AustralieFrV1, Main, Exploration, bibRecord, 008F00

ALK+ histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy

Identifieur interne : 008F00 ( Main/Exploration ); précédent : 008E99; suivant : 008F01

ALK+ histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy

Auteurs : John K. C. Chan [Hong Kong] ; Laurence Lamant [France] ; Elizabeth Algar [Australie] ; Georges Delsol [France] ; William Y. W. Tsang [Hong Kong] ; King C. Lee [Hong Kong] ; Karin Tiedemann [Australie] ; Chung W. Chow [Australie]

Source :

Blood [ 0006-4971 ] ; 2008.

RBID : Pascal:09-0005466

Descripteurs français

Pascal (Inist)
- Histiocytose, Maladie autoimmune, Translocation chromosomique, Enfant, Nourrisson, Gène ALK.
Wicri :
- topic : Enfant.

English descriptors

KwdEn :
- Autoimmune disease, Child, Chromosome translocation, Histiocytosis, Infant.

Abstract

We report 3 cases of a previously uncharacterized form of histiocytosis presenting in early infancy and showing ALK immunoreactivity. The patients presented with pallor, massive hepatosplenomegaly, anemia, and thrombocytopenia. Liver biopsy showed infiltration of the sinusoids by large histiocytes with markedly folded nuclei, fine chromatin, small nucleoli, and voluminous lightly eosinophilic cytoplasm that sometimes was vacuolated or contained phagocytosed blood cells. One patient developed cutaneous infiltrates that morphologically resembled juvenile xanthogranuloma. The histiocytes were immunoreactive for histiocytic markers (CD68, CD163, lysozyme), S100 protein, ALK (membranous and cytoplasmic pattern), and dendritic cell markers (fascin, factor Xllla), but not CD1a and langerin. One case successfully analyzed by molecular techniques revealed TPM3-ALK fusion. Thus the spectrum of diseases exhibiting ALK translocation should be expanded to include ALK⁺ histiocytosis. The disease in the 3 patients (2 having been given chemotherapy) resolved slowly over many months.

Affiliations:

