease associated with a proliferative non-neoplastic lesion. The patient was referred to a specialized dental surgeon and underwent periodontal therapy including surgical procedures. After that, according to follow-up with the patient, there were no signs of disease remission. The lesion increased in size, although the patient did not complain of any symptoms. A second biopsy was performed and the microscopic features again showed a rich inflammatory infiltrate with mononuclear cells and histiocytic cells, characterized by pale histiocytes with lobed nuclei, resembling a bean. A varying number of eosinophils also were observed, without any evidence of atypical cells present in this infiltrate. An immunohistochemical staining panel was done to determine the nature of this inflammatory infiltrate by using antibodies S-100, CD1a, CD-68 and CD45RO that were positive. These immunohistochemical findings were fundamental for the final diagnosis of LCH. The treatment included surgical extraction of all superior teeth, radiation and systemic corticoid therapies. After 8 years of treatment, the patient is free of disease.
Although LCH is an unusual lesion in an oral cavity, it can be present. Biopsy and a histological exam are essential to establish the diagnosis. Immunohistochemicals were fundamental to exclude malignant lesion and to confirm the diagnosis of LCH.
Url:
DOI: 10.1186/s13104-015-1799-z
PubMed: 26749317
PubMed Central: 4706661
Affiliations:
Links toward previous steps (curation, corpus...)
- to stream Pmc, to step Corpus: 000959
- to stream Pmc, to step Curation: 000959
- to stream Pmc, to step Checkpoint: 000447
- to stream Ncbi, to step Merge: 006283
- to stream Ncbi, to step Curation: 006283
- to stream Ncbi, to step Checkpoint: 006283
- to stream Main, to step Merge: 000504
- to stream Main, to step Curation: 000504
Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Solitary langerhans cell histiocytosis in an adult: case report and literature review</title><author><name sortKey="Goncalves, Cintia Ferreira" sort="Goncalves, Cintia Ferreira" uniqKey="Goncalves C" first="Cíntia Ferreira" last="Gonçalves">Cíntia Ferreira Gonçalves</name><affiliation><nlm:aff id="Aff1">Avenida Teotônio Segurado, Cj. 01, Lt. 01, Sl 508, Plano Diretor Sul, Palmas, Tocantins CEP 77061-002 Brazil</nlm:aff><wicri:noCountry code="subfield">Tocantins CEP 77061-002 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Morais, Marilia Oliveira" sort="Morais, Marilia Oliveira" uniqKey="Morais M" first="Marília Oliveira" last="Morais">Marília Oliveira Morais</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="De Cassia Goncalves Alencar, Rita" sort="De Cassia Goncalves Alencar, Rita" uniqKey="De Cassia Goncalves Alencar R" first="Rita" last="De Cássia Gonçalves Alencar">Rita De Cássia Gonçalves Alencar</name><affiliation><nlm:aff id="Aff3">Hospital Araújo Jorge, Goiânia, Goiás Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Batista, Aline Carvalho" sort="Batista, Aline Carvalho" uniqKey="Batista A" first="Aline Carvalho" last="Batista">Aline Carvalho Batista</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Mendonca, Elismauro Francisco" sort="Mendonca, Elismauro Francisco" uniqKey="Mendonca E" first="Elismauro Francisco" last="Mendonça">Elismauro Francisco Mendonça</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">26749317</idno><idno type="pmc">4706661</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4706661</idno><idno type="RBID">PMC:4706661</idno><idno type="doi">10.1186/s13104-015-1799-z</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">000959</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000959</idno><idno type="wicri:Area/Pmc/Curation">000959</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">000959</idno><idno type="wicri:Area/Pmc/Checkpoint">000447</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">000447</idno><idno type="wicri:Area/Ncbi/Merge">006283</idno><idno type="wicri:Area/Ncbi/Curation">006283</idno><idno type="wicri:Area/Ncbi/Checkpoint">006283</idno><idno type="wicri:Area/Main/Merge">000504</idno><idno type="wicri:Area/Main/Curation">000504</idno><idno type="wicri:Area/Main/Exploration">000504</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Solitary langerhans cell histiocytosis in an adult: case report and