Case Reports in Hematology最新文献

{"title":"Metachronous B-Cell Acute Lymphoblastic Leukemia After Localized Neuroblastoma Treated Without Cytotoxic Therapy: A Case Report.","authors":"Reihaneh Sedghi, Ahmad Nazar Zadeh, Mohammadreza Bordbar, Omid Reza Zekavat, Shirin Parand","doi":"10.1155/crh/5543785","DOIUrl":"10.1155/crh/5543785","url":null,"abstract":"<p><p>A 7-month-old infant developed therapy-independent acute lymphoblastic leukemia (ALL) 8 months after complete resection of an International Neuroblastoma Risk Group (INRG) Stage L1 adrenal neuroblastoma (N-MYC nonamplified) treated with surgery alone. The patient presented with leukemia cutis and discordant bone marrow findings: the initial aspirate revealed > 90% lymphoblasts (CD45+/CD19+/CD10+/TdT+), whereas two subsequent marrow examinations showed normocellular hematopoiesis, suggesting spatially heterogeneous marrow involvement with predominant extramedullary disease. Skin biopsy confirmed leukemic infiltration (PAX5+/TdT+/CD34+/CD10-). Germline testing was not available. Standard-risk chemotherapy achieved complete remission, and the patient is currently receiving maintenance therapy. This case highlights that second primary ALL can occur in the absence of cytotoxic therapy after neuroblastoma and underscores the need for long-term surveillance and consideration of genetic evaluation in selected patients.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5543785"},"PeriodicalIF":0.7,"publicationDate":"2026-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13201254/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148044113","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":"Hydroxyurea-Associated Wunderlich Syndrome in Triple-Negative Myelofibrosis: A Case Report and Literature Review.","authors":"Veysel Erol, Zeki Güzel, İbrahim Ethem Akgün, Gökhan Ecer","doi":"10.1155/crh/5518867","DOIUrl":"10.1155/crh/5518867","url":null,"abstract":"<p><strong>Background: </strong>Wunderlich syndrome (WS) is a spontaneous, nontraumatic intrarenal or perirenal hemorrhage most commonly related to renal neoplasms, vascular disorders, infection, cyst rupture, or anticoagulation. Clinical severity ranges from self-limited bleeding to hemorrhagic shock.</p><p><strong>Case: </strong>We report a 58-year-old woman with triple-negative myelofibrosis (MF) who was started on hydroxyurea (HU) 500 mg/day and low-dose acetylsalicylic acid (ASA). On Day 60, she presented with bilateral perirenal and intrarenal hemorrhage confirmed by contrast-enhanced abdominal computed tomography. ASA was discontinued and bleeding ceased spontaneously. One month later, she represented with recurrent bilateral hemorrhage without ASA exposure and marked neutrophilic leukocytosis. Concomitant purpuric skin lesions, positive antinuclear antibodies (ANA, 1:160), and low C3 levels raised suspicion for a HU-associated vasculitic process; HU was discontinued. Coagulation parameters at the time of bleeding (INR: 1.2, aPTT: 26.4 s, fibrinogen: 348 mg/dL, and D-dimer: 0.6 μg/mL) were not consistent with disseminated intravascular coagulation. Despite supportive management, the patient's condition deteriorated and she died shortly thereafter. The exact cause of death could not be definitively determined.</p><p><strong>Conclusion: </strong>To our knowledge, this is the first reported case of WS in a patient with MF receiving HU therapy. Clinicians should consider WS in HU-treated patients presenting with acute flank pain or hematuria and evaluate for potential vasculitic features.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5518867"},"PeriodicalIF":0.7,"publicationDate":"2026-05-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13191779/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148018094","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":"Brain Abscess Following CPX-351 Treatment in a Patient With Acute Myeloid Leukemia.","authors":"Yusuke Yamaga, Shoichiro Okazaki, Yuri Sakuma, Daimon Siraisi, Takahiro Nishiyama","doi":"10.1155/crh/3668400","DOIUrl":"10.1155/crh/3668400","url":null,"abstract":"<p><p>We describe a rare case of a brain abscess occurring during CPX-351 induction therapy for acute myeloid leukemia (AML). The patient experienced recurrent febrile neutropenia during the myelosuppression period and developed left-sided spatial neglect during hematologic recovery. Brain magnetic resonance imaging revealed a solitary abscess in the right parietal lobe. Burr hole drainage yielded <i>Bacillus</i> species, and subsequent treatment with vancomycin and linezolid, followed by endoscopic abscess resection, led to gradual neurological recovery. Complete remission of AML was achieved, and consolidation therapy with venetoclax plus azacitidine was initiated. CPX-351 is associated with prolonged myelosuppression and relatively low gastrointestinal toxicity, which may reduce the risk of Gram-negative sepsis but increase susceptibility to Gram-positive infections, such as catheter-related bacteremia. Clinicians should consider the possibility of a brain abscess in patients presenting with neurological symptoms during AML treatment, as early diagnosis and prompt surgical and antimicrobial management are critical for favorable outcomes.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"3668400"},"PeriodicalIF":0.7,"publicationDate":"2026-05-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13194186/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148018078","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":"Extranodal Male Genital Involvement With Diffuse Large B-Cell Lymphoma.","authors":"James J Fradin, Kavanya Feustel, Mehdi Hamadani, Thomas A Giever","doi":"10.1155/crh/5511675","DOIUrl":"10.1155/crh/5511675","url":null,"abstract":"<p><p>Primary penile diffuse large B-cell lymphoma (DLBCL) is an exceptionally rare extranodal presentation that can be challenging to diagnose due to its nonspecific symptoms. We report the case of an 84-year-old male who initially presented with phimosis who subsequently developed a nonhealing surgical wound and B symptoms. Imaging and biopsy ultimately revealed Stage IVB activated B-cell type DLBCL. He was treated with dose-reduced rituximab, cyclophosphamide, doxorubicin, prednisone, and polatuzumab vedotin (modified R-mini-CHOP). Despite his advanced disease, he achieved a partial metabolic response with negative minimal residual disease. We also summarize prior reported cases of penile DLBCL, highlighting patterns in disease presentation and treatment approaches. This case emphasizes the importance of maintaining a broad differential for penile lesions to avoid delays in diagnosis and treatment, and it illustrates the integration of novel agents such as polatuzumab vedotin for frail or elderly patients.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5511675"},"PeriodicalIF":0.7,"publicationDate":"2026-05-