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1. Jegalian AG, Buxbaum NP, Facchetti F, Raffeld M, Pittaluga S, Wayne AS, Jaffe ES: Blastic plasmacytoid dendritic cell neoplasm in children: diagnostic features and clinical implications. Haematologica; 2010 Nov;95(11):1873-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Blastic plasmacytoid dendritic cell neoplasm in children: diagnostic features and clinical implications.
  • BACKGROUND: Blastic plasmacytoid dendritic cell neoplasm is a rare malignancy that typically follows a highly aggressive clinical course in adults, whereas experience in children with this disease is very limited.
  • DESIGN AND METHODS: This retrospective study analyzed the pathological and clinical findings of nine cases of blastic plasmacytoid dendritic cell neoplasm presenting in patients under the age of 18 years who were reviewed at our institution.
  • RESULTS: In the combined analysis, the overall survival rate among the 25 patients with available follow-up, all having received chemotherapy, was 72% (follow-up ranging from 9 months to 13 years, with a median of 30 months).
  • Nine patients were alive 5 years after the original diagnosis, although only three of them had undergone hematopoietic stem cell transplantation--one in first complete remission and two in second remission.
  • Of the seven patients who lacked cutaneous disease at presentation, 100% survived, including five who were alive more than 5 years after diagnosis, although only two had undergone stem cell transplantation.
  • Among the 18 patients who presented with cutaneous disease and for whom follow-up data were available, only 11 survived (61%).
  • Unexpectedly, three of four cases of blastic plasmacytoid dendritic cell neoplasm tested showed focal positivity for S-100.
  • S-100 was negative in 28 cases of acute myeloid leukemia evaluated for this marker.
  • CONCLUSIONS: In contrast to adult cases, in which long-term survival depends on stem cell transplantation in first complete remission, blastic plasmacytoid dendritic cell neoplasms in children are clinically less aggressive.
  • Treatment with high-risk acute lymphoblastic leukemia-type chemotherapy appears to be effective, and stem cell transplantation may be reserved for children who relapse and achieve a second remission.
  • Outcomes were more favorable in cases that lacked cutaneous disease at presentation, although a comparison of cutaneous and non-cutaneous cases might be confounded by differences in treatment regimens.
  • Focal expression of S-100 may be seen in concert with other markers of plasmacytoid dendritic cells.

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  • (PMID = 20663945.001).
  • [ISSN] 1592-8721
  • [Journal-full-title] Haematologica
  • [ISO-abbreviation] Haematologica
  • [Language] ENG
  • [Grant] United States / Intramural NIH HHS / /
  • [Publication-type] Journal Article; Research Support, N.I.H., Intramural
  • [Publication-country] Italy
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / S100 Proteins
  • [Other-IDs] NLM/ PMC2966909
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2. Su O, Onsun N, Demirkesen C, Aydin Y, Pirmit S, Gereli M: A case of CD4+/CD56+ hematodermic neoplasm (plasmacytoid dendritic cell neoplasm). Dermatol Online J; 2010;16(4):8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] A case of CD4+/CD56+ hematodermic neoplasm (plasmacytoid dendritic cell neoplasm).
  • CD4+/CD56+ hematodermic neoplasm (blastic plasmacytoid dendritic cell neoplasm) involving the skin is relatively rare and has been of significant interest in the recent literature.
  • Histological examination of skin biopsies showed an intense hematolymphoid infiltration in the dermis and in the subcutaneous tissue.
  • Stains were positive for CD4 (weak), CD56, and terminal deoxynucleotidyl transferase (TdT).
  • These cells were negative for CD2, CD3, CD5, CD10, CD20, CD30, CD68, and T cell intracellular antigen (TIA).
  • In situ hybridization (ISH) for Epstein-Barr virus was negative and the diagnosis was blastic NK cell lymphoma.
  • The patient was treated with a hyper-CVAD regimen (cyclophosphamide, vincristine, doxorubicine, dexamethasone, methotrexate, and cytarabine).This treatment regimen achieved partial remission but the patient died eight months after the diagnosis.
  • The patient presented with exclusively cutaneous involvement at the beginning but progressed rapidly and died shortly after despite aggressive chemotherapy.
  • Due to its rarity, we present here a case of CD4+/CD56+ hematodermic neoplasm.
  • [MeSH-major] Antigens, CD4 / immunology. Antigens, CD56 / immunology. Lymphoma, T-Cell, Cutaneous / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Cyclophosphamide / therapeutic use. DNA Nucleotidylexotransferase / analysis. Dendritic Cells / immunology. Dendritic Cells / pathology. Dexamethasone / therapeutic use. Doxorubicin / therapeutic use. Fatal Outcome. Humans. Male. Middle Aged. Vincristine / therapeutic use

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  • (PMID = 20409415.001).
  • [ISSN] 1087-2108
  • [Journal-full-title] Dermatology online journal
  • [ISO-abbreviation] Dermatol. Online J.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56; 5J49Q6B70F / Vincristine; 7S5I7G3JQL / Dexamethasone; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; EC 2.7.7.31 / DNA Nucleotidylexotransferase; CVAD protocol
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3. Chang HJ, Lee MD, Yi HG, Lim JH, Lee MH, Shin JH, Choi SJ, Moon Y, Nahm CH, Kim CS: A case of blastic plasmacytoid dendritic cell neoplasm initially mimicking cutaneous lupus erythematosus. Cancer Res Treat; 2010 Dec;42(4):239-43

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] A case of blastic plasmacytoid dendritic cell neoplasm initially mimicking cutaneous lupus erythematosus.
  • Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare disease.
  • The prognosis is poor in most cases with rapid progression despite administering chemotherapy.
  • A 67-year-old man complained of skin rashes on his back and this spread to the trunk, face, arms and thighs, and he was initially diagnosed with cutaneous lupus erythematosus according to the skin biopsy.
  • Repeated skin biopsy revealed a diffuse infiltration of lymphoid cells with medium sized nuclei, positive for CD4 and CD56, negative for Epstein-Barr virus (EBV), indicating a diagnosis of BPDCN.
  • He was treated with six cycles of combination chemotherapy consisting of ifosphamide, methotrexate, etoposide, prednisolone and L-asparaginase, and he achieved a partial response.
  • Herein we report on a rare case of BPDCN that was initially misinterpreted as cutaneous lupus erythematosus.