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Le document en format XML

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 histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy</title>
<author><name sortKey="Chan, John K C" sort="Chan, John K C" uniqKey="Chan J" first="John K. C." last="Chan">John K. C. Chan</name>
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<author><name sortKey="Tsang, William Y W" sort="Tsang, William Y W" uniqKey="Tsang W" first="William Y. W." last="Tsang">William Y. W. Tsang</name>
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<author><name sortKey="Tiedemann, Karin" sort="Tiedemann, Karin" uniqKey="Tiedemann K" first="Karin" last="Tiedemann">Karin Tiedemann</name>
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<author><name sortKey="Chow, Chung W" sort="Chow, Chung W" uniqKey="Chow C" first="Chung W." last="Chow">Chung W. Chow</name>
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<s3>AUS</s3>
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<sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">ALK<sup>+</sup>
 histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy</title>
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<wicri:noRegion>Kowloon</wicri:noRegion>
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<s2>Toulouse</s2>
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<author><name sortKey="Delsol, Georges" sort="Delsol, Georges" uniqKey="Delsol G" first="Georges" last="Delsol">Georges Delsol</name>
<affiliation wicri:level="3"><inist:fA14 i1="02"><s1>Inserm, U563, Centre de Physiopathologie de Toulouse Purpan, and Université Toulouse III Paul Sabatier</s1>
<s2>Toulouse</s2>
<s3>FRA</s3>
<sZ>2 aut.</sZ>
<sZ>4 aut.</sZ>
</inist:fA14>
<country>France</country>
<placeName><region type="region">Occitanie (région administrative)</region>
<region type="old region">Midi-Pyrénées</region>
<settlement type="city">Toulouse</settlement>
</placeName>
</affiliation>
</author>
<author><name sortKey="Tsang, William Y W" sort="Tsang, William Y W" uniqKey="Tsang W" first="William Y. W." last="Tsang">William Y. W. Tsang</name>
<affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Department of Pathology, Queen Elizabeth Hospital</s1>
<s2>Kowloon</s2>
<s3>HKG</s3>
<sZ>1 aut.</sZ>
<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
</inist:fA14>
<country>Hong Kong</country>
<wicri:noRegion>Kowloon</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Lee, King C" sort="Lee, King C" uniqKey="Lee K" first="King C." last="Lee">King C. Lee</name>
<affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Department of Pathology, Queen Elizabeth Hospital</s1>
<s2>Kowloon</s2>
<s3>HKG</s3>
<sZ>1 aut.</sZ>
<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
</inist:fA14>
<country>Hong Kong</country>
<wicri:noRegion>Kowloon</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Tiedemann, Karin" sort="Tiedemann, Karin" uniqKey="Tiedemann K" first="Karin" last="Tiedemann">Karin Tiedemann</name>
<affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Children's Cancer Center, Royal Children's Hospital</s1>
<s2>Melbourne</s2>
<s3>AUS</s3>
<sZ>3 aut.</sZ>
<sZ>7 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<placeName><settlement type="city">Melbourne</settlement>
<region type="état">Victoria (État)</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Chow, Chung W" sort="Chow, Chung W" uniqKey="Chow C" first="Chung W." last="Chow">Chung W. Chow</name>
<affiliation wicri:level="4"><inist:fA14 i1="03"><s1>Department of Pediatrics, University of Melbourne</s1>
<s3>AUS</s3>
<sZ>3 aut.</sZ>
<sZ>8 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<placeName><settlement type="city">Melbourne</settlement>
<region type="état">Victoria (État)</region>
</placeName>
<orgName type="university">Université de Melbourne</orgName>
</affiliation>
<affiliation wicri:level="3"><inist:fA14 i1="05"><s1>Department of Anatomical Pathology, Royal Children's Hospital</s1>
<s2>Melbourne</s2>
<s3>AUS</s3>
<sZ>8 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<placeName><settlement type="city">Melbourne</settlement>
<region type="état">Victoria (État)</region>
</placeName>
</affiliation>
</author>
</analytic>
<series><title level="j" type="main">Blood</title>
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<idno type="ISSN">0006-4971</idno>
<imprint><date when="2008">2008</date>
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<seriesStmt><title level="j" type="main">Blood</title>
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</fileDesc>
<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Autoimmune disease</term>
<term>Child</term>
<term>Chromosome translocation</term>
<term>Histiocytosis</term>
<term>Infant</term>
</keywords>
<keywords scheme="Pascal" xml:lang="fr"><term>Histiocytose</term>
<term>Maladie autoimmune</term>
<term>Translocation chromosomique</term>
<term>Enfant</term>
<term>Nourrisson</term>
<term>Gène ALK</term>
</keywords>
<keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Enfant</term>
</keywords>
</textClass>
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</teiHeader>
<front><div type="abstract" xml:lang="en">We report 3 cases of a previously uncharacterized form of histiocytosis presenting in early infancy and showing ALK immunoreactivity. The patients presented with pallor, massive hepatosplenomegaly, anemia, and thrombocytopenia. Liver biopsy showed infiltration of the sinusoids by large histiocytes with markedly folded nuclei, fine chromatin, small nucleoli, and voluminous lightly eosinophilic cytoplasm that sometimes was vacuolated or contained phagocytosed blood cells. One patient developed cutaneous infiltrates that morphologically resembled juvenile xanthogranuloma. The histiocytes were immunoreactive for histiocytic markers (CD68, CD163, lysozyme), S100 protein, ALK (membranous and cytoplasmic pattern), and dendritic cell markers (fascin, factor Xllla), but not CD1a and langerin. One case successfully analyzed by molecular techniques revealed TPM3-ALK fusion. Thus the spectrum of diseases exhibiting ALK translocation should be expanded to include ALK<sup>+</sup>
 histiocytosis. The disease in the 3 patients (2 having been given chemotherapy) resolved slowly over many months.</div>
</front>
</TEI>
<affiliations><list><country><li>Australie</li>
<li>France</li>
<li>Hong Kong</li>
</country>
<region><li>Midi-Pyrénées</li>
<li>Occitanie (région administrative)</li>
<li>Victoria (État)</li>
</region>
<settlement><li>Melbourne</li>
<li>Toulouse</li>
</settlement>
<orgName><li>Université de Melbourne</li>
</orgName>
</list>
<tree><country name="Hong Kong"><noRegion><name sortKey="Chan, John K C" sort="Chan, John K C" uniqKey="Chan J" first="John K. C." last="Chan">John K. C. Chan</name>
</noRegion>
<name sortKey="Lee, King C" sort="Lee, King C" uniqKey="Lee K" first="King C." last="Lee">King C. Lee</name>
<name sortKey="Tsang, William Y W" sort="Tsang, William Y W" uniqKey="Tsang W" first="William Y. W." last="Tsang">William Y. W. Tsang</name>
</country>
<country name="France"><region name="Occitanie (région administrative)"><name sortKey="Lamant, Laurence" sort="Lamant, Laurence" uniqKey="Lamant L" first="Laurence" last="Lamant">Laurence Lamant</name>
</region>
<name sortKey="Delsol, Georges" sort="Delsol, Georges" uniqKey="Delsol G" first="Georges" last="Delsol">Georges Delsol</name>
</country>
<country name="Australie"><region name="Victoria (État)"><name sortKey="Algar, Elizabeth" sort="Algar, Elizabeth" uniqKey="Algar E" first="Elizabeth" last="Algar">Elizabeth Algar</name>
</region>
<name sortKey="Algar, Elizabeth" sort="Algar, Elizabeth" uniqKey="Algar E" first="Elizabeth" last="Algar">Elizabeth Algar</name>
<name sortKey="Chow, Chung W" sort="Chow, Chung W" uniqKey="Chow C" first="Chung W." last="Chow">Chung W. Chow</name>
<name sortKey="Chow, Chung W" sort="Chow, Chung W" uniqKey="Chow C" first="Chung W." last="Chow">Chung W. Chow</name>
<name sortKey="Tiedemann, Karin" sort="Tiedemann, Karin" uniqKey="Tiedemann K" first="Karin" last="Tiedemann">Karin Tiedemann</name>
</country>
</tree>
</affiliations>
</record>

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ALK+ histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy

ALK+ histiocytosis : a novel type of systemic histiocytic proliferative disorder of early infancy

Source :

Descripteurs français

English descriptors

Abstract

Links toward previous steps (curation, corpus...)

Le document en format XML

Pour manipuler ce document sous Unix (Dilib)

Pour mettre un lien sur cette page dans le réseau Wicri