literature review</title><author><name sortKey="Goncalves, Cintia Ferreira" sort="Goncalves, Cintia Ferreira" uniqKey="Goncalves C" first="Cíntia Ferreira" last="Gonçalves">Cíntia Ferreira Gonçalves</name><affiliation><nlm:aff id="Aff1">Avenida Teotônio Segurado, Cj. 01, Lt. 01, Sl 508, Plano Diretor Sul, Palmas, Tocantins CEP 77061-002 Brazil</nlm:aff><wicri:noCountry code="subfield">Tocantins CEP 77061-002 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Morais, Marilia Oliveira" sort="Morais, Marilia Oliveira" uniqKey="Morais M" first="Marília Oliveira" last="Morais">Marília Oliveira Morais</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="De Cassia Goncalves Alencar, Rita" sort="De Cassia Goncalves Alencar, Rita" uniqKey="De Cassia Goncalves Alencar R" first="Rita" last="De Cássia Gonçalves Alencar">Rita De Cássia Gonçalves Alencar</name><affiliation><nlm:aff id="Aff3">Hospital Araújo Jorge, Goiânia, Goiás Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Batista, Aline Carvalho" sort="Batista, Aline Carvalho" uniqKey="Batista A" first="Aline Carvalho" last="Batista">Aline Carvalho Batista</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author><author><name sortKey="Mendonca, Elismauro Francisco" sort="Mendonca, Elismauro Francisco" uniqKey="Mendonca E" first="Elismauro Francisco" last="Mendonça">Elismauro Francisco Mendonça</name><affiliation><nlm:aff id="Aff2">Faculdade de Odontologia, Praça Universitária S/N, Setor Universitário, Universidade Federal de Goiás, Goiânia, Goiás CEP 74605-220 Brazil</nlm:aff><wicri:noCountry code="subfield">Goiás CEP 74605-220 Brazil</wicri:noCountry></affiliation></author></analytic><series><title level="j">BMC Research Notes</title><idno type="eISSN">1756-0500</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>Background</title><p>Langerhans cell histiocytosis (LCH) is a disease that often affects children, but can also occur in adults and smokers. Oral manifestations are unusual and are characterized by bone pain, tooth mobility, necrotic ulcers and local edema. The aim of this paper is to describe a clinical case of LCH in an oral cavity that mimicked oral squamous cell carcinoma.</p></sec><sec><title>Case presentation</title><p>A male, 63 years old, complaining about a “wound in the mouth” for 6 months, without any pain or spontaneous bleeding. His medical history was free of disease. The patient was a smoker for 33 years. Intraoral examination revealed a destructive ulcerative lesion around the upper left first and second molars that resembled an oral squamous cell carcinoma. Biopsy of the ulcerative lesion was performed and the microscopic features showed an inflammatory infiltrate rich in plasma cells. Based on this microscopical finding, the final diagnosis was periodontal disease associated with a proliferative non-neoplastic lesion. The patient was referred to a specialized dental surgeon and underwent periodontal therapy including surgical procedures. After that, according to follow-up with the patient, there were no signs of disease remission. The lesion increased in size, although the patient did not complain of any symptoms. A second biopsy was performed and the microscopic features again showed a rich inflammatory infiltrate with mononuclear cells and histiocytic cells, characterized by pale histiocytes with lobed nuclei, resembling a bean. A varying number of eosinophils also were observed, without any evidence of atypical cells present in this infiltrate. An immunohistochemical staining panel was done to determine the nature of this inflammatory infiltrate by using antibodies S-100, CD1a, CD-68 and CD45RO that were positive. These immunohistochemical findings were fundamental for the final diagnosis of LCH. The treatment included surgical extraction of all superior teeth, radiation and systemic corticoid therapies. After 8 years of treatment, the patient is free of disease.</p></sec><sec><title>Conclusion</title><p>Although LCH is an unusual lesion in an oral cavity, it can be present. Biopsy and a histological exam are essential to establish the diagnosis. Immunohistochemicals were fundamental to exclude malignant lesion and to confirm the diagnosis of LCH.