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  • (PMID = 21253327.001).
  • [ISSN] 2005-9256
  • [Journal-full-title] Cancer research and treatment : official journal of Korean Cancer Association
  • [ISO-abbreviation] Cancer Res Treat
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Korea (South)
  • [Other-IDs] NLM/ PMC3021744
  • [Keywords] NOTNLM ; Cutaneous lupus erythematosus / Drug therapy / Neoplasm / Plasmacytoid dendritic cells
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4. Ruggiero A, Maurizi P, Larocca LM, Arlotta A, Riccardi R: Childhood CD4+/CD56+ hematodermic neoplasm: case report and review of the literature. Haematologica; 2006 Dec;91(12 Suppl):ECR48
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Childhood CD4+/CD56+ hematodermic neoplasm: case report and review of the literature.
  • Recently, rare CD4+/CD56+ hematodermic neoplasm has been described as a distinct clinico-pathologic entity, with aggressive course and poor outcome.
  • To date, no standardized therapeutic approach to this disease has been established.
  • We report an additional case of CD4+/CD56+ hematodermic tumour that showed a good response to chemotherapy based on a lymphoma protocol.
  • Moreover, we try to analyse features and outcome of a few other paediatric CD4+/CD56+ hematodermic tumours as they are reported in the literature.
  • [MeSH-major] Lymphoma, Non-Hodgkin / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] 6-Mercaptopurine / administration & dosage. Antigens, CD4 / analysis. Antigens, CD56 / analysis. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Asparaginase / administration & dosage. Child. Cyclophosphamide / administration & dosage. Cytarabine / administration & dosage. Daunorubicin / administration & dosage. Dendritic Cells / pathology. Dexamethasone / administration & dosage. Humans. Male. Methotrexate / administration & dosage. Prednisone / administration & dosage. Remission Induction. Vincristine / administration & dosage

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  • (PMID = 17194654.001).
  • [ISSN] 1592-8721
  • [Journal-full-title] Haematologica
  • [ISO-abbreviation] Haematologica
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Italy
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56; 04079A1RDZ / Cytarabine; 5J49Q6B70F / Vincristine; 7S5I7G3JQL / Dexamethasone; 8N3DW7272P / Cyclophosphamide; E7WED276I5 / 6-Mercaptopurine; EC 3.5.1.1 / Asparaginase; VB0R961HZT / Prednisone; YL5FZ2Y5U1 / Methotrexate; ZS7284E0ZP / Daunorubicin
  • [Number-of-references] 25
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5. Yoshimasu T, Manabe A, Tanaka R, Mochizuki S, Ebihara Y, Ishikawa K, Iseki T, Oyaizu N, Aritaki K, Tanaka K, Tsuruta T, Hoshika A, Asano S, Tsuji K: Successful treatment of relapsed blastic natural killer cell lymphoma with unrelated cord blood transplantation. Bone Marrow Transplant; 2002 Jul;30(1):41-4
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  • [Title] Successful treatment of relapsed blastic natural killer cell lymphoma with unrelated cord blood transplantation.
  • The prognosis for blastic natural killer (NK) cell lymphoma is generally dismal.
  • A 15-year-old boy was diagnosed as having blastic NK cell lymphoma in the cervical lymph nodes.
  • Autologous peripheral blood stem cell transplantation was performed on achieving a complete remission.
  • Chemotherapy induced a second remission and the patient received UCBT with a conditioning regimen consisting of total body irradiation, thiotepa and cyclophosphamide.
  • At the time of writing, he remains in remission 18 months after UCBT with an excellent performance status.
  • UCBT may be an option for patients with blastic NK cell lymphoma.
  • [MeSH-major] Blast Crisis / therapy. Cord Blood Stem Cell Transplantation. Killer Cells, Natural / pathology. Lymphoma, T-Cell / therapy
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Disease-Free Survival. Humans. Immunophenotyping. Male. Remission Induction / methods

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  • (PMID = 12105776.001).
  • [ISSN] 0268-3369
  • [Journal-full-title] Bone marrow transplantation
  • [ISO-abbreviation] Bone Marrow Transplant.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 23
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6. Tsukune Y, Isobe Y, Yasuda H, Shimizu S, Katsuoka Y, Hosone M, Oshimi K, Komatsu N, Sugimoto K: Activity and safety of combination chemotherapy with methotrexate, ifosfamide, l-asparaginase and dexamethasone (MILD) for refractory lymphoid malignancies: a pilot study. Eur J Haematol; 2010 Apr;84(4):310-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Activity and safety of combination chemotherapy with methotrexate, ifosfamide, l-asparaginase and dexamethasone (MILD) for refractory lymphoid malignancies: a pilot study.
  • Optimal salvage chemotherapy has not been established for lymphoid malignancy, which is refractory to the conventional cyclophosphamide, doxorubicin, vincristine, and prednisone regimen.
  • To explore an effective regimen, we conducted a phase I pilot study of combination chemotherapy with methotrexate, ifosfamide, l-asparaginase and dexamethasone (MILD), which are unaffected by MDR1-encoded P-glycoprotein.
  • Eleven patients had T/NK-cell malignancies, six had B-cell malignancies, and one was diagnosed with a blastic plasmacytoid dendritic cell neoplasm.
  • Eleven patients completed two courses of MILD therapy.
  • Treatment-related death because of systemic mucormycosis was observed in one patient.
  • Major treatment-related adverse events were grade 3 or more hematologic toxicities, which included lymphopenia corresponding to dose-limiting toxicity.
  • It was very interesting that all of seven responders had T/NK-cell malignancies.
  • MILD therapy was feasible and presented acceptable toxicity in patients with refractory or lethal lymphoid malignancies.
  • The efficacy for T/NK-cell malignancies should be further evaluated.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Hematologic Neoplasms / drug therapy

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  • (PMID = 20015242.001).
  • [ISSN] 1600-0609
  • [Journal-full-title] European journal of haematology
  • [ISO-abbreviation] Eur. J. Haematol.
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase I; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] EC 3.5.1.1 / Asparaginase; UM20QQM95Y / Ifosfamide; YL5FZ2Y5U1 / Methotrexate
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7. Eros N, Marschalkó M, Balassa K, Hídvégi B, Szakonyi J, Ilniczky S, Borka K, Kovács A, Bottlik G, Hársing J, Csomor J, Szepesi A, Matolcsy A, Kárpáti S, Demeter J: Central nervous system involvement in CD4+/CD56+ hematodermic neoplasm: a report of two cases. J Neurooncol; 2010 Apr;97(2):301-4

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Central nervous system involvement in CD4+/CD56+ hematodermic neoplasm: a report of two cases.
  • CD4+/CD56+ hematodermic neoplasm, formerly known as blastic NK-cell lymphoma, is an uncommon, aggressive non-Hodgkin's lymphoma with cutaneous, lymph node, and bone marrow involvement at presentation.
  • The disease is characterized by early leukemic phase; however, central nervous system involvement is rarely reported.
  • Herein we describe two cases of CD4+/CD56+ hematodermic neoplasm with meningeal manifestation.
  • Authors recommend routine cerebrospinal fluid analysis and prophylactic intrathecal chemotherapy in patients with this highly aggressive disease.
  • [MeSH-major] Lymphoma, Extranodal NK-T-Cell / pathology. Lymphoma, Extranodal NK-T-Cell / physiopathology. Meningeal Neoplasms / pathology. Meningeal Neoplasms / physiopathology
  • [MeSH-minor] Aged. Antigens, CD56 / metabolism. Antineoplastic Agents / therapeutic use. CD4-Positive T-Lymphocytes / metabolism. CD4-Positive T-Lymphocytes / pathology. Cell Separation. Fatal Outcome. Female. Flow Cytometry. Humans. Immunophenotyping. Male

