Spontaneous Regression of Blastic Plasmacytoid Dendritic Cell Neoplasm Following Sepsis by &lt;i&gt;Serratia marcescens&lt;/i&gt;: A Case Report and Literature Review

Suzuki, Akane; Abe, Shunsuke; Koyama, Kaori; Suzuki, Shinju; Nagao, Munenori; Kobayashi, Masahiro; Nomura, Jun; Tsutsumi, Tomomi; Takeda, Tomoki; Oka, Yasu; Shirota, Yuko; Takasawa, Naruhiko; Kodera, Takao; Okitsu, Yoko; Takahashi, Shinichiro; Ichinohasama, Ryo; Kameoka, Junichi

doi:10.2169/internalmedicine.5820-20

Cited by 3 publications

(6 citation statements)

References 30 publications

(37 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…(3) patients usually achieve at least partial remission in the skin, but similar bone marrow responses are unusual [16,[21][22][23][24]. Here, we present a case of SR of BPDCN, probably one of the few cases related to tissue infection.…”

Section: Discussionmentioning

confidence: 85%

See 1 more Smart Citation

Spontaneous Remission of Blastic Plasmacytoid Dendritic Cell Neoplasm: A Case Report

Castaño-Bonilla,

Mata,

Láinez-González

et al. 2024

Medicina

View full text Add to dashboard Cite

Spontaneous remissions (SRs) in blastic plasmacytoid dendritic cell neoplasms (BPDCNs) are infrequent, poorly documented, and transient. We report a 40-year-old man presenting with bycitopenia and soft tissue infection. The bone marrow exhibited 3% abnormal cells. Immunophenotyping of these cells revealed the antigens CD45+ (dim), CD34+, CD117+, CD123+ (bright), HLA-DR+ (bimodal), CD56+ (bright), CD33+, CD13+, CD2+, and CD22+ (dim) and the partial expression of the CD10+, CD36+, and CD7+ antigens. All other myeloid, monocytic, and lymphoid antigens were negative. Genetic studies showed a complex karyotype and mutations in the TP53R337C and KRASG12D genes. On hospital admission, the patient showed a subcutaneous nodule on the right hand and left lower limb. Flow cytometry multiparameter (FCM) analysis showed the presence of 29% abnormal cells with the previously described immunophenotype. The patient was diagnosed with BPDCN. The patient was treated with broad-spectrum antibiotics for soft tissue infection, which delayed therapy for BPDCN. No steroids or chemotherapeutic or hypomethylating agents were administered. His blood cell counts improved and skin lesions disappeared, until the patient relapsed five months after achieving spontaneous remission. About 60% of abnormal cells were identified. No changes in immunophenotype or the results of genetic studies were observed. The patient underwent a HyperCVAD chemotherapy regimen for six cycles. Consolidation therapy was performed via allogeneic bone marrow transplantation with an HLA-unrelated donor. One year after the bone marrow transplant, the patient died due to the progression of his underlying disease, coinciding with a respiratory infection caused by SARS-CoV-2. In the available literature, SRs are often linked to infections or other stimulators of the immune system, suggesting that powerful immune activation could play a role in controlling the leukemic clone. Nevertheless, the underlying mechanism of this phenomenon is not clearly understood. We hypothesize that the immune system would force the leukemic stem cell (LSC) to undergo a state of quiescence. This loss of replication causes the LSC progeny to die off, resulting in the SR of BPDCN.

show abstract

Section: Discussionmentioning

confidence: 85%

“…To date, there have only been five documented cases of spontaneous remission in BPDCN [16]. Here, we report one of the few cases described in the literature with a spontaneous complete remission of BPDCN, followed by relapse of the same disease within a short period of time.…”

Section: Introductionmentioning

confidence: 83%

Spontaneous Remission of Blastic Plasmacytoid Dendritic Cell Neoplasm: A Case Report

Castaño-Bonilla,

Mata,

Láinez-González

et al. 2024

Medicina

View full text Add to dashboard Cite

show abstract

“…CT has been mainly used to assess the extent and depth of the skin lesions, as well as the metastases in patients with BPDCN [4] , [5] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] . CT shows round and ovoid homogeneous cutaneous mass with soft tissue density with or without subcutaneous invasion as seen in the present case.…”

