Blastic plasmacytoid dendritic cell neoplasm: is transplantation the treatment of choice?
Summary Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with...
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| Published in | British journal of dermatology (1951) Vol. 162; no. 1; pp. 74 - 79 |
|---|---|
| Main Authors | , , , , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
Oxford, UK
Blackwell Publishing Ltd
01.01.2010
Wiley-Blackwell |
| Subjects | |
| Online Access | Get full text |
| ISSN | 0007-0963 1365-2133 1365-2133 |
| DOI | 10.1111/j.1365-2133.2009.09373.x |
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| Abstract | Summary
Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor.
Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008.
Methods Forty‐seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded.
Results The mean survival was 16·7 months (95% confidence interval 12·6–20·8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher.
Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients’ prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome. |
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| AbstractList | Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor. Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008. Methods Forty-seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded. Results The mean survival was 16.7 months (95% confidence interval 12.6-20.8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher. Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients' prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome. SummaryBackground Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor.Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008.Methods Forty-seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded.Results The mean survival was 16.7 months (95% confidence interval 12.6-20.8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher.Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients' prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome. Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor. Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008. Methods Forty-seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded. Results The mean survival was 16.7 months (95% confidence interval 12.6-20.8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher. Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients' prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome.Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor. Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008. Methods Forty-seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded. Results The mean survival was 16.7 months (95% confidence interval 12.6-20.8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher. Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients' prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome. Summary Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare entity is usually revealed and diagnosed on cutaneous lesions associated or not with a leukaemic component. The prognosis associated with BPDCN is very poor. Objectives To perform a retrospective review of BPDCN cases registered in the French Study Group on Cutaneous Lymphoma database from June 1995 to May 2008. Methods Forty‐seven patients were included. Demographic data, initial staging, therapeutic management and outcome were recorded. Results The mean survival was 16·7 months (95% confidence interval 12·6–20·8). Only eight (17%) and one (2%) patients reached respectively 2 and 5 years of survival. Initial spreading of the disease did not represent, in this cohort, a reliable prognosis factor. The outcome was overall influenced by treatment provided. While radiation therapy, monochemotherapy or even polychemotherapy regimens did not significantly affect the course of the disease, the survival of bone marrow transplanted patients was significantly higher. Conclusions Despite the fact that BPDCN is often initially limited to the skin, only an aggressive initial therapy may improve the patients’ prognosis. Local treatments, such radiation therapy, are definitively useless. Regardless of the initial extension of the disease, in our experience only bone marrow transplantation significantly improved the outcome. |
| Author | Beylot-Barry, M. Dereure, O. Fraitag, S. Machet, L. Dompmartin, A. Grange, F. Maubec, E. Petrella, T. Joly, P. Dalle, S. Bagot, M. D'Incan, M. Barete, S. Lipsker, D. Prey, S. Wetterwald, M. |
| Author_xml | – sequence: 1 givenname: S. surname: Dalle fullname: Dalle, S. organization: Department of Dermatology, Université Claude Bernard Lyon 1, Hospices Civils de Lyon, INSERM U590, Lyon, France – sequence: 2 givenname: M. surname: Beylot-Barry fullname: Beylot-Barry, M. organization: Department of Dermatology, Hôpital du Haut-Lévêque, CHU de Bordeaux, Pessac, France – sequence: 3 givenname: M. surname: Bagot fullname: Bagot, M. organization: Department of Dermatology, Assistance Publique-Hôpitaux de Paris, INSERM U976, Créteil, France – sequence: 4 givenname: D. surname: Lipsker fullname: Lipsker, D. organization: Faculté de Médecine, Clinique Dermatologique, Hôpitaux Universitaires de Strasbourg, Université de Strasbourg, Strasbourg, France – sequence: 5 givenname: L. surname: Machet fullname: Machet, L. organization: Department of Dermatology, Hôpital Trousseau, CHRU de Tours, Université François-Rabelais, Tours, France – sequence: 6 givenname: P. surname: Joly fullname: Joly, P. organization: Department of Dermatology, INSERM U901, University