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  • (PMID = 19798469.001).
  • [ISSN] 1573-7373
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antigens, CD56; 0 / Antineoplastic Agents
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8. Male HJ, Davis MB, McGuirk JP, Abhyankar S, Aljitawi OS, Zhang D, Ganguly S: Blastic plasmacytoid dendritic cell neoplasm should be treated with acute leukemia type induction chemotherapy and allogeneic stem cell transplantation in first remission. Int J Hematol; 2010 Sep;92(2):398-400
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Blastic plasmacytoid dendritic cell neoplasm should be treated with acute leukemia type induction chemotherapy and allogeneic stem cell transplantation in first remission.
  • Blastic plasmacytoid dendritic cell (BPDC) neoplasm is a rare but clinically aggressive tumor known to be derived from the precursors of plasmacytoid dendritic cells (CD123+) with a high frequency of cutaneous and bone marrow involvement.
  • Though majority of the patients initially respond to multi-agent chemotherapy, most would relapse within a year.
  • We hereby report a patient with disseminated cutaneous BPDC with marrow involvement diagnosed by typical histo-pathological and flow-cytometric findings.
  • He was subsequently treated with leukemia type induction regimen followed by allogeneic stem cell transplantation in first complete remission.
  • We recognize that BPDC with marrow involvement behaves like acute myeloid leukemia and aggressive treatment followed by stem cell transplantation may lead to long-term remission in selected cases.
  • [MeSH-major] Dendritic Cells / pathology. Hematopoietic Stem Cell Transplantation / methods. Leukemia / therapy
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Bone Marrow. Humans. Male. Middle Aged. Remission Induction. Skin Neoplasms / therapy. Transplantation Chimera. Transplantation, Homologous

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  • [Cites] Blood. 2002 Mar 1;99(5):1556-63 [11861268.001]
  • [Cites] Am J Clin Pathol. 2005 May;123(5):662-75 [15981806.001]
  • [Cites] Blood. 2009 Jul 30;114(5):937-51 [19357394.001]
  • (PMID = 20697854.001).
  • [ISSN] 1865-3774
  • [Journal-full-title] International journal of hematology
  • [ISO-abbreviation] Int. J. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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9. Kaune KM, Baumgart M, Bertsch HP, Mitteldorf C, Müller-Hermelink HK, Haase D, Ghadimi BM, Schön MP, Neumann C: Solitary cutaneous nodule of blastic plasmacytoid dendritic cell neoplasm progressing to overt leukemia cutis after chemotherapy: immunohistology and FISH analysis confirmed the diagnosis. Am J Dermatopathol; 2009 Oct;31(7):695-701
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Solitary cutaneous nodule of blastic plasmacytoid dendritic cell neoplasm progressing to overt leukemia cutis after chemotherapy: immunohistology and FISH analysis confirmed the diagnosis.
  • Blastic plasmacytoid dendritic cell (BPDC) neoplasm, formerly called blastic natural killer cell lymphoma or CD4+/CD56+ hematodermic neoplasm, is a rare tumor entity, now regarded to be derived from the plasmacytoid dendritic cell (PDC) lineage.
  • Because over 90% of patients present with skin lesions usually early in their disease, dermatologists have to be familiar with the specific diagnostic features and the clinical course of this devastating disease.
  • We present a woman with a long standing solitary skin tumor of BPDC neoplasm, who experienced a deleterious clinical course, which is typical for this disease.
  • Phenotypic and karyotypic characteristics distinguishing this tumor from myelomonocytic leukemia with skin involvement are presented.
  • [MeSH-major] Dendritic Cells / pathology. Leukemia / pathology. Lymphoma / pathology. Neoplasms, Second Primary / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] Aged. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Arthritis, Rheumatoid / complications. Biomarkers, Tumor / analysis. Diabetes Mellitus, Type 2 / complications. Disease Progression. Fatal Outcome. Female. Humans. Immunohistochemistry. In Situ Hybridization, Fluorescence

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  • (PMID = 19684511.001).
  • [ISSN] 1533-0311
  • [Journal-full-title] The American Journal of dermatopathology
  • [ISO-abbreviation] Am J Dermatopathol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
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10. Nizza D, Simoneaux SF: Blastic plasmacytoid dendritic cell neoplasm presenting as a subcutaneous mass in an 8-year-old boy. Pediatr Radiol; 2010 Dec;40 Suppl 1:S40-2
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Blastic plasmacytoid dendritic cell neoplasm presenting as a subcutaneous mass in an 8-year-old boy.
  • Blastic plasmacytoid dendritic cell neoplasm (also referred to as CD4+/CD56+ hematodermic neoplasm) is a rare hematological malignancy typically seen in older adults.
  • The disease presents with nonspecific cutaneous lesions and advances toward a fatal leukemic phase despite an often favorable initial response to chemotherapy.
  • We report a case of histopathologically proven blastic plasmacytoid dendritic cell neoplasm arising in an otherwise healthy and asymptomatic 8-year-old boy who noticed a painless mass within the subcutaneous tissues below the left calf.
  • The case is notable both for the young age of the patient and for the absence of characteristic cutaneous manifestations of this disease.
  • [MeSH-major] Dendritic Cells / diagnostic imaging. Dendritic Cells / pathology. Diagnostic Imaging / methods. Plasmacytoma / diagnosis. Soft Tissue Neoplasms / diagnosis
  • [MeSH-minor] Child. Diagnosis, Differential. Humans. Leg / diagnostic imaging. Leg / pathology. Male. Radionuclide Imaging

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  • (PMID = 20552185.001).
  • [ISSN] 1432-1998
  • [Journal-full-title] Pediatric radiology
  • [ISO-abbreviation] Pediatr Radiol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
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11. Machet L, De Muret A, Wiezberka E, Bernez A, Abdallah-Lotf M, Linassier C, Petrella T: [Agranular CD4+ CD56+ CD123+ hematodermic neoplasm (blastic NK-cell lymphoma) revealed by cutaneous localization: 2 cases]. Ann Dermatol Venereol; 2004 Nov;131(11):969-73
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  • [Title] [Agranular CD4+ CD56+ CD123+ hematodermic neoplasm (blastic NK-cell lymphoma) revealed by cutaneous localization: 2 cases].
  • [Transliterated title] Localisations cutanées révélatrices d'une hématodermie CD4+ CD56+ CD123+: 2 cas.
  • BACKGROUND: Agranular CD4+ CD56+ hematodermic neoplasm (blastic NK-cell lymphoma) has been recently described.
  • OBSERVATIONS: Two old men of respectively 70 and 77 years consulted for infiltrated cutaneous lesions.
  • Preliminary histological examination of cutaneous biopsy taken in both patients showed a malignant proliferation suggesting a cutaneous lymphoma, and the patients were referred.
  • Histological examination of new biopsies showed a very similar proliferation in the 2 cases of monotonous medium-sized mononuclear cells without expression of the common antigens CD3 and CD20 and the expression of CD4, CD56, and CD123.
  • No rearrangement of the T-cell receptor gene or the immunoglobulin heavy chain gene were evidenced.
  • Thrombocytopenia associated with the abnormal presence of 15 p. 100 of circulating CD4+ CD56+ cells was initially found in the second patient.
  • The first patient was treated with chemotherapy, with complete remission.
  • A cutaneous relapse promptly occurred, followed by bone and cerebral localizations.
  • The patient died one year after the diagnosis of the disease, in spite of intensification of the treatment.
  • Treatment is still ongoing in the second patient.
  • COMMENTS: The histological presentation of these two patients was very similar with an unusual phenotype of tumor cells expressing CD4, CD56, CD123, but not expressing CD3 and CD20.
  • Some cases have been published under the "term of blastic NK lymphoma" which is the actual term for the disease in the WHO classification.
  • However, the tumor cells derive from the dendritic plasmacytoid cells, also called type 2 dendritic cells, and perhaps from a common precursor to lymphocyte T and dendritic plasmacytoid cells.
  • In spite of complete cutaneous response in the 2 cases presented, as in other reports, extra-cutaneous involvement occurs quickly.
  • [MeSH-major] Antigens, CD4 / analysis. Antigens, CD56 / analysis. Killer Cells, Natural. Lymphoma, Non-Hodgkin / immunology. Lymphoma, Non-Hodgkin / pathology. Receptors, Interleukin-3 / analysis. Skin Neoplasms / immunology. Skin Neoplasms / pathology
  • [MeSH-minor] Aged. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Fatal Outcome. Humans. Interleukin-3 Receptor alpha Subunit. Male. Phenotype