Section: Discussionmentioning

confidence: 99%

“…Although there has been at least eleven reports of BPDCN with CT findings, knowledge of chronological changes of CT findings has been limited. To our knowledge, only two case reports demonstrated the sequential CTs, both of which showed the chronological changes of the involved spleen [ 13 , 14 ], we first report the time course of skin lesions of BPDCN with sequential CT examinations [7] . CT demonstrated that multiple skin nodules fused to each other, resulting in a thickening of the entire skin, with some surface erosions and crusting in this case.…”

Section: Discussionmentioning

confidence: 99%

“…Rapid progression to the terminal leukemic phase is common, resulting in a short mean survival of 12–16 months [3] . To our knowledge, there have been at least 11 reports with CT findings [4] , [5] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] , including 4 18 F- fluorodeoxyglucose positron emission tomography / CT ( 18 F-FDG PET/CT) reports, associated with BPDCN; however, knowledge of chronological changes on sequential CT examinations have been limited [ 13 , 14 ]. We herein report a case of a patient with rapidly progressing BPDCN with a sequential evaluation on CT.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Rapidly progressing blastic plasmacytoid dendritic cell neoplasm causing diffuse skin thickening: A case report with sequential computed tomography examinations

Yoshioka

Kurokawa

Amemiya

et al. 2021

Radiology Case Reports

View full text Add to dashboard Cite

Blastic plasmacytoid dendritic cell neoplasm is a rare aggressive myeloid malignancy thought to be derived from precursor plasmacytoid dendritic cells. Rapid progression and poor prognosis have been known. We herein present a case of BPDCN in a previously healthy man who presented with suddenly developed multiple deep purple skin rashes, with sequential computed tomography examinations. The follow-up computed tomography demonstrated that multiple skin nodules observed in the initial MRI fused, resulting in a thickening of the entire skin, with some surface erosions and crusting. Blastic plasmacytoid dendritic cell neoplasm should be considered in the differentials in patients with a sudden onset and rapidly progressing skin rash or thickening.

show abstract

Mechanisms of Spontaneous Regression of Malignant Tumors in Inflammation

M.D.

2022

Global'nyye problemy sovremennosti

View full text Add to dashboard Cite

Annotation: The study of the mechanisms of spontaneous regression of malignant tumors is a topical issue of our century. Disclosure of the pathogenesis of regression will allow progress in the treatment of such diseases. The article discusses the mechanisms of regression of malignant tumors in inflammation, discloses the role of bacteria in this issue, and analyzes the possible mechanism of immunity sensitization in such diseases. The review of immune mechanisms was carried out on the basis of these articles, which studied the Coli toxin, BCG vaccine. The results showed the effectiveness of the use of these funds in the treatment of cancer patients. The possibilities of using bacteria for the transfer of drugs to the tumor area and their production of cytokines are also described. In addition, the correlation between the time of onset of inflammation and regression of tumor formation was analyzed. The main antitumor compounds and substances (IL-1, IL-12, TNF family, LIGHT and CCL21) were also noted, their effect on tumor cells and their role in the regression of malignant tumors was described. As a result, it was concluded that bacteria increase the ability of the immune system to recognize and attack tumor cells, however, the individual characteristics of the reactivity of the human immune system should be taken into account before such therapy.

show abstract

Spontaneous Regression of Blastic Plasmacytoid Dendritic Cell Neoplasm Following Sepsis by <i>Serratia marcescens</i>: A Case Report and Literature Review

Cited by 3 publications

References 30 publications

Spontaneous Remission of Blastic Plasmacytoid Dendritic Cell Neoplasm: A Case Report

Spontaneous Remission of Blastic Plasmacytoid Dendritic Cell Neoplasm: A Case Report

Rapidly progressing blastic plasmacytoid dendritic cell neoplasm causing diffuse skin thickening: A case report with sequential computed tomography examinations

Mechanisms of Spontaneous Regression of Malignant Tumors in Inflammation

Contact Info

Product

Resources

About