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  1. Article ; Online: Reply to Zhou et al. 'A novel approach for characterization of KSHV-associated multicentric Castleman disease from effusions'.

    Vanjak, Anthony / Garzaro, Margaux / Knapp, Silene / Silvestrini, Marc-Antoine / Martin de Fremont, Gregoire / Bertinchamp, Remi / Calvani, Julien / Meignin, Véronique / Dossier, Antoine / Galicier, Lionel / Oksenhendler, Eric / Boutboul, David

    British journal of haematology

    2023  Volume 201, Issue 2, Page(s) 357–359

    MeSH term(s) Humans ; Castleman Disease/diagnosis ; Castleman Disease/complications ; Sarcoma, Kaposi
    Language English
    Publishing date 2023-02-14
    Publishing country England
    Document type Letter
    ZDB-ID 80077-6
    ISSN 1365-2141 ; 0007-1048
    ISSN (online) 1365-2141
    ISSN 0007-1048
    DOI 10.1111/bjh.18698
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Uh III Zs.182: Show issues Location:
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  2. Article ; Online: Paraneoplastic pemphigus uncovers distinct clinical and biological phenotypes of western unicentric Castleman disease.

    Dieudonné, Yannick / Silvestrini, Marc-Antoine / Dossier, Antoine / Meignin, Véronique / Jouenne, Fanélie / Mahévas, Thibault / Bouaziz, Jean-David / Jackson, Margaret A / Mordant, Pierre / Poirot, Justine / Onodi, Fanny / Calvani, Julien / Hourseau, Muriel / Evrard, Diane / Berisha, Mirlinda / Perrin, François / Danel, Claire / Borie, Raphael / Galicier, Lionel /
    Mourah, Samia / Bengoufa, Djaouida / Oksenhendler, Eric / Grootenboer-Mignot, Sabine / Boutboul, David

    British journal of haematology

    2023  Volume 202, Issue 2, Page(s) 267–278

    Abstract: Unicentric Castleman disease (UCD) is a lymphoproliferative disease of unknown cause. Paraneoplastic pemphigus (PNP) is a major complication shown to be associated with a poor prognosis, with particular severity in patients with bronchiolitis obliterans ( ...

    Abstract Unicentric Castleman disease (UCD) is a lymphoproliferative disease of unknown cause. Paraneoplastic pemphigus (PNP) is a major complication shown to be associated with a poor prognosis, with particular severity in patients with bronchiolitis obliterans (BO). This study describes the clinical and biological characteristics of UCD-PNP patients in a large Western cohort. A total of 148 patients diagnosed with UCD were identified, including 14 patients with a defined PNP. PNP was significantly associated with myasthenia gravis (MG) and FDC sarcoma during follow-up (FDCS). PNP was also significantly associated with reduced survival. These data, together with a multivariate analysis by principal components, led to the identification of UCD-PNP as a group at risk of MG, FDCS and death. PDGFRB sequencing performed on UCD lesions from six patients found the gain-of-function p.N666S variant in two. Interestingly, both patients had hyaline-vascular UCD subtype, were in the UCD-PNP subgroup and had FDCS. Sera from 25 UCD-PNP patients and 6 PNP patients without UCD were tested for PNP-associated autoantibodies. Sera from UCD-PNP patients had a strong reactivity against the N-terminal domain of recombinant periplakin (rPPL, 82%) and showed reactivity against at least two domains of rPPL. These features were not found in patients with UCD alone or in the PNP group without UCD. These data indicate that UCD-PNP patients belong to a subgroup sharing strong clinical and biological identity that might help to decipher the different dynamics of UCD natural history.
    MeSH term(s) Humans ; Pemphigus/diagnosis ; Pemphigus/etiology ; Castleman Disease/pathology ; Autoantibodies ; Myasthenia Gravis/diagnosis ; Paraneoplastic Syndromes/etiology ; Paraneoplastic Syndromes/diagnosis
    Chemical Substances Autoantibodies
    Language English
    Publishing date 2023-05-23
    Publishing country England
    Document type Journal Article ; Research Support, Non-U.S. Gov't
    ZDB-ID 80077-6
    ISSN 1365-2141 ; 0007-1048
    ISSN (online) 1365-2141
    ISSN 0007-1048
    DOI 10.1111/bjh.18847
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

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  3. Article ; Online: Characteristics of circulating KSHV-infected viroblasts during active KSHV+ multicentric Castleman disease.

    Martin de Frémont, Gregoire / Vanjak, Anthony / Sbihi, Zineb / Knapp, Silene / Garzaro, Margaux / Chbihi, Marwa / Fournier, Benjamin / Poirot, Justine / Dossier, Antoine / Silvestrini, Marc-Antoine / Villemonteix, Juliette / Meignin, Véronique / Galicier, Lionel / Bertinchamp, Rémi / Le Goff, Jerome / Salmona, Maud / Flamarion, Edouard / Cassius, Charles / Lebbé, Celeste /
    Ronchetti, Anne Marie / Latour, Sylvain / Oksenhendler, Eric / Carcelain, Guislaine / Boutboul, David

    Blood advances

    2022  Volume 7, Issue 9, Page(s) 1682–1691

    Abstract: Kaposi sarcoma-associated herpesvirus (KSHV)/human herpesvirus 8-associated multicentric Castleman disease (MCD) is a polyclonal B-cell lymphoproliferative disorder that mainly occurs in immunocompromised hosts. The diagnosis relies on lymph node biopsy ... ...

    Abstract Kaposi sarcoma-associated herpesvirus (KSHV)/human herpesvirus 8-associated multicentric Castleman disease (MCD) is a polyclonal B-cell lymphoproliferative disorder that mainly occurs in immunocompromised hosts. The diagnosis relies on lymph node biopsy demonstrating KSHV-infected cells located in the mantle zone with a marked interfollicular plasma cell infiltration. Infected cells are large cells positive for immunoglobulin M (IgM), λ light chain, and CD38, described initially as infected plasmablasts. We show that IgM+λ+CD38high cells were also detectable in the peripheral blood of 14 out of 18 (78%) patients with active KSHV-MCD and absent in 40 controls. Using immunofluorescence and flow-fluorescence in situ hybridization, we demonstrate that these cells are KSHV infected and express both latent and lytic KSHV transcripts. These KSHV-infected viroblasts (KIVs) harbor a distinct phenotype compared with conventional plasmablasts. We also identified several putative mechanisms of immune escape used by KSHV, because KIVs displayed an overall decrease of costimulatory molecules, with a remarkable lack of CD40 expression and are interleukin-10-producing cells. The identification of this specific and easily accessible KSHV+ circulating population brings new elements to the understanding of KSHV-MCD but also raises new questions that need to be clarified.
    MeSH term(s) Humans ; Herpesvirus 8, Human/genetics ; Herpesvirus 8, Human/metabolism ; Castleman Disease/complications ; In Situ Hybridization, Fluorescence ; Immunoglobulin M
    Chemical Substances Immunoglobulin M
    Language English
    Publishing date 2022-12-12
    Publishing country United States
    Document type Journal Article ; Research Support, Non-U.S. Gov't
    ZDB-ID 2915908-8
    ISSN 2473-9537 ; 2473-9529
    ISSN (online) 2473-9537
    ISSN 2473-9529
    DOI 10.1182/bloodadvances.2022008456
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    Zs.A 7153: Show issues Location:
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