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  • (PMID = 15602384.001).
  • [ISSN] 0151-9638
  • [Journal-full-title] Annales de dermatologie et de vénéréologie
  • [ISO-abbreviation] Ann Dermatol Venereol
  • [Language] fre
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] France
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56; 0 / IL3RA protein, human; 0 / Interleukin-3 Receptor alpha Subunit; 0 / Receptors, Interleukin-3
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12. Löffler H, Kosely F, Ho AD, Krämer A: [Blastic plasmacytoid dendritic cell neoplasm - a rare differential diagnosis of neoplastic skin infiltrations associated with systemic symptoms]. Dtsch Med Wochenschr; 2009 Sep;134(39):1927-30
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  • [Title] [Blastic plasmacytoid dendritic cell neoplasm - a rare differential diagnosis of neoplastic skin infiltrations associated with systemic symptoms].
  • [Transliterated title] Blastische plasmozytoide Neoplasie dendritischer Zellen - seltene Differenzialdiagnose neoplastischer Hautinfiltrate mit systemischer Beteiligung.
  • HISTORY AND CLINICAL FINDINGS: A 70-year-old female patient developed a non-pruritic, indolent rash associated with infections and peripheral blood abnormalities.
  • A skin biopsy was suggestive of malignant lymphoma of the skin.
  • Additional immunohistochemistry of the skin specimen as well as cytologic and flow cytometric examination of the bone marrow revealed an immature cell population expressing CD4 and CD56 which infiltrated both the dermis and, with an infiltration grade of about 85 %, the bone marrow.
  • DIAGNOSIS: Blastic plasmacytoid dendritic cell neoplasm (formerly known as blastic NK cell lymphoma).
  • TREATMENT AND COURSE: After the first course of induction chemotherapy with daunorubicin and cytarabin, both the rash and the hematologic findings of bone marrow and peripheral blood showed a complete remission.
  • CONCLUSION: The blastic plasmacytoid dendritic cell neoplasm is a rare, aggressive hematopoietic neoplasm most likely related to acute myeloid leukemia (AML).
  • Since cutaneous involvement is regularly present at diagnosis, the differential diagnosis of unexplained skin lesions should include this disease entity, especially if peripheral blood abnormalities are present.
  • Despite the initial response to cytostatic therapy being mostly excellent, the prognosis is poor.
  • Hence, treatment as high-risk AML seems advisable.
  • [MeSH-major] Leukemia, Myeloid, Acute / diagnosis. Skin / pathology. Skin Neoplasms / diagnosis
  • [MeSH-minor] Aged. Antineoplastic Agents / therapeutic use. Biopsy. Bone Marrow / pathology. Diagnosis, Differential. Exanthema. Female. Flow Cytometry. Humans. Immunohistochemistry. Immunophenotyping. Pancytopenia. Prognosis. Remission Induction

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  • [Copyright] Georg Thieme Verlag KG Stuttgart, New York.
  • (PMID = 19760552.001).
  • [ISSN] 1439-4413
  • [Journal-full-title] Deutsche medizinische Wochenschrift (1946)
  • [ISO-abbreviation] Dtsch. Med. Wochenschr.
  • [Language] ger
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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13. Tse E, Liang RH: Natural killer cell neoplasms. Clin Lymphoma; 2004 Dec;5(3):197-201
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  • [Title] Natural killer cell neoplasms.
  • Lymphoid neoplasms that are derived from natural killer (NK) cells are uncommon but distinct clinicopathologic disease entities.
  • Three types have been recognized and categorized in the latest World Health Organization classification: extranodal NK cell lymphoma, nasal-type; aggressive NK cell leukemia; and blastic NK cell lymphoma.
  • All NK tumor cells express the NK cell marker CD56, but they lack the expression of surface CD3 and the rearrangement of T-cell receptor genes, which distinguish them from T-lymphoid neoplasms.
  • There is also a strong association with the Epstein-Barr virus, except in blastic NK cell lymphoma.
  • Extranodal involvement by the NK cell tumor is common, especially in the nasal cavity, the skin, and the gastrointestinal tract.
  • All 3 NK cell neoplasms are characterized by aggressive clinical course and poor response to treatment.
  • Although the optimal treatment modality remains to be determined, good initial response to combined radiation therapy and chemotherapy has been observed in localized disease.
  • Further studies in the basic biology of the NK cell and the pathology of NK cell neoplasms may shed light on the development of newer and more effective therapy.
  • [MeSH-major] Killer Cells, Natural / pathology. Lymphoma / immunology. Lymphoma / pathology

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  • (PMID = 15636697.001).
  • [ISSN] 1526-9655
  • [Journal-full-title] Clinical lymphoma
  • [ISO-abbreviation] Clin Lymphoma
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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14. Calvo M, González C, Martín E, Marqués A, Jaén P: [Blastic NK-cell lymphoma]. Actas Dermosifiliogr; 2006 May;97(4):253-6
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  • [Title] [Blastic NK-cell lymphoma].
  • [Transliterated title] Linfoma NK blástico.
  • Blastic NK (<<natural killer>>) cell lymphoma is a rare type of lymphoma, recognized as an independent entity in the new World Health Organization-European Organization for Research and Treatment of Cancer (WHO-EORTC) classification for cutaneous lymphomas.
  • It frequently starts with extranodal involvement (primarily cutaneous), often presents with splenomegaly and pancytopenia, and initially responds well to chemotherapy, although the medium-term prognosis is usually poor.
  • It was first considered to originate from immature NK lymphocytes, but now its origin is thought to be plasmacytoid dendritic cell precursors.
  • We present the clinicopathological characteristics of a new case of this entity in a 78-year-old male patient.
  • [MeSH-major] Killer Cells, Natural / pathology. Lymphoma, Non-Hodgkin / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] Aged. Antigens, CD / analysis. Antigens, Neoplasm / analysis. Humans. Immunophenotyping. Male

