{"title":"Isolated Central Nervous System Involvement after Brentuximab Vedotin Treatment for HIV-Positive ALK-Negative Anaplastic Large Cell Lymphoma.","authors":"Takuya Suyama, Kumiko Matsui, Kosuke Makihara, Masatoshi Tsuru","doi":"10.1155/2024/5534556","DOIUrl":"10.1155/2024/5534556","url":null,"abstract":"<p><p>Human immunodeficiency virus (HIV)-associated lymphoma poses a high mortality risk despite antiretroviral therapy (ART). Although intermediate- or high-grade B-cell lymphomas are common, anaplastic large-cell lymphomas (ALCLs) are rare and seldom affect the central nervous system (CNS). Herein, we present a case of HIV-associated ALCL with isolated CNS involvement that occurred following the discontinuation of ART that was administered after treatment with brentuximab vedotin (BV)-which does not cross the blood-brain barrier. At the time of CNS recurrence, the patient's CD4 count was 9 cells/mm<sup>3</sup>. This is the first report of CNS recurrence in HIV-associated ALCL. Considering the high risk of CNS relapse, we suggest initiating CNS prophylaxis in cases of HIV-associated ALCL, particularly in patients receiving CNS-impermeable agents such as BV.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2024 ","pages":"5534556"},"PeriodicalIF":0.7,"publicationDate":"2024-02-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10904676/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140022946","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
D. Rieke, L. Schmalbrock, J. Ihlow, Karsten Kleo, Ann-Christin von Brünneck, Florian Nolte, Ulrich Keller, Sebastian Ochsenreither
{"title":"Renal Extramedullary Hematopoiesis in Mast Cell Leukemia with Bone Marrow Fibrosis","authors":"D. Rieke, L. Schmalbrock, J. Ihlow, Karsten Kleo, Ann-Christin von Brünneck, Florian Nolte, Ulrich Keller, Sebastian Ochsenreither","doi":"10.1155/2024/3502887","DOIUrl":"https://doi.org/10.1155/2024/3502887","url":null,"abstract":"Systemic mastocytosis is defined by the clonal proliferation of abnormal mast cells. The clinical course can range from indolent forms with normal life expectancy to advanced mast cell leukemia with dismal prognosis. An association with other diseases, including myeloproliferative neoplasia, has been described. We present a case of a 75-year patient with a history of cutaneous mastocytosis who was diagnosed with mast cell leukemia more than 9 years ago and did not receive treatment. The patient presented to our clinic with acute kidney failure because of renal extramedullary hematopoiesis. Bone marrow histopathology revealed extensive fibrosis and 50% infiltration by mast cells with a c-KIT D816V mutation. No mutations supporting primary myelofibrosis were identified. Treatment with midostaurin was started, and the patient was discharged after improvement of renal function. Here, we discuss diagnostic challenges between different forms of mast cell leukemia and overlaps with other hematological malignancies.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"37 2","pages":""},"PeriodicalIF":0.7,"publicationDate":"2024-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139388327","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kritika Krishnamurthy, Jui Choudhuri, K H Ramesh, Yanhua Wang
{"title":"MPO Expression of Background Neutrophils in MPO Negative Acute Promyelocytic Leukemia, An Easy Clue to Corroborate a Challenging Diagnosis: A Case Report and Review of Literature.","authors":"Kritika Krishnamurthy, Jui Choudhuri, K H Ramesh, Yanhua Wang","doi":"10.1155/2023/7979261","DOIUrl":"10.1155/2023/7979261","url":null,"abstract":"<p><p>Acute promyelocytic leukemia (APL) is characterized by the pathogenic driver fusion transcript PML-RARA resulting from the t(15;17) translocation. Early recognition of APL with prompt ATRA induction has a decisive impact on the early death rate. The preliminary diagnosis of APL relies heavily on cytomorphology and flow cytometry. In APL with variant morphology, such as the microgranular variant, immunophenotype, especially the bright MPO positivity is the basis of diagnosis. Till date, only five cases of APL with reduced/absent MPO have been described in literature. The identification of MPO deficiency based on genetic testing would involve at the least a MPO gene scanning with NGS, followed by microarray to identify somatic uniparental disomy in heterozygotes. This testing is not only redundant given the scant clinical implications of heterozygous MPO deficiency but also time consuming. An easy way to identify background MPO deficiency confounding the immunophenotype of a myeloid neoplasm is the MPO expression in background neutrophils gated on the initial flow cytometry. A dim MPO in the background neutrophils, in the morphological setting of APL, can identify underlying MPO deficiency, clarifying the immunophenotypic ambiguity and thus establishing an unequivocal diagnosis as seen in the current case.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"7979261"},"PeriodicalIF":0.7,"publicationDate":"2023-12-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10761215/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139088969","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Moeen Mohammadi-Oroujeh, Ansa Mehreen, David L Grinblatt
