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Bone marrow immunophenotyping by flow cytometry in refractory cytopenia of childhood

Identifieur interne : 002E26 ( Pmc/Curation ); précédent : 002E25; suivant : 002E27

Bone marrow immunophenotyping by flow cytometry in refractory cytopenia of childhood

Auteurs : Anna M. Aalbers [Pays-Bas] ; Marry M. Van Den Heuvel-Eibrink [Pays-Bas] ; Irith Baumann [Allemagne] ; Michael Dworzak [Autriche] ; Henrik Hasle [Danemark] ; Franco Locatelli [Italie] ; Barbara De Moerloose [Belgique] ; Markus Schmugge [Suisse] ; Ester Mejstrikova [République tchèque] ; Michaela Nováková [République tchèque] ; Marco Zecca [Italie] ; C. Michel Zwaan [Pays-Bas] ; Jeroen G. Te Marvelde [Pays-Bas] ; Anton W. Langerak [Pays-Bas] ; Jacques J. M. Van Dongen [Pays-Bas] ; Rob Pieters [Pays-Bas] ; Charlotte M. Niemeyer [Allemagne] ; Vincent H. J. Van Der Velden [Pays-Bas]

Source :

RBID : PMC:4349269

Abstract

Refractory cytopenia of childhood is the most common type of childhood myelodysplastic syndrome. Because the majority of children with refractory cytopenia have a normal karyotype and a hypocellular bone marrow, differentiating refractory cytopenia from the immune-mediated bone marrow failure syndrome (very) severe aplastic anemia can be challenging. Flow cytometric immunophenotyping of bone marrow has been shown to be a valuable diagnostic tool in differentiating myelodysplastic syndrome from non-clonal cytopenias in adults. Here, we performed the first comprehensive flow cytometric analysis of immature myeloid, lymphoid cells and erythroid cells, and granulocytes, monocytes, and lymphoid cells in bone marrow obtained from a large prospective cohort of 81 children with refractory cytopenia. Children with refractory cyotopenia had a strongly reduced myeloid compartment, but not as severe as children with aplastic anemia. Furthermore, the number of flow cytometric abnormalities was significantly higher in children with refractory cytopenia than in healthy controls and in children with aplastic anemia, but lower than in advanced myelodysplastic syndrome. We conclude that flow cytometric immunophenotyping could be a relevant addition to histopathology in the diagnosis of refractory cytopenia of childhood. (The multi-center studies EWOG-MDS RC06 and EWOG-MDS 2006 are registered at clinicaltrials.gov identifiers 00499070 and 00662090, respectively).