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  • (PMID = 16801018.001).
  • [ISSN] 0001-7310
  • [Journal-full-title] Actas dermo-sifiliográficas
  • [ISO-abbreviation] Actas Dermosifiliogr
  • [Language] spa
  • [Publication-type] Case Reports; English Abstract; Journal Article; Review
  • [Publication-country] Spain
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / Antigens, Neoplasm
  • [Number-of-references] 12
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15. Child FJ, Mitchell TJ, Whittaker SJ, Calonje E, Spittle M, Crocker J, Russell-Jones R: Blastic natural killer cell and extranodal natural killer cell-like T-cell lymphoma presenting in the skin: report of six cases from the UK. Br J Dermatol; 2003 Mar;148(3):507-15
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  • [Title] Blastic natural killer cell and extranodal natural killer cell-like T-cell lymphoma presenting in the skin: report of six cases from the UK.
  • BACKGROUND: Some lymphomas express natural killer (NK)-cell markers such as the neural cell adhesion molecule, which is recognized by the CD56 antibody.
  • The new World Health Organization classification of lymphoma/leukaemia recognizes several types of NK/T-cell neoplasm, including blastic NK-cell lymphoma, which characteristically presents with cutaneous lesions.
  • OBJECTIVES: To describe the clinical, pathological and molecular features in six cases of CD56+ lymphoma with cutaneous presentation.
  • In addition, in situ hybridization (ISH) to identify Epstein-Barr virus (EBV) mRNA, and polymerase chain reaction analysis to identify the presence of a clonal population of T cells or B cells were performed on lesional skin.
  • Four patients developed disseminated disease, three with neurological involvement.
  • These four patients died between 14 and 46 months following diagnosis (median 30 months).
  • In four of six cases the histopathological and immunohistological features were in keeping with a blastic NK-cell lymphoma.
  • No clonal immunoglobulin heavy chain (IgH) or T-cell receptor (TCR) gene rearrangement was detected in the four cases consistent with an origin from NK cells.
  • A further case fitted the criteria for an extranodal NK/T-cell lymphoma of nasal type and was also the only case to show evidence of EBV mRNA by ISH.
  • A clonal T-cell population was identified in the final case.
  • This patient also exhibited molecular evidence of a clonal B-cell population and a t(14;18) translocation confirmed by sequence analysis.
  • CONCLUSIONS: Our data confirm that NK-cell lymphomas presenting in the skin are a heterogeneous group, and that in the U.K., blastic NK-cell lymphoma is more common than extranodal NK/T-cell lymphoma of nasal type.
  • These lymphomas pursue an aggressive course, with rapid development of disseminated disease, and resistance to chemotherapy.
  • Detailed immunophenotyping is needed to distinguish the different types.
  • Our molecular data indicate that blastic NK-cell lymphoma cases lack clonal TCR/IgH gene rearrangements consistent with an NK-cell origin.
  • Our ISH findings indicate that EBV plays a pathogenetic role only in extranodal NK/T-cell lymphoma of nasal type.
  • [MeSH-major] Killer Cells, Natural / immunology. Lymphoma, T-Cell, Cutaneous / immunology. Ribosomal Proteins. Skin Neoplasms / immunology
  • [MeSH-minor] Adult. Aged. Antigens, CD56 / immunology. Female. Gene Rearrangement, gamma-Chain T-Cell Antigen Receptor / genetics. Humans. Immunoglobulin Heavy Chains / genetics. In Situ Hybridization. Male. Middle Aged. Polymerase Chain Reaction. RNA-Binding Proteins / analysis. Translocation, Genetic

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  • (PMID = 12653743.001).
  • [ISSN] 0007-0963
  • [Journal-full-title] The British journal of dermatology
  • [ISO-abbreviation] Br. J. Dermatol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antigens, CD56; 0 / Immunoglobulin Heavy Chains; 0 / RNA-Binding Proteins; 0 / Ribosomal Proteins; 135844-68-7 / RPL22 protein, human
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16. Greer JP, Mosse CA: Natural killer-cell neoplasms. Curr Hematol Malig Rep; 2009 Oct;4(4):245-52
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Natural killer-cell neoplasms.
  • The natural killer (NK)-cell neoplasms are rare, representing less than 1% of non-Hodgkin lymphoma, except in Asia and Latin America, where they represent 3% to 6%.
  • NK-cell neoplasms include immature acute leukemias; a blastic NK-cell lymphoma, which is obsolete because of its plasmacytoid dendritic-cell origin; and mature NK neoplasms, comprising extranodal NK/T-cell lymphoma (ENKL), nasal-type; aggressive NK-cell leukemia; and chronic NK-cell lymphoproliferative disorders, which are often reactive.
  • Epstein-Barr virus is usually detected in tumor cells of ENKL and aggressive NK-cell leukemia.
  • The latter two mature NK neoplasms are relatively chemoresistant because of the frequent expression of P-glycoprotein.
  • Early radiation is advocated for localized nasal ENKL.
  • Stem cell transplantation is recommended for advanced disease, owing to a poor prognosis.
  • Novel agents, including chemotherapy, inhibitors of molecular pathways, and monoclonal antibodies, are under investigation.
  • [MeSH-major] Killer Cells, Natural / pathology. Leukemia / pathology. Lymphoma / pathology. Lymphoproliferative Disorders / pathology
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Cell Lineage. Cell Transformation, Neoplastic. Humans. Stem Cell Transplantation

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  • (PMID = 20425414.001).
  • [ISSN] 1558-822X
  • [Journal-full-title] Current hematologic malignancy reports
  • [ISO-abbreviation] Curr Hematol Malig Rep
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents
  • [Number-of-references] 42
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17. Northup JK, Gadre SA, Ge Y, Lockhart LH, Velagaleti GV: Do cytogenetic abnormalities precede morphologic abnormalities in a developing malignant condition? Eur J Haematol; 2007 Feb;78(2):152-6
The Lens. Cited by Patents in .

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Do cytogenetic abnormalities precede morphologic abnormalities in a developing malignant condition?
  • Cytogenetic evaluation of bone marrow and neoplastic tissues plays a critical role in determining patient management and prognosis.
  • Here, we highlight two cases in which the cytogenetic studies challenge the common practice of using hematologic and morphologic changes as key factors in malignant disease management.
  • The first case is that of a lymph node sample from a 40-yr-old non-Hodgkin's lymphoma (NHL) patient sent for determination of disease progress.
  • Cytogenetic studies of lymph node showed multiple clonal abnormalities, most notably a der(18) from a t(14;18) which is associated with high-grade NHL.
  • After two cycles of chemotherapy with fludarabine, the patient did not show any clinical response, suggesting possible progression to high-grade lymphoma.
  • The second case is of a patient with a history of human immunodeficiency virus and blastic natural killer leukemia/lymphoma.
  • Hematologic studies of ascitic fluid classified the patient as having pleural effusion lymphoma whereas bone marrow analysis showed no malignancy.
  • Bone marrow cytogenetic studies showed multiple clonal abnormalities including a t(8;14), which is commonly associated with Burkitt's lymphoma (BL).
  • To our knowledge, this is the first case wherein a morphologically normal bone marrow showed presence of clonal abnormalities consistent with BL or Pleural effusion lymphoma.
  • After two cycles of CHOP (cyclophosphamide, doxorubicin, vincristine and prednisone) chemotherapy, the patient's general condition and ascitis improved and she was discharged.
  • These studies clearly demonstrate that genetic changes often precede morphologic changes in a developing malignant condition.
  • Therefore, the critical information needed for care of patients with malignant disorders may be incomplete or inaccurate if cytogenetic evaluation is overlooked.
  • [MeSH-major] Burkitt Lymphoma / genetics. Lymphoma, AIDS-Related / genetics. Lymphoma, Follicular / genetics. Lymphoma, Non-Hodgkin / genetics. Translocation, Genetic
  • [MeSH-minor] Adult. Antibodies, Monoclonal / administration & dosage. Antibodies, Monoclonal, Murine-Derived. Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bone Marrow / pathology. Chromosomes, Human, Pair 12 / ultrastructure. Chromosomes, Human, Pair 14 / genetics. Chromosomes, Human, Pair 14 / ultrastructure. Chromosomes, Human, Pair 18 / genetics. Chromosomes, Human, Pair 18 / ultrastructure. Chromosomes, Human, Pair 8 / genetics. Chromosomes, Human, Pair 8 / ultrastructure. Chromosomes, Human, X. Clone Cells / pathology. Cyclophosphamide / administration & dosage. Disease Progression. Doxorubicin / administration & dosage. Drug Resistance, Neoplasm. Female. Genes, myc. Humans. Karyotyping. Lymph Nodes / pathology. Male. Mutagenesis, Insertional. Pleural Effusion, Malignant / drug therapy. Pleural Effusion, Malignant / genetics. Pleural Effusion, Malignant / pathology. Prednisone / administration & dosage. Rituximab. Trisomy. Vidarabine / administration & dosage. Vidarabine / analogs & derivatives. Vincristine / administration & dosage