{"title":"Epstein-Barr Virus Driven Hodgkin's Lymphoma after a Short Course of Daratumumab Treatment for Relapsed Multiple Myeloma.","authors":"Moeen Mohammadi-Oroujeh, Ansa Mehreen, David L Grinblatt","doi":"10.1155/2023/6669174","DOIUrl":"10.1155/2023/6669174","url":null,"abstract":"<p><p>In this case, we describe the potential risk of developing an infectious complication leading to a secondary malignancy after a short course of immunotherapy. We report a patient who presented with Epstein-Barr virus (EBV) driven Hodgkin's lymphoma after treatment with a short course of daratumumab along with pomalidomide and dexamethasone for relapsed multiple myeloma. Although there have been limited documented cases of daratumumab treatment leading to EBV reactivation, in patients presenting with infectious symptoms or neutropenia on a daratumumab-based regimen, testing for EBV should not be overlooked.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"6669174"},"PeriodicalIF":0.7,"publicationDate":"2023-12-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10749716/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139038053","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kim Abbegail Tan Aldecoa, Chef Stan L Macaraeg, Akash Dadlani, Sri Yadlapalli
{"title":"Spontaneous Hyphema during Ibrutinib Treatment in a CLL Patient.","authors":"Kim Abbegail Tan Aldecoa, Chef Stan L Macaraeg, Akash Dadlani, Sri Yadlapalli","doi":"10.1155/2023/1691996","DOIUrl":"10.1155/2023/1691996","url":null,"abstract":"<p><p>Ibrutinib is an oral, first-line, targeted therapy for chronic lymphocytic leukemia (CLL). Commonly reported adverse events are diarrhea, fatigue, and musculoskeletal pain, but rarely it has been associated with visual disturbances. Here, we present a rare case of spontaneous hyphema in a 60-year-old patient with a known diagnosis of CLL on ibrutinib treatment.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"1691996"},"PeriodicalIF":0.7,"publicationDate":"2023-12-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10735723/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138832215","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Circulating Inhibitor against Factor X: A Rare Cause of Hemorrhagic Diathesis.","authors":"P Rossignon, F Grandjean, A Claessens, N Weynants","doi":"10.1155/2023/5510654","DOIUrl":"https://doi.org/10.1155/2023/5510654","url":null,"abstract":"<p><p>Acquired coagulopathies resulting from factor X deficiency are rare and typically associated with amyloidosis or plasma cell dyscrasia. Factor X plays a pivotal role in the coagulation cascade, converting prothrombin into thrombin and facilitating the formation of fibrinogen and thrombus. While its occurrence following common infections is extremely rare, isolated cases have been documented. We present a rare case of bleeding diathesis in a patient with community-acquired pneumonia, where prolonged activated partial thromboplastin time (aPTT) and prothrombin time (PT) led to the diagnosis of an infectious-triggered acquired circulating inhibitor targeting factor X. Prompt treatment with methylprednisolone effectively controlled the inhibitor without recurrence. This case report provides insights into the diagnostic strategies, differential algorithm, and therapeutic approaches for managing this rare coagulopathy.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"5510654"},"PeriodicalIF":0.7,"publicationDate":"2023-12-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10732816/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138832213","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Gilteritinib Monotherapy as a Transplant Bridging Option for a Patient with <i>FLT3</i>-Mutated Acute Promyelocytic Leukemia Who Developed a Second Relapse after All-Trans Retinoic Acid + Chemotherapy, Arsenic Trioxide, and High-Dose Cytarabine Therapy.","authors":"Hirofumi Kobayashi, Hiroki Tsutsumi, Yukiko Misaki, Takashi Maekawa, Naoko Inoshita, Machiko Kawamura, Nobuo Maseki","doi":"10.1155/2023/8568587","DOIUrl":"https://doi.org/10.1155/2023/8568587","url":null,"abstract":"<p><p>We report a case of FLT3-mutated APL who developed disease relapse despite all-trans retinoic acid (ATRA) + chemotherapy, and re-induction chemotherapy with arsenic trioxide (ATO) and high-dose (HD) cytarabine (Ara-C) therapy failed to yield complete remission. Because the leukemic cells were resistant to all the aforementioned therapies, we started the patient on monotherapy with gilteritinib, a selective FLT3-inhibitor, as an alternative re-induction treatment option rather than further intensive chemotherapy. The patient showed complete hematologic remission in response to this therapy. This case serves as supporting evidence for the use of single-agent therapy with gilteritinib as a bridge to transplantation in patients with refractory <i>FLT3</i>-mutated APL.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"8568587"},"PeriodicalIF":0.7,"publicationDate":"2023-12-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10732830/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138832214","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Junaid Ahmad Wali, Brian M Quigley, Beverly Schaefer