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Arceci, Rj" uniqKey="Arceci R">RJ Arceci</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Satter, Ek" uniqKey="Satter E">EK Satter</name></author><author><name sortKey="High, Wa" uniqKey="High W">WA High</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Aric, M" uniqKey="Aric M">M Aricò</name></author><author><name sortKey="Girschikofsky, M" uniqKey="Girschikofsky M">M Girschikofsky</name></author><author><name sortKey="Genereau, T" uniqKey="Genereau T">T Généreau</name></author><author><name sortKey="Klersy, C" uniqKey="Klersy C">C Klersy</name></author><author><name sortKey="Mcclain, K" uniqKey="Mcclain K">K McClain</name></author><author><name sortKey="Grois, N" uniqKey="Grois N">N Grois</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Morimoto, A" uniqKey="Morimoto A">A Morimoto</name></author><author><name sortKey="Oh, Y" uniqKey="Oh Y">Y Oh</name></author><author><name sortKey="Shioda, Y" uniqKey="Shioda Y">Y Shioda</name></author><author><name sortKey="Kudo, K" uniqKey="Kudo K">K Kudo</name></author><author><name sortKey="Imamura, T" uniqKey="Imamura T">T Imamura</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Howarth, Dm" uniqKey="Howarth D">DM Howarth</name></author><author><name sortKey="Gilchrist, Gs" uniqKey="Gilchrist G">GS Gilchrist</name></author><author><name sortKey="Mullan, Bp" uniqKey="Mullan B">BP Mullan</name></author><author><name sortKey="Wiseman, Ga" uniqKey="Wiseman G">GA Wiseman</name></author><author><name sortKey="Edmonson, Jh" uniqKey="Edmonson J">JH Edmonson</name></author><author><name sortKey="Schomberg, Pj" uniqKey="Schomberg P">PJ Schomberg</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Routy, B" uniqKey="Routy B">B Routy</name></author><author><name sortKey="Hoang, J" uniqKey="Hoang J">J Hoang</name></author><author><name sortKey="Gruber, J" uniqKey="Gruber J">J Gruber</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Preliasco, Vf" uniqKey="Preliasco V">VF Preliasco</name></author><author><name sortKey="Benchuya, C" uniqKey="Benchuya C">C Benchuya</name></author><author><name sortKey="Pavan, V" uniqKey="Pavan V">V Pavan</name></author><author><name sortKey="De La Cal, C" uniqKey="De La Cal C">C de la Cal</name></author><author><name sortKey="Ganzinelli, S" uniqKey="Ganzinelli S">S Ganzinelli</name></author><author><name sortKey="Sterin Borda, L" uniqKey="Sterin Borda L">L Sterin-Borda</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Doz, E" uniqKey="Doz E">E Doz</name></author><author><name sortKey="Noulin, N" uniqKey="Noulin N">N Noulin</name></author><author><name sortKey="Boichot, E" uniqKey="Boichot E">E Boichot</name></author><author><name sortKey="Guenon, I" uniqKey="Guenon I">I Guenon</name></author><author><name sortKey="Fick, L" uniqKey="Fick L">L Fick</name></author><author><name sortKey="Le Bert, M" uniqKey="Le Bert M">M Le Bert</name></author><author><name sortKey="Lagente, V" uniqKey="Lagente V">V Lagente</name></author><author><name sortKey="Ryffel, B" uniqKey="Ryffel B">B Ryffel</name></author><author><name sortKey="Schnyder, B" uniqKey="Schnyder B">B Schnyder</name></author><author><name sortKey="Quesniaux, Vf" uniqKey="Quesniaux V">VF Quesniaux</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Cantu, Ma" uniqKey="Cantu M">MA Cantu</name></author><author><name sortKey="Lupo, Pj" uniqKey="Lupo P">PJ Lupo</name></author><author><name sortKey="Bilgi, M" uniqKey="Bilgi M">M Bilgi</name></author><author><name sortKey="Hicks, Mj" uniqKey="Hicks M">MJ Hicks</name></author><author><name sortKey="Allen, Ce" uniqKey="Allen C">CE Allen</name></author><author><name sortKey="Mcclain, Kl" uniqKey="Mcclain K">KL McClain</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kaltsas, Ga" uniqKey="Kaltsas G">GA Kaltsas</name></author><author><name sortKey="Powles, Tb" uniqKey="Powles T">TB Powles</name></author><author><name sortKey="Evanson, J" uniqKey="Evanson J">J Evanson</name></author><author><name sortKey="Plowman, Pn" uniqKey="Plowman P">PN Plowman</name></author><author><name sortKey="Drinkwater, Je" uniqKey="Drinkwater J">JE Drinkwater</name></author><author><name sortKey="Jenkins, Pj" uniqKey="Jenkins P">PJ Jenkins</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ma, J" uniqKey="Ma J">J Ma</name></author><author><name sortKey="Jiang, Y" uniqKey="Jiang Y">Y Jiang</name></author><author><name sortKey="Chen, X" uniqKey="Chen X">X Chen</name></author><author><name