Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion

B-cell prolymphocytic leukemia (B-PLL) is a rare lymphoid neoplasm with an aggressive clinical course. Treatment strategies for B-PLL remain to be established, and, until recently, alemtuzumab was the only effective therapeutic option in patients harboring 17p deletions. Herein, we describe, for the...

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Main Authors: H. Coelho, M. Badior, T. Melo
Format: Article
Language:English
Published: Wiley 2017-01-01
Series:Case Reports in Hematology
Online Access:http://dx.doi.org/10.1155/2017/8563218
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author H. Coelho
M. Badior
T. Melo
author_facet H. Coelho
M. Badior
T. Melo
author_sort H. Coelho
collection DOAJ
description B-cell prolymphocytic leukemia (B-PLL) is a rare lymphoid neoplasm with an aggressive clinical course. Treatment strategies for B-PLL remain to be established, and, until recently, alemtuzumab was the only effective therapeutic option in patients harboring 17p deletions. Herein, we describe, for the first time, a case of B-cell prolymphocytic leukemia harboring a 17p deletion in a 48-year-old man that was successfully treated sequentially with idelalisib-rituximab/ibrutinib followed by allogeneic hematopoietic stem cell transplant (allo-HSCT). After 5 months of therapy with idelalisib-rituximab, clinical remission was achieved, but the development of severe diarrhea led to its discontinuation. Subsequently, the patient was treated for 2 months with ibrutinib and the quality of the response was maintained with no severe adverse effects reported. A reduced-intensity conditioning allo-HSCT from a HLA-matched unrelated donor was performed, and, thereafter, the patient has been in complete remission for 10 months now. In conclusion, given the poor prognosis of B-PLL and the lack of effective treatment modalities, the findings here suggest that both ibrutinib and idelalisib should be considered as upfront therapy of B-PLL and as a bridge to allo-HSCT.
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spelling doaj-art-af76b9007836441fac3022e6f8a8eef02025-02-03T06:42:01ZengWileyCase Reports in Hematology2090-65602090-65792017-01-01201710.1155/2017/85632188563218Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p DeletionH. Coelho0M. Badior1T. Melo2Serviço de Hematologia, Centro Hospitalar de Vila Nova de Gaia/Espinho, Vila Nova Gaia, PortugalServiço de Hematologia, Centro Hospitalar de Vila Nova de Gaia/Espinho, Vila Nova Gaia, PortugalServiço de Hematologia, Centro Hospitalar de Vila Nova de Gaia/Espinho, Vila Nova Gaia, PortugalB-cell prolymphocytic leukemia (B-PLL) is a rare lymphoid neoplasm with an aggressive clinical course. Treatment strategies for B-PLL remain to be established, and, until recently, alemtuzumab was the only effective therapeutic option in patients harboring 17p deletions. Herein, we describe, for the first time, a case of B-cell prolymphocytic leukemia harboring a 17p deletion in a 48-year-old man that was successfully treated sequentially with idelalisib-rituximab/ibrutinib followed by allogeneic hematopoietic stem cell transplant (allo-HSCT). After 5 months of therapy with idelalisib-rituximab, clinical remission was achieved, but the development of severe diarrhea led to its discontinuation. Subsequently, the patient was treated for 2 months with ibrutinib and the quality of the response was maintained with no severe adverse effects reported. A reduced-intensity conditioning allo-HSCT from a HLA-matched unrelated donor was performed, and, thereafter, the patient has been in complete remission for 10 months now. In conclusion, given the poor prognosis of B-PLL and the lack of effective treatment modalities, the findings here suggest that both ibrutinib and idelalisib should be considered as upfront therapy of B-PLL and as a bridge to allo-HSCT.http://dx.doi.org/10.1155/2017/8563218
spellingShingle H. Coelho
M. Badior
T. Melo
Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
Case Reports in Hematology
title Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
title_full Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
title_fullStr Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
title_full_unstemmed Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
title_short Sequential Kinase Inhibition (Idelalisib/Ibrutinib) Induces Clinical Remission in B-Cell Prolymphocytic Leukemia Harboring a 17p Deletion
title_sort sequential kinase inhibition idelalisib ibrutinib induces clinical remission in b cell prolymphocytic leukemia harboring a 17p deletion
url http://dx.doi.org/10.1155/2017/8563218
work_keys_str_mv AT hcoelho sequentialkinaseinhibitionidelalisibibrutinibinducesclinicalremissioninbcellprolymphocyticleukemiaharboringa17pdeletion
AT mbadior sequentialkinaseinhibitionidelalisibibrutinibinducesclinicalremissioninbcellprolymphocyticleukemiaharboringa17pdeletion
AT tmelo sequentialkinaseinhibitionidelalisibibrutinibinducesclinicalremissioninbcellprolymphocyticleukemiaharboringa17pdeletion