of Rouen, Rouen, France – sequence: 7 givenname: A. surname: Dompmartin fullname: Dompmartin, A. organization: Department of Dermatology, Université de Caen Basse Normandie, Centre Hospitalier Universitaire Caen, Caen, France – sequence: 8 givenname: M. surname: D'Incan fullname: D'Incan, M. organization: Department of Dermatology, Université Clermont-Ferrand 1, CHU Clermont-Ferrand, Clermont-Ferrand, France – sequence: 9 givenname: E. surname: Maubec fullname: Maubec, E. organization: Department of Dermatology, Hôpital Bichat, Assistance Publique-Hôpitaux de Paris, Université Paris-VII, Paris, France – sequence: 10 givenname: F. surname: Grange fullname: Grange, F. organization: Department of Dermatology, Hôpital Robert Debré, Reims, France – sequence: 11 givenname: O. surname: Dereure fullname: Dereure, O. organization: Department of Dermatology, University of Montpellier, Montpellier, France – sequence: 12 givenname: S. surname: Prey fullname: Prey, S. organization: Department of Dermatology, Paul Sabatier University and Purpan Hospital, Toulouse, France – sequence: 13 givenname: S. surname: Barete fullname: Barete, S. organization: Department of Dermatology and Allergy, Hôpital Tenon, Assistance Publique-Hôpitaux de Paris, France – sequence: 14 givenname: M. surname: Wetterwald fullname: Wetterwald, M. organization: Hematology Department, Centre Hospitalier de Dunkerque, Dunkerque, France – sequence: 15 givenname: S. surname: Fraitag fullname: Fraitag, S. organization: Hôpital Necker-Enfants-Malades, Assistance Publique-Hôpitaux de Paris, Université René-Descartes, Paris, France – sequence: 16 givenname: T. surname: Petrella fullname: Petrella, T. organization: Department of Pathology, Dijon University Hospital and Centre de Pathologie Dijon, Dijon, France |
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| Keywords | Dendritic cell multicentric Dermatology Leukemia Homograft Malignant hemopathy Lymphoma Treatment Lymphoproliferative syndrome Graft Tumor leukaemia Skin blastic plasmacytoid dendritic cell neoplasm retrospective study Cancer cutaneous lymphoma |
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| Notes | ark:/67375/WNG-T3MSNKRX-M ArticleID:BJD9373 istex:EF304436555B8720921AF630FE7B5B7426822B52 Conflicts of interest S.D., M.B.‐B., M.B., L.M., P.J., A.D., M.d’I., E.M., F.G., O.D., S.B., S.F. and T.P. are members of the French Study Group on Cutaneous Lymphoma. None declared. ObjectType-Article-2 SourceType-Scholarly Journals-1 ObjectType-Feature-1 content type line 23 ObjectType-Article-1 ObjectType-Feature-2 |
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| References_xml | – reference: Kadowaki N, Antonenko S, Lau JY, Liu YJ. Natural interferon alpha/beta-producing cells link innate and adaptive immunity. J Exp Med 2000; 192:219-26. – reference: Huguet F, Leguay T, Raffoux E et al. Pediatric-inspired therapy in adults with Philadelphia chromosome-negative acute lymphoblastic leukemia: the GRAALL-2003 study. J Clin Oncol 2009; 27:911-18. – reference: Petrella T, Wechsler J, Courville P et al. [Hematodermic CD4/CD56 neoplasm.]. Ann Pathol 2004; 24:241-55. – reference: Ito T, Amakawa R, Inaba M et al. Plasmacytoid dendritic cells regulate Th cell responses through OX40 ligand and type I IFNs. J Immunol 2004; 172:4253-9. – reference: Villadangos JA, Young L. Antigen-presentation properties of plasmacytoid dendritic cells. Immunity 2008; 29:352-61. – reference: Willemze R, Jaffe ES, Burg G et al. WHO-EORTC classification for cutaneous lymphomas. Blood 2005; 105:3768-85. – reference: Urosevic M, Conrad C, Kamarashev J et al. 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4 article-title: Primary cutaneous CD4+/CD56+ hematodermic neoplasm (blastic NK‐cell lymphoma): a report of five cases publication-title: Haematologica – volume: 104 start-page: 2467 year: 2004 end-page: 74 article-title: Randomized comparison of double induction and timed‐sequential induction to a ‘3 + 7’ induction in adults with AML: long‐term analysis of the Acute Leukemia French Association (ALFA) 9000 study publication-title: Blood – volume: 109 start-page: 1720 year: 2007 end-page: 7 article-title: Gene‐expression profiling and array‐based CGH classify CD4+CD56+ hematodermic neoplasm and cutaneous myelomonocytic leukemia as distinct disease entities publication-title: Blood |
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Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This... Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This rare... SummaryBackground Blastic plasmacytoid dendritic cell neoplasm (BPDCN) represents the malignant counterpart derived from plasmacytoid dendritic cells. This... |
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| SubjectTerms | Adult Aged Aged, 80 and over Antineoplastic Agents - therapeutic use Biological and medical sciences blastic plasmacytoid dendritic cell neoplasm Bone Marrow Transplantation Child Cohort Studies cutaneous lymphoma Dendritic Cells Dermatology Female Hematologic and hematopoietic diseases Humans leukaemia Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis lymphoma Lymphoma - drug therapy Lymphoma - mortality Lymphoma - radiotherapy Lymphoma - surgery Male Medical sciences Middle Aged multicentric Prognosis Retrospective Studies retrospective study Skin Neoplasms - drug therapy Skin Neoplasms - mortality Skin Neoplasms - radiotherapy Skin Neoplasms - surgery Survival Analysis |
| Title | Blastic plasmacytoid dendritic cell neoplasm: is transplantation the treatment of choice? |
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