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Novel treatment of blastic plasmacytoid dendritic cell neoplasm: A case report.

Abstract Blastic plasmacytoid dendritic cell neoplasm (BPDCN), derived from precursors of plasmacytoid dendritic cells, is a rare and aggressive malignancy with frequent cutaneous involvement. Although cutaneous lesions are often chemosensitive, BPDCN portends a poor prognosis as most patients relapse after developing drug resistance.
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Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start




PMID- 29390581
OWN - NLM
STAT- MEDLINE
DCOM- 20180212
LR  - 20180212
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 51
DP  - 2017 Dec
TI  - Novel treatment of blastic plasmacytoid dendritic cell neoplasm: A case report.
PG  - e9452
LID - 10.1097/MD.0000000000009452 [doi]
AB  - RATIONALE: Blastic plasmacytoid dendritic cell neoplasm (BPDCN), derived from
      precursors of plasmacytoid dendritic cells, is a rare and aggressive malignancy
      with frequent cutaneous involvement. Although cutaneous lesions are often
      chemosensitive, BPDCN portends a poor prognosis as most patients relapse after
      developing drug resistance. PATIENT CONCERNS: We report a case of a 65-year-old
      man who presented with a rapidly enlarging hyperpigmented plaque on his shoulder 
      with subsequent similarly appearing macules and plaques on his chest, back, and
      neck. DIAGNOSIS: Skin biopsy revealed a dense adnexocentric dermal infiltrate of 
      immature blastoid cells without epidermal involvement. The infiltrate was
      immunoreactive for CD4, CD56, CD123, and Bcl-2, but negative for CD3, CD8, CD30, 
      MPO, EBER, and ISH. The patient was diagnosed with BPDCN based on these cell
      markers. INTERVENTION: Bone marrow biopsy and radiologic work-up showed no
      evidence of extracutaneous involvement. The patient attained partial remission
      after undergoing 2 rounds of cyclophosphamide, doxorubicin, vincristine, and
      prednisone (CHOP regimen) before autologous stem cell transplantation, however,
      he quickly relapsed and developed new cutaneous lesions. OUTCOMES: The patient
      was treated with venetoclax, a Bcl-2 inhibitor, and exhibits complete resolution 
      of prior skin findings and continues to remain free of new cutaneous lesions 10
      months posttreatment initiation with venetoclax. LESSONS: Herein, we present a
      case that supports the use of venetoclax, a Bcl-2 inhibitor, in the off-label
      treatment of BPDCN with Bcl-2 overexpression. Only 1 prior case has reported the 
      off-label use of venetoclax for the treatment of BPDCN. This case highlights a
      novel therapeutic option for BPDCN patients unresponsive to traditional
      treatment.
CI  - Copyright (c) 2017 The Authors. Published by Wolters Kluwer Health, Inc. All
      rights reserved.
FAU - Grushchak, Solomiya
AU  - Grushchak S
FAU - Joy, Cossette
AU  - Joy C
FAU - Gray, Arielle
AU  - Gray A
FAU - Opel, Dan
AU  - Opel D
FAU - Speiser, Jodi
AU  - Speiser J
FAU - Reserva, Jeave
AU  - Reserva J
FAU - Tung, Rebecca
AU  - Tung R
FAU - Smith, Scott E
AU  - Smith SE
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 0 (CD4 Antigens)
RN  - 0 (CD56 Antigen)
SB  - AIM
SB  - IM
MH  - Aged
MH  - CD4 Antigens/metabolism
MH  - CD56 Antigen/metabolism
MH  - Dendritic Cells/*pathology
MH  - Humans
MH  - Male
MH  - Skin/cytology/pathology
MH  - Skin Neoplasms/*diagnosis/pathology
PMC - PMC5758283
EDAT- 2018/02/03 06:00
MHDA- 2018/02/13 06:00
CRDT- 2018/02/03 06:00
PHST- 2018/02/03 06:00 [entrez]
PHST- 2018/02/03 06:00 [pubmed]
PHST- 2018/02/13 06:00 [medline]
AID - 10.1097/MD.0000000000009452 [doi]
AID - 00005792-201712220-00168 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Dec;96(51):e9452. doi: 10.1097/MD.0000000000009452.