sortKey="Gong, G" uniqKey="Gong G">G Gong</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ardekian, L" uniqKey="Ardekian L">L Ardekian</name></author><author><name sortKey="Peled, M" uniqKey="Peled M">M Peled</name></author><author><name sortKey="Rosen, D" uniqKey="Rosen D">D Rosen</name></author><author><name sortKey="Rachmiel, A" uniqKey="Rachmiel A">A Rachmiel</name></author><author><name sortKey="Abu El Naaj, I" uniqKey="Abu El Naaj I">I Abu el-Naaj</name></author><author><name sortKey="Laufer, D" uniqKey="Laufer D">D Laufer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hicks, J" uniqKey="Hicks J">J Hicks</name></author><author><name sortKey="Flaitz, Cm" uniqKey="Flaitz C">CM Flaitz</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Aric, M" uniqKey="Aric M">M Aricò</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gadner, H" uniqKey="Gadner H">H Gadner</name></author><author><name sortKey="Grois, N" uniqKey="Grois N">N Grois</name></author><author><name sortKey="Arico, M" uniqKey="Arico M">M Arico</name></author><author><name sortKey="Broadbent, V" uniqKey="Broadbent V">V Broadbent</name></author><author><name sortKey="Ceci, A" uniqKey="Ceci A">A Ceci</name></author><author><name sortKey="Jakobson, A" uniqKey="Jakobson A">A Jakobson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ben Slama, L" uniqKey="Ben Slama L">L Ben Slama</name></author><author><name sortKey="Ruhin, B" uniqKey="Ruhin B">B Ruhin</name></author><author><name sortKey="Zoghbani, A" uniqKey="Zoghbani A">A Zoghbani</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Minkov, M" uniqKey="Minkov M">M Minkov</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Grana, N" uniqKey="Grana N">N Grana</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Murakami, I" uniqKey="Murakami I">I Murakami</name></author><author><name sortKey="Matsushita, M" uniqKey="Matsushita M">M Matsushita</name></author><author><name sortKey="Iwasaki, T" uniqKey="Iwasaki T">T Iwasaki</name></author><author><name sortKey="Kuwamoto, S" uniqKey="Kuwamoto S">S Kuwamoto</name></author><author><name sortKey="Kato, M" uniqKey="Kato M">M Kato</name></author><author><name sortKey="Nagata, K" uniqKey="Nagata K">K Nagata</name></author><author><name sortKey="Horie, Y" uniqKey="Horie Y">Y Horie</name></author><author><name sortKey="Hayashi, K" uniqKey="Hayashi K">K Hayashi</name></author><author><name sortKey="Imamura, T" uniqKey="Imamura T">T Imamura</name></author><author><name sortKey="Morimoto, A" uniqKey="Morimoto A">A Morimoto</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list></list><tree><noCountry><name sortKey="Batista, Aline Carvalho" sort="Batista, Aline Carvalho" uniqKey="Batista A" first="Aline Carvalho" last="Batista">Aline Carvalho Batista</name><name sortKey="De Cassia Goncalves Alencar, Rita" sort="De Cassia Goncalves Alencar, Rita" uniqKey="De Cassia Goncalves Alencar R" first="Rita" last="De Cássia Gonçalves Alencar">Rita De Cássia Gonçalves Alencar</name><name sortKey="Goncalves, Cintia Ferreira" sort="Goncalves, Cintia Ferreira" uniqKey="Goncalves C" first="Cíntia Ferreira" last="Gonçalves">Cíntia Ferreira Gonçalves</name><name sortKey="Mendonca, Elismauro Francisco" sort="Mendonca, Elismauro Francisco" uniqKey="Mendonca E" first="Elismauro Francisco" last="Mendonça">Elismauro Francisco Mendonça</name><name sortKey="Morais, Marilia Oliveira" sort="Morais, Marilia Oliveira" uniqKey="Morais M" first="Marília Oliveira" last="Morais">Marília Oliveira Morais</name></noCountry></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
EXPLOR_STEP=$WICRI_ROOT/Wicri/Santé/explor/EdenteV2/Data/Main/Exploration
HfdSelect -h $EXPLOR_STEP/biblio.hfd -nk 000504 | SxmlIndent | more
Ou
HfdSelect -h $EXPLOR_AREA/Data/Main/Exploration/biblio.hfd -nk 000504 | SxmlIndent | more
Pour mettre un lien sur cette page dans le réseau Wicri
{{Explor lien
|wiki= Wicri/Santé
|area= EdenteV2
|flux= Main
|étape= Exploration
|type= RBID
|clé= PMC:4706661
|texte= Solitary langerhans cell histiocytosis in an adult: case report and literature review
}}
Pour générer des pages wiki
HfdIndexSelect -h $EXPLOR_AREA/Data/Main/Exploration/RBID.i -Sk "pubmed:26749317" \
| HfdSelect -Kh $EXPLOR_AREA/Data/Main/Exploration/biblio.hfd \
| NlmPubMed2Wicri -a EdenteV2
| This area was generated with Dilib version V0.6.32. |  |