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  • (PMID = 17313561.001).
  • [ISSN] 0902-4441
  • [Journal-full-title] European journal of haematology
  • [ISO-abbreviation] Eur. J. Haematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Denmark
  • [Chemical-registry-number] 0 / Antibodies, Monoclonal; 0 / Antibodies, Monoclonal, Murine-Derived; 4F4X42SYQ6 / Rituximab; 5J49Q6B70F / Vincristine; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; FA2DM6879K / Vidarabine; P2K93U8740 / fludarabine; VB0R961HZT / Prednisone; CHOP protocol
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18. Oshimi K: NK cell lymphoma. Int J Hematol; 2002 Aug;76 Suppl 2:118-21

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] NK cell lymphoma.
  • Natural killer (NK) cells are lymphocytes with large granular lymphocyte morphology, CD3-CD56+ phenotype, non-MHC-restricted cytotoxicity, and germ-line configuration T-cell receptor genes.
  • Two types of lymphomas originating from NK cells have been described; blastic NK-cell lymphoma, and nasal-type NK-cell lymphoma.
  • Because recent reports indicate that blastic NK-cell lymphoma originates from the precursors of plasmacytoid dendritic cells, I will focus mainly on nasal-type NK-cell lymphoma, and discuss its pathogenesis, diagnostic problems, treatment strategy, and outcome.
  • Nasal-type NK-cell lymphoma develops mostly in the nasal cavity and rarely in other sites, such as the skin and intestinal tract.
  • Epstein-Barr virus (EBV) is found in lymphoma cells of almost all the patients, and is considered to be the etiologic agent.
  • Indeed, EBV easily infects NK cells in the absence of CD21 antigen, or EBV receptor, on the surface of NK cells.
  • Further, various types of oncogenes and suppressor oncogenes are found to be involved in its pathogenesis.
  • Based on the data obtained from paraffin-embedded specimens, it is difficult to determine whether the lymphoma cells are of T-cell or NK-cell lineage, because immunohistochemical staining of cytoplasmic CD3 is positive both in T and NK cells, and CD56 is positive in a part of T cells.
  • The presence of CD5 antigen indicates T-cell lineage.
  • When the disease is limited, radiation therapy is effective, but not satisfactory.
  • A new trial to use simultaneously both radiation and chemotherapy has started in Japan.
  • In advanced stages, a combination chemotherapy including L-asparaginase seems to be promising, and high-dose chemotherapy with autologous or allogeneic stem cell support is under investigation.
  • A recent report described the expression of short-length P-glycoprotein (P-gp), but not full-length P-gp in NK cells, and this mini-P-gp is unable to extrude daunorubicin.
  • These findings may change the treatment strategy.
  • Finally, I will present the results on interim analysis of 166 cases of nasal-type NK-cell lymphoma collected in Japan between 1994 and 1998.
  • [MeSH-major] Killer Cells, Natural. Lymphoma, T-Cell / pathology
  • [MeSH-minor] Combined Modality Therapy. Humans. Nose Neoplasms / diagnosis. Nose Neoplasms / etiology. Nose Neoplasms / pathology

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  • (PMID = 12430911.001).
  • [ISSN] 0925-5710
  • [Journal-full-title] International journal of hematology
  • [ISO-abbreviation] Int. J. Hematol.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Japan
  • [Number-of-references] 23
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19. An G, Qi JY, Zou DH, Zhao YZ, Chen HS, Qiu LG: [Blastic plasmacytoid dendritic cell neoplasm: two cases report and review of literatures]. Zhonghua Nei Ke Za Zhi; 2009 Mar;48(3):189-92
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  • [Title] [Blastic plasmacytoid dendritic cell neoplasm: two cases report and review of literatures].
  • OBJECTIVE: To identify the clinical and pathological features of blastic plasmacytoid dendritic cell neoplasm (BPDC).
  • METHODS: The characteristics of BPDC hematodermic neoplasm were discussed with a report of two new cases and review the literatures.
  • Lineage specific markers for B- and T-cell were negative and the tumors did not express myeloperoxidase.
  • Systemic chemotherapy resulted in complete remission, but the disease relapsed quickly and were unresponsive to further chemotherapy.
  • The patients died 26 months and 11 months respectively after diagnosis.
  • CONCLUSION: BPDC hematodermic neoplasm is a rare subtype of lymphoma with distinct clinicopathologic and immunophenotypic features.
  • The disease often has a fulminant course with a poor prognosis.
  • More recent studies suggest that there is a derivation from a plasmacytoid dendritic cell precursor.
  • [MeSH-major] Dendritic Cells / pathology. Lymphoma, T-Cell, Cutaneous / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] Humans. Killer Cells, Natural / pathology. Male. Middle Aged

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  • (PMID = 19576083.001).
  • [ISSN] 0578-1426
  • [Journal-full-title] Zhonghua nei ke za zhi
  • [ISO-abbreviation] Zhonghua Nei Ke Za Zhi
  • [Language] chi
  • [Publication-type] Case Reports; English Abstract; Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] China
  • [Number-of-references] 12
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20. Yamaguchi M, Maekawa M, Nakamura Y, Ueda M: Long-term remission of blastic natural killer-cell lymphoma after autologous peripheral blood stem-cell transplantation. Am J Hematol; 2005 Oct;80(2):124-7
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  • [Title] Long-term remission of blastic natural killer-cell lymphoma after autologous peripheral blood stem-cell transplantation.
  • We report here a case of blastic natural killer (NK)-cell lymphoma treated successfully with autologous peripheral blood stem-cell transplantation (APBSCT).
  • A 57-year-old man had skin tumors and was diagnosed as having blastic NK-cell lymphoma by tumor biopsy.
  • Chemotherapy led to complete remission (CR).
  • To sustain CR, the patient underwent high-dose chemotherapy in combination with 12 Gy of total-body irradiation (TBI) followed by autologous peripheral blood stem-cell rescue.
  • From this case, we concluded that APBSCT with preconditioning by TBI-containing regimens might be a cure-attaining treatment for disseminated blastic NK-cell lymphoma and should be considered as a choice of treatment in cases where no suitable donors for allogeneic transplantation are available.
  • [MeSH-major] Killer Cells, Natural / pathology. Lymphoma, T-Cell / therapy. Peripheral Blood Stem Cell Transplantation / methods