Url:
DOI: 10.3324/haematol.2014.107706
PubMed: 25425683
PubMed Central: 4349269

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PMC:4349269

Le document en format XML

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<name sortKey="Mejstrikova, Ester" sort="Mejstrikova, Ester" uniqKey="Mejstrikova E" first="Ester" last="Mejstrikova">Ester Mejstrikova</name>
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<name sortKey="Van Der Velden, Vincent H J" sort="Van Der Velden, Vincent H J" uniqKey="Van Der Velden V" first="Vincent H. J." last="Van Der Velden">Vincent H. J. Van Der Velden</name>
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<nlm:aff id="af6-1000315">Department of Pediatric Hematology-Oncology, IRCCS Ospedale Bambino Gesù, Rome, University of Pavia, Italy</nlm:aff>
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<wicri:regionArea>Department of Pediatric Hematology-Oncology, IRCCS Ospedale Bambino Gesù, Rome, University of Pavia</wicri:regionArea>
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<name sortKey="De Moerloose, Barbara" sort="De Moerloose, Barbara" uniqKey="De Moerloose B" first="Barbara" last="De Moerloose">Barbara De Moerloose</name>
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<name sortKey="Mejstrikova, Ester" sort="Mejstrikova, Ester" uniqKey="Mejstrikova E" first="Ester" last="Mejstrikova">Ester Mejstrikova</name>
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<name sortKey="Novakova, Michaela" sort="Novakova, Michaela" uniqKey="Novakova M" first="Michaela" last="Nováková">Michaela Nováková</name>
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<name sortKey="Zecca, Marco" sort="Zecca, Marco" uniqKey="Zecca M" first="Marco" last="Zecca">Marco Zecca</name>
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<name sortKey="Zwaan, C Michel" sort="Zwaan, C Michel" uniqKey="Zwaan C" first="C. Michel" last="Zwaan">C. Michel Zwaan</name>
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<nlm:aff id="af2-1000315">Department of Pediatric Oncology/Hematology, Sophia Children’s Hospital - Erasmus University Medical Center, Rotterdam, The Netherlands</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Pediatric Oncology/Hematology, Sophia Children’s Hospital - Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<name sortKey="Te Marvelde, Jeroen G" sort="Te Marvelde, Jeroen G" uniqKey="Te Marvelde J" first="Jeroen G." last="Te Marvelde">Jeroen G. Te Marvelde</name>
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<nlm:aff id="af1-1000315">Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam, The Netherlands</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<name sortKey="Langerak, Anton W" sort="Langerak, Anton W" uniqKey="Langerak A" first="Anton W." last="Langerak">Anton W. Langerak</name>
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<wicri:regionArea>Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<name sortKey="Van Dongen, Jacques J M" sort="Van Dongen, Jacques J M" uniqKey="Van Dongen J" first="Jacques J. M." last="Van Dongen">Jacques J. M. Van Dongen</name>
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<nlm:aff id="af1-1000315">Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam, The Netherlands</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<name sortKey="Pieters, Rob" sort="Pieters, Rob" uniqKey="Pieters R" first="Rob" last="Pieters">Rob Pieters</name>
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<nlm:aff id="af2-1000315">Department of Pediatric Oncology/Hematology, Sophia Children’s Hospital - Erasmus University Medical Center, Rotterdam, The Netherlands</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Pediatric Oncology/Hematology, Sophia Children’s Hospital - Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<name sortKey="Niemeyer, Charlotte M" sort="Niemeyer, Charlotte M" uniqKey="Niemeyer C" first="Charlotte M." last="Niemeyer">Charlotte M. Niemeyer</name>
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<nlm:aff id="af11-1000315">Department of Pediatrics and Adolescent Medicine, Division of Pediatric Hematology and Oncology, University of Freiburg, Germany</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Pediatrics and Adolescent Medicine, Division of Pediatric Hematology and Oncology, University of Freiburg</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Van Der Velden, Vincent H J" sort="Van Der Velden, Vincent H J" uniqKey="Van Der Velden V" first="Vincent H. J." last="Van Der Velden">Vincent H. J. Van Der Velden</name>
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<nlm:aff id="af1-1000315">Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam, The Netherlands</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam</wicri:regionArea>
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<title level="j">Haematologica</title>
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<front>
