Progression of a CD4+/CD56+ blastic plasmacytoid DC neoplasm after initiation of extracorporeal photopheresis in an allogeneic transplant recipient

Dohm, Andrea; Hasenkamp, Justin; Bertsch, Hans Peter; Maas, Jens-Holger; Truemper, Lorenz; Wulf, Gerald

doi:10.1038/bmt.2010.196

Cited by 5 publications

(3 citation statements)

References 13 publications

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“…The potential role of maintenance therapy warrants investigation in BPDCN. Table 1 summarizes outcomes using various conventional chemotherapy regimens that include at least 4 patients [21][22][23][24][25][26][27][28][29][30][31][32][33][34].…”

Section: Conventional Therapymentioning

confidence: 99%

Diagnostic and Therapeutic Advances in Blastic Plasmacytoid Dendritic Cell Neoplasm: A Focus on Hematopoietic Cell Transplantation

Kharfan‐Dabaja

Lazarus

Nishihori

et al. 2013

Biology of Blood and Marrow Transplantation

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Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is an exceedingly rare disorder categorized under acute myeloid leukemia by the World Health Organization. Phenotypically, malignant cells coexpress CD4(+) and CD56(+) without coexpressing common lymphoid or myeloid lineage markers. BPDCN frequently expresses CD123, TCL1, BDCA-2, and CD2AP. Restriction of CD2AP expression to plasmacytoid dendritic cells makes it a useful tool to help confirm diagnosis. Clonal complex chromosome aberrations are described in two-thirds of cases. Eighty percent of BPDCN cases present with nonspecific dermatological manifestations, prompting inclusion in the differential diagnosis of atypical skin rashes refractory to standard treatment. Prognosis is poor, with a median survival of less than 18 months. No prospective randomized data exist to define the most optimal frontline chemotherapy. Current practice considers acute myeloid leukemia-like or acute lymphoblastic leukemia-like regimens acceptable for induction treatment. Unfortunately, responses are short-lived, with second remissions difficult to achieve, underscoring the need to consider hematopoietic cell transplantation early in the disease course. Allografting, especially if offered in first remission, can result in long-term remissions. Preclinical data suggest a potential role for immunomodulatory agents in BPCDN. However, further research efforts are needed to better understand BPDCN biology and to establish evidence-based treatment algorithms that might ultimately improve overall prognosis of this disease.

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Section: Conventional Therapymentioning

confidence: 99%

Diagnostic and Therapeutic Advances in Blastic Plasmacytoid Dendritic Cell Neoplasm: A Focus on Hematopoietic Cell Transplantation

Kharfan‐Dabaja

Lazarus

Nishihori

et al. 2013

Biology of Blood and Marrow Transplantation

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“…One month later, fulminant leukemia, treated with etoposide combined with fludarabine/cyclophosphamide and dexamethasoneNo14DODDalle et al 4 . 2010/9M/37NSLeg/noduleLomustine, doxorubicin, cytarabine-PR3/ifosfamide, etoposide, cytarabine, MTXAllo-SCT54DCDM/70Cheek/noduleCytarabine, idarubicin, lomustine-PDPD/Mini-CHANo19DCDM/73Shoulder/noduleRT-PDPD/Idarubicin, cytarabine, lomustineNo36DCDM/75Arm/noduleRefused treatmentNSNo12DCDM/83Cheek/noduleRT-CR8/Refused treatmentNo8AliveF/25Leg/tumorHyper-CVAD-CR6/Autologous BMTAutologous BMT38AliveM/82Scalp/tumorRT-CR9/EtoposideNo19DCDM/78Leg/tumorCHOP-CR12/NSNo12AliveF/60Cheek/noduleMTX, asparaginase, RT-CRNSNo11LTFDohm et al 5 . 2011/1M/32NSArm/nodule/6 × 4CHOEP-14 × 6, +RT (36 Gy) – CR11/First relapse: 4 cycles of ifosfamide, carboplatin and etoposide + RT (36 Gy): Second relapse: 4 cycles of fludarabine and cyclophosphamide.…”

Section: Discussionmentioning

confidence: 99%

“…Only 19 patients with LS-BPDCN have been reported in the literature since the latest classification of BPDCN in 2008, 3 , 4 , 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 (their data are summarized in Table I ). Median age of the reported patients at presentation was 46 years (range, 5-83), which is younger than the median age documented for all patients with BPDCN (67.8 years).…”

Section: Discussionmentioning

confidence: 99%

Localized skin-limited blastic plasmacytoid dendritic cell neoplasm: A subset with possible durable remission without transplantation

et al. 2017

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Ulcerating skin lesions from blastic plasmacytoid dendritic cell neoplasm responding to low-dose radiotherapy—a case report and literature review

Hoffmann,

Böke,

De-Colle

et al. 2024

Strahlenther Onkol

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Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare hematologic malignancy that can manifest with skin nodules and erythematous plaques. In most cases BPDCN progresses rapidly, causing multiple skin lesions and also affecting internal organs and bone marrow, warranting initiation of systemic therapies or hematopoietic stem cell transplantation (HCT). Although not curative, radiotherapy for isolated lesions might be indicated in case of (imminent) ulceration and large or symptomatic lesions. To this end, doses of 27.0–51.0 Gy have been reported. Here, we present the case of an 80-year-old male with BPDCN with multiple large, nodular, and ulcerating lesions of the thorax, abdomen, and face. Low-dose radiotherapy of 2 × 4.0 Gy was administered to several lesions, which resolved completely within 1 week with only light residual hyperpigmentation of the skin in affected areas and reliably prevented further ulceration. Radiotoxicity was not reported. Therefore, low-dose radiotherapy can be an effective and low-key treatment in selected cases of BPDCN, especially in a palliative setting, with a favorable toxicity profile.

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Progression of a CD4+/CD56+ blastic plasmacytoid DC neoplasm after initiation of extracorporeal photopheresis in an allogeneic transplant recipient

Cited by 5 publications

References 13 publications

Diagnostic and Therapeutic Advances in Blastic Plasmacytoid Dendritic Cell Neoplasm: A Focus on Hematopoietic Cell Transplantation

Diagnostic and Therapeutic Advances in Blastic Plasmacytoid Dendritic Cell Neoplasm: A Focus on Hematopoietic Cell Transplantation

Localized skin-limited blastic plasmacytoid dendritic cell neoplasm: A subset with possible durable remission without transplantation

Ulcerating skin lesions from blastic plasmacytoid dendritic cell neoplasm responding to low-dose radiotherapy—a case report and literature review

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