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  • [Copyright] (c) 2005 Wiley-Liss, Inc.
  • (PMID = 16184583.001).
  • [ISSN] 0361-8609
  • [Journal-full-title] American journal of hematology
  • [ISO-abbreviation] Am. J. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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21. Yamada O, Ichikawa M, Okamoto T, Park C, Motoji T, Mizoguchi H, Shibuya A: Killer T-cell induction in patients with blastic natural killer cell lymphoma/leukaemia: implications for successful treatment and possible therapeutic strategies. Br J Haematol; 2001 Apr;113(1):153-60
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  • [Title] Killer T-cell induction in patients with blastic natural killer cell lymphoma/leukaemia: implications for successful treatment and possible therapeutic strategies.
  • A rare form of putative natural killer (NK) cell lymphoma called blastic NK cell lymphoma appears to be clinicopathologically distinctive in showing a homogenous lymphoblast, variable expression of CD2, CD4, CD56 and TdT, negative for surface CD3, T-cell receptor antigen, CD16, CD34 and lack of association with Epstein-Barr virus (EBV).
  • We report two patients with blastic NK cell lymphoma and describe the interesting clinical studies.
  • The patients presented with cutaneous plaques.
  • Unlike in many NK and NK-like T-cell disorders, azurophilic cytoplasmic granules were absent.
  • In addition, the cells were positive for HLA-DR, CD2, CD4, CD56 and TdT, and negative for EBV transcripts.
  • In spite of the advanced clinical stage, complete remission was achieved by conventional chemotherapy.
  • After interleukin 2 expansion of tumour-infiltrating bone marrow and lymph node cells from the patients, cytotoxic T-cell lines with rearranged T-cell receptor genes were established.
  • They showed specific killing activity against autologous tumour cells in an MHC-restricted fashion, with possible implications for treatment.
  • In addition, upon cessation of maintenance chemotherapy, one patient developed overt leukaemia with blasts expressing CD33 antigens, suggesting a continuous spectrum of blastic NK cell lymphoma to myeloid/NK cell precursor acute leukaemia.
  • [MeSH-major] Killer Cells, Natural / immunology. Precursor Cell Lymphoblastic Leukemia-Lymphoma / immunology
  • [MeSH-minor] Aged. Antigens, CD. Antigens, CD2. Antigens, Differentiation, Myelomonocytic. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Blotting, Southern. CD4-Positive T-Lymphocytes / immunology. Cyclophosphamide / therapeutic use. Cytotoxicity Tests, Immunologic. Doxorubicin / therapeutic use. Female. Flow Cytometry. Gene Rearrangement, T-Lymphocyte. Humans. Immunophenotyping. Karyotyping. Male. Middle Aged. Prednisone / therapeutic use. Remission Induction. Sialic Acid Binding Ig-like Lectin 3. Vincristine / therapeutic use

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  • (PMID = 11328295.001).
  • [ISSN] 0007-1048
  • [Journal-full-title] British journal of haematology
  • [ISO-abbreviation] Br. J. Haematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / Antigens, CD2; 0 / Antigens, Differentiation, Myelomonocytic; 0 / CD33 protein, human; 0 / Sialic Acid Binding Ig-like Lectin 3; 5J49Q6B70F / Vincristine; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; VB0R961HZT / Prednisone; CHOP protocol
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22. Chamaillard M, Beylot-Barry M, Viallard JF, Cogrel O, Vergier B, Pellegrin JL, Beylot C: [Agranular CD4+ CD56+ hematodermic neoplasm: a new case report]. Ann Dermatol Venereol; 2004 Dec;131(12):1074-6
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  • [Title] [Agranular CD4+ CD56+ hematodermic neoplasm: a new case report].
  • [Transliterated title] Hématodermie CD4+ CD56+. Une nouvelle observation.
  • BACKGROUND: "Agranular CD4+ CD56+ hematodermic neoplasm" are rare hematologic neoplasms which were recently shown to correspond to the plasmocytoid dendritic cells.
  • The biopsy has shown a dense dermal infiltration with malignant cells CD4+ CD56+ CD43+.
  • A chemotherapy permitted a clinical remission after six courses.
  • After a short success of chemotherapy by DHAP, the patient died three month later.
  • DISCUSSION: "Agranular CD4+ CD56+ hematodermic neoplasm" is a distinct entity from the cutaneous primary lymphomas.
  • Recently plasmocytoid monocyte cells have been identified as the precursor of the malignant population with the high expression of CD123, IL3 receptor.
  • It is a distinct clinicopathologic entity by its clinical presentation with skin tropism, bone marrow involvement with or without leukemic phase and poor prognosis independent of the kind of treatment and its particular phenotype CD4+ CD56+ CD43+.
  • It would be interesting to use antibodies linked to CD123 in therapeutic because any treatment have efficacity in this disease.
  • [MeSH-major] Antigens, CD4. Antigens, CD56. Skin Neoplasms

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  • (PMID = 15692441.001).
  • [ISSN] 0151-9638
  • [Journal-full-title] Annales de dermatologie et de vénéréologie
  • [ISO-abbreviation] Ann Dermatol Venereol
  • [Language] fre
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] France
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56
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23. Kimura S, Kakazu N, Kuroda J, Akaogi T, Hayashi H, Nishida K, Abe T: Agranular CD4+CD56+ blastic natural killer leukemia/lymphoma. Ann Hematol; 2001 Apr;80(4):228-31
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  • [Title] Agranular CD4+CD56+ blastic natural killer leukemia/lymphoma.
  • Blastic natural killer cell leukemia/lymphoma (blastic NKL/L) is characterized by blastic morphology and a distinctive immunophenotype combining blastic features and cytologically resembling acute myeloid or lymphoid leukemia.
  • The clinical, pathologic, and cytogenetic features of blastic NKL/L have not yet been systematically identified.
  • We report herein a case of blastic NKL/L with skin lesion, adenopathy, and systemic lymphoadenopathy.
  • The identified tumor cells were positive for CD4 and CD56, and negative for T-cell, B-cell, and myeloid markers.
  • T-cell receptor beta, gamma, delta, and immunoglobulin heavy chain genes in the bone marrow cells showed germ-line configurations.
  • Southern blot analysis with a terminal probe did not reveal any Epstein-Barr virus infection.
  • Although patients diagnosed as blastic NKL/L have generally shown chemotherapy resistance and poor prognosis, our patient was treated with a combined chemotherapy, which is also used for acute lymphoblastic leukemia, and has maintained complete remission (CR) for more than 13 months.
  • [MeSH-major] Antigens, CD4 / analysis. Antigens, CD56 / analysis. Killer Cells, Natural. Lymphoma / diagnosis. Precursor Cell Lymphoblastic Leukemia-Lymphoma / diagnosis
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Chromosome Banding. Chromosomes, Human, Pair 13. Chromosomes, Human, Pair 14. Humans. Immunoglobulin Heavy Chains / analysis. In Situ Hybridization, Fluorescence. Karyotyping. Male. Nucleic Acid Hybridization. Receptors, Antigen, T-Cell / analysis. Remission Induction. Translocation, Genetic