<div type="abstract" xml:lang="en">
<p>Refractory cytopenia of childhood is the most common type of childhood myelodysplastic syndrome. Because the majority of children with refractory cytopenia have a normal karyotype and a hypocellular bone marrow, differentiating refractory cytopenia from the immune-mediated bone marrow failure syndrome (very) severe aplastic anemia can be challenging. Flow cytometric immunophenotyping of bone marrow has been shown to be a valuable diagnostic tool in differentiating myelodysplastic syndrome from non-clonal cytopenias in adults. Here, we performed the first comprehensive flow cytometric analysis of immature myeloid, lymphoid cells and erythroid cells, and granulocytes, monocytes, and lymphoid cells in bone marrow obtained from a large prospective cohort of 81 children with refractory cytopenia. Children with refractory cyotopenia had a strongly reduced myeloid compartment, but not as severe as children with aplastic anemia. Furthermore, the number of flow cytometric abnormalities was significantly higher in children with refractory cytopenia than in healthy controls and in children with aplastic anemia, but lower than in advanced myelodysplastic syndrome. We conclude that flow cytometric immunophenotyping could be a relevant addition to histopathology in the diagnosis of refractory cytopenia of childhood. (The multi-center studies EWOG-MDS RC06 and EWOG-MDS 2006 are registered at
<italic>
<ext-link ext-link-type="uri" xlink:href="clinicaltrials.gov">clinicaltrials.gov</ext-link>
identifiers 00499070</italic>
and
<italic>00662090</italic>
, respectively).</p>
</div>
</front>
</TEI>
<pmc article-type="research-article">
<pmc-comment>The publisher of this article does not allow downloading of the full text in XML form.</pmc-comment>
<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">Haematologica</journal-id>
<journal-id journal-id-type="iso-abbrev">Haematologica</journal-id>
<journal-id journal-id-type="hwp">haematol</journal-id>
<journal-id journal-id-type="publisher-id">Haematologica</journal-id>
<journal-title-group>
<journal-title>Haematologica</journal-title>
</journal-title-group>
<issn pub-type="ppub">0390-6078</issn>
<issn pub-type="epub">1592-8721</issn>
<publisher>
<publisher-name>Ferrata Storti Foundation</publisher-name>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="pmid">25425683</article-id>
<article-id pub-id-type="pmc">4349269</article-id>
<article-id pub-id-type="doi">10.3324/haematol.2014.107706</article-id>
<article-id pub-id-type="publisher-id">1000315</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Articles</subject>
<subj-group subj-group-type="heading">
<subject>Myelodysplastic Syndromes</subject>
</subj-group>
</subj-group>
</article-categories>
<title-group>
<article-title>Bone marrow immunophenotyping by flow cytometry in refractory cytopenia of childhood</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name>
<surname>Aalbers</surname>
<given-names>Anna M.</given-names>
</name>
<xref ref-type="aff" rid="af1-1000315">1</xref>
<xref ref-type="aff" rid="af2-1000315">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>van den Heuvel-Eibrink</surname>
<given-names>Marry M.</given-names>
</name>
<xref ref-type="aff" rid="af2-1000315">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Baumann</surname>
<given-names>Irith</given-names>
</name>
<xref ref-type="aff" rid="af3-1000315">3</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Dworzak</surname>
<given-names>Michael</given-names>
</name>
<xref ref-type="aff" rid="af4-1000315">4</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Hasle</surname>
<given-names>Henrik</given-names>
</name>
<xref ref-type="aff" rid="af5-1000315">5</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Locatelli</surname>
<given-names>Franco</given-names>
</name>
<xref ref-type="aff" rid="af6-1000315">6</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>De Moerloose</surname>
<given-names>Barbara</given-names>
</name>
<xref ref-type="aff" rid="af7-1000315">7</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Schmugge</surname>
<given-names>Markus</given-names>
</name>
<xref ref-type="aff" rid="af8-1000315">8</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Mejstrikova</surname>
<given-names>Ester</given-names>
</name>
<xref ref-type="aff" rid="af9-1000315">9</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Nováková</surname>
<given-names>Michaela</given-names>
</name>
<xref ref-type="aff" rid="af9-1000315">9</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Zecca</surname>
<given-names>Marco</given-names>
</name>
<xref ref-type="aff" rid="af10-1000315">10</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Zwaan</surname>
<given-names>C. Michel</given-names>
</name>
<xref ref-type="aff" rid="af2-1000315">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>te Marvelde</surname>
<given-names>Jeroen G.</given-names>