{"title":"Successful Use of Bortezomib in an Adolescent with Refractory TTP.","authors":"Junaid Ahmad Wali, Brian M Quigley, Beverly Schaefer","doi":"10.1155/2023/8173903","DOIUrl":"10.1155/2023/8173903","url":null,"abstract":"<p><p>With increasing early and upfront use of rituximab and caplacizumab in the modern management of immune-mediated thrombotic thrombocytopenic purpura (iTTP), the risk of refractory disease is expected to decline. However, despite the use of adequate initial therapy, a small subset of patients develop a refractory disease which is difficult to manage. Bortezomib has come to be known as a safe and effective treatment option for refractory iTTP, but its use in children is limited. Here, we describe the case of an adolescent patient with refractory iTTP who had a satisfactory and sustained response to the use of bortezomib.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"8173903"},"PeriodicalIF":0.7,"publicationDate":"2023-11-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10693465/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138478919","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Christopher B. Ryder, Hayder Saeed, Mohammad Hussaini
{"title":"Composite Lymphoma with Follicular Lymphoma Transformation to Clonally Related Epstein–Barr Virus (EBV) Positive Diffuse Large B-Cell Lymphoma and EBV-PositiveClassic Hodgkin Lymphoma","authors":"Christopher B. Ryder, Hayder Saeed, Mohammad Hussaini","doi":"10.1155/2023/8833273","DOIUrl":"https://doi.org/10.1155/2023/8833273","url":null,"abstract":"While the Epstein–Barr virus (EBV) is known to drive de novo lymphomagenesis, it may rarely contribute to transformation of indolent lymphoma as well. Some EBV-related lymphomas represent a diagnostic challenge with important prognostic and therapeutic implications. We describe a case of follicular lymphoma (FL) transformation to both EBV + diffuse large B-cell lymphoma (DLBCL) and EBV + classic Hodgkin lymphoma (cHL), the latter of which was only identified retrospectively after selective outgrowth during DLBCL therapy. Finally, we describe successful salvage therapy with brentuximab vedotin plus nivolumab. This is the first known case of composite lymphoma with FL, EBV + DLBCL, and EBV + cHL within a single lymph node. The disease course highlights the importance of careful morphologic examination and comprehensive immunophenotypic characterization of EBV + lymphomas to ensure proper clinical care and underscores the potential for novel therapies currently under investigation. This trial is registered with NCT01671813.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":" 8","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-11-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135341287","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Bhvaneet Jhajj, Ryan Henrie, Youness El-Khalidy, Habib Moshref Razavi
{"title":"A Case of Acute Myeloid Leukemia Mimicking Blastic Plasmacytoid Dendritic Cell Neoplasm: Utility of the Proposed Upcoming WHO-5 Diagnostic Criteria.","authors":"Bhvaneet Jhajj, Ryan Henrie, Youness El-Khalidy, Habib Moshref Razavi","doi":"10.1155/2023/5014728","DOIUrl":"10.1155/2023/5014728","url":null,"abstract":"<p><p>Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is an aggressive hematologic malignancy which is associated with a distinctive morphologic appearance. However, the morphology is not specific, and diagnostic characterization requires integration of immunophenotypic and genetic testing. We herein report a case of a 35-year-old female patient who presented with worsening cytopenia. A bone marrow aspirate identified medium-sized blastic cells with perinuclear microvacuoles (\"pearl neckless\"). Occasional blasts demonstrated a \"hand mirror\" appearance. Tandem flow cytometry showed an atypical population of dim CD45 events with expression of CD4, CD56, CD117, CD123, and monocytic markers such as CD64. Fluorescence in situ hybridization (FISH) showed evidence of a KMT2A rearrangement with an unknown partner on chromosome 19. Expression of MPO and muramidase was present. The final diagnosis was acute monocytic leukemia (AMoL). Due to the overlapping features of acute myeloid leukemia and BPDCN, the 5<sup>th</sup> Edition of the World Health Organization (WHO) Classification of Haematolymphoid Tumours provides new criteria for the diagnosis of BPDCN. Our case highlights the utility of these criteria.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2023 ","pages":"5014728"},"PeriodicalIF":0.7,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10632061/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"72016718","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}