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  • (PMID = 11401089.001).
  • [ISSN] 0939-5555
  • [Journal-full-title] Annals of hematology
  • [ISO-abbreviation] Ann. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56; 0 / Immunoglobulin Heavy Chains; 0 / Receptors, Antigen, T-Cell
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24. Kim Y, Kang MS, Kim CW, Sung R, Ko YH: CD4+CD56+ lineage negative hematopoietic neoplasm: so called blastic NK cell lymphoma. J Korean Med Sci; 2005 Apr;20(2):319-24
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  • [Title] CD4+CD56+ lineage negative hematopoietic neoplasm: so called blastic NK cell lymphoma.
  • Blastic natural killer (NK) cell lymphoma is a rare neoplasm characterized by blastoid tumor cells expressing CD4 and CD56, with predominant skin involvement.
  • Although this tumor has been regarded as a neoplasm related to NK cell, recent studies suggested that it is derived from plasmacytoid dendritic cells, but not from NK cell.
  • Herein we report 4 cases of CD4+CD56+ lineage marker-blastic NK cell lymphomas with a review of literatures.
  • All four tumors were CD4+ and CD56+.
  • Lineage specific markers for B- and T cell were negative.
  • T-cell receptor gene rearrangement, EBV, CD13 and CD33 were negative.
  • In one patient, tumor cells arranged in Homer-Wright type pseudorosette and expressed terminal deoxynucleotidyl transferase(TdT).
  • Despite the standard lymphoma chemotherapy, the tumors, except one lost during follow-up, progressed and relapsed.
  • The patients died 8-60 months after diagnosis.
  • [MeSH-major] Antigens, CD4 / analysis. Antigens, CD56 / analysis. Killer Cells, Natural / pathology. Lymphoma, T-Cell / pathology
  • [MeSH-minor] Adolescent. Cell Lineage. Female. Humans. Male. Middle Aged

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  • (PMID = 15832009.001).
  • [ISSN] 1011-8934
  • [Journal-full-title] Journal of Korean medical science
  • [ISO-abbreviation] J. Korean Med. Sci.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Korea (South)
  • [Chemical-registry-number] 0 / Antigens, CD4; 0 / Antigens, CD56
  • [Other-IDs] NLM/ PMC2808614
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25. Giagounidis AA, Heinsch M, Haase S, Aul C: Early plasmacytoid dendritic cell leukemia/lymphoma coexpressing myeloid antigenes. Ann Hematol; 2004 Nov;83(11):716-21
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  • [Title] Early plasmacytoid dendritic cell leukemia/lymphoma coexpressing myeloid antigenes.
  • Early plasmacytoid dendritic cell (pDC) leukemia/lymphoma has recently been described as a CD4(+)CD56(+) lineage negative malignancy with characteristic clinical, morphologic, immunophenotypic, and biological features.
  • We present a case of a 72-year-old man who was diagnosed with isolated skin involvement 30 months ago and received numerous chemotherapy cycles that did not prevent three relapses of the disease, the last two involving the bone marrow.
  • Flow cytometry showed CD4(+), CD56(+), CD45(+), CD38(+), HLA-DR(+), CD33(+), CD123(+), CD2(-), cyCD3(-), CD7(-), CD10(-), CD11b(-), CD13(-), CD14(-), CD16(-), CD19(-), cyCD22(-), CD24(-), CD34(-), CD57(-), CD61(-), CD64(-), CD65(-), cyCD79a(-), CD117(-), MPO(-), and TdT(-) population.
  • Our patient was initially treated with acute myeloid leukemia-type chemotherapy, later he was given acute lymphoblastic leukemia-type treatment, and at the last relapse he received CHOP chemotherapy.
  • Each treatment led to rapid response of tumor manifestations with disease-free intervals of 7 months, 9 months, and 8 months, respectively.
  • Although patients usually have an ominous prognosis, with only 25% living more than 24 months, our patient is alive after 30+ months and has again achieved complete remission after the last chemotherapy.
  • [MeSH-major] Antigens, CD / metabolism. Dendritic Cells / metabolism. Leukemia, Plasma Cell / metabolism. Skin Neoplasms / metabolism. Skin Neoplasms / secondary
  • [MeSH-minor] Aged. Bone Marrow / metabolism. Bone Marrow / pathology. Humans. Male. Neoplasm Staging

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  • (PMID = 15316755.001).
  • [ISSN] 0939-5555
  • [Journal-full-title] Annals of hematology
  • [ISO-abbreviation] Ann. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antigens, CD
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26. Barrionuevo C, Zaharia M, Martinez MT, Taxa L, Misad O, Moscol A, Sarria G, Guerrero I, Casanova L, Flores C, Zevallos-Giampietri EA: Extranodal NK/T-cell lymphoma, nasal type: study of clinicopathologic and prognosis factors in a series of 78 cases from Peru. Appl Immunohistochem Mol Morphol; 2007 Mar;15(1):38-44
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  • [Title] Extranodal NK/T-cell lymphoma, nasal type: study of clinicopathologic and prognosis factors in a series of 78 cases from Peru.
  • It is well known that extranodal NK/T-cell lymphoma (NK/TCL) nasal type clusters in Asian countries.
  • A large series of 78 cases of nasal NK/TCL from Peru is analyzed in the present study.
  • Two histologic groups 1 (monomorphic) and 2 (polymorphic), were segregated according to the proportion of large cells (above and below 30%, respectively).
  • Epstein-Barr virus (EBV) sequences and types were investigated using polymerase chain reaction.
  • Clinical characteristics, stage, outcome, and response to treatment were evaluated in both groups.
  • Both groups showed CD3c+ CD2+ CD56+ CD3s- CD20- immunophenotype.
  • In this series type-2 EBV was found more frequent than type-1 EBV, contrarily to that observed in Asian series.
  • However, about one-third of cases simultaneously harbored both viral types.
  • Both groups received an average of 50-Gy dose of radiation therapy (RT), with or without chemotherapy.
  • Complete therapeutic response was achieved in 89% of group 1 and in 74% of group 2, but this difference was not statistically significant.
  • The overall survival, in both groups, was longer for patients treated with RT alone compared with those treated with combined RT therapy and chemotherapy.
  • The present study has shown that dividing nasal NK/TCL in monomorphic and polymorphic variants, according to frequency of large cells, does not correlate with clinical and prognostic factors.

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  • (PMID = 17536305.001).
  • [ISSN] 1541-2016
  • [Journal-full-title] Applied immunohistochemistry & molecular morphology : AIMM
  • [ISO-abbreviation] Appl. Immunohistochem. Mol. Morphol.
  • [Language] ENG
  • [Publication-type] Journal Article
  • [Publication-country] United States
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