</name>
<xref ref-type="aff" rid="af1-1000315">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Langerak</surname>
<given-names>Anton W.</given-names>
</name>
<xref ref-type="aff" rid="af1-1000315">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>van Dongen</surname>
<given-names>Jacques J.M.</given-names>
</name>
<xref ref-type="aff" rid="af1-1000315">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Pieters</surname>
<given-names>Rob</given-names>
</name>
<xref ref-type="aff" rid="af2-1000315">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Niemeyer</surname>
<given-names>Charlotte M.</given-names>
</name>
<xref ref-type="aff" rid="af11-1000315">11</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>van der Velden</surname>
<given-names>Vincent H.J.</given-names>
</name>
<xref ref-type="aff" rid="af1-1000315">1</xref>
<xref ref-type="corresp" rid="c1-1000315"></xref>
</contrib>
</contrib-group>
<aff id="af1-1000315">
<label>1</label>
Department of Immunology, Erasmus MC, Erasmus University Medical Center, Rotterdam, The Netherlands</aff>
<aff id="af2-1000315">
<label>2</label>
Department of Pediatric Oncology/Hematology, Sophia Children’s Hospital - Erasmus University Medical Center, Rotterdam, The Netherlands</aff>
<aff id="af3-1000315">
<label>3</label>
Department of Pathology, Clinical Centre South West, Böblingen Clinics, Germany</aff>
<aff id="af4-1000315">
<label>4</label>
St. Anna Children’s Hospital and Children’s Cancer Research Institute, Department of Pediatrics, Medical University of Vienna, Austria</aff>
<aff id="af5-1000315">
<label>5</label>
Department of Pediatrics, Aarhus University Hospital Skejby, Aarhus, Denmark</aff>
<aff id="af6-1000315">
<label>6</label>
Department of Pediatric Hematology-Oncology, IRCCS Ospedale Bambino Gesù, Rome, University of Pavia, Italy</aff>
<aff id="af7-1000315">
<label>7</label>
Department of Pediatric Hematology/Oncology, Ghent University Hospital, Ghent, Belgium</aff>
<aff id="af8-1000315">
<label>8</label>
Department of Hematology, University Children’s Hospital, Zurich, Switzerland</aff>
<aff id="af9-1000315">
<label>9</label>
Department of Pediatric Hematology/Oncology, Charles University and University Hospital Motol, Prague, Czech Republic</aff>
<aff id="af10-1000315">
<label>10</label>
Pediatric Hematology, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy</aff>
<aff id="af11-1000315">
<label>11</label>
Department of Pediatrics and Adolescent Medicine, Division of Pediatric Hematology and Oncology, University of Freiburg, Germany</aff>
<author-notes>
<corresp id="c1-1000315">Correspondence:
<email>v.h.j.vandervelden@erasmusmc.nl</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>3</month>
<year>2015</year>
</pub-date>
<volume>100</volume>
<issue>3</issue>
<fpage>315</fpage>
<lpage>323</lpage>
<history>
<date date-type="received">
<day>23</day>
<month>3</month>
<year>2014</year>
</date>
<date date-type="accepted">
<day>13</day>
<month>11</month>
<year>2014</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright© Ferrata Storti Foundation</copyright-statement>
<copyright-year>2015</copyright-year>
</permissions>
<self-uri content-type="pdf" xlink:type="simple" xlink:href="100315.pdf"></self-uri>
<abstract>
<p>Refractory cytopenia of childhood is the most common type of childhood myelodysplastic syndrome. Because the majority of children with refractory cytopenia have a normal karyotype and a hypocellular bone marrow, differentiating refractory cytopenia from the immune-mediated bone marrow failure syndrome (very) severe aplastic anemia can be challenging. Flow cytometric immunophenotyping of bone marrow has been shown to be a valuable diagnostic tool in differentiating myelodysplastic syndrome from non-clonal cytopenias in adults. Here, we performed the first comprehensive flow cytometric analysis of immature myeloid, lymphoid cells and erythroid cells, and granulocytes, monocytes, and lymphoid cells in bone marrow obtained from a large prospective cohort of 81 children with refractory cytopenia. Children with refractory cyotopenia had a strongly reduced myeloid compartment, but not as severe as children with aplastic anemia. Furthermore, the number of flow cytometric abnormalities was significantly higher in children with refractory cytopenia than in healthy controls and in children with aplastic anemia, but lower than in advanced myelodysplastic syndrome. We conclude that flow cytometric immunophenotyping could be a relevant addition to histopathology in the diagnosis of refractory cytopenia of childhood. (The multi-center studies EWOG-MDS RC06 and EWOG-MDS 2006 are registered at
<italic>
<ext-link ext-link-type="uri" xlink:href="clinicaltrials.gov">clinicaltrials.gov</ext-link>
identifiers 00499070</italic>
and
<italic>00662090</italic>
, respectively).</p>
</abstract>
</article-meta>
</front>
</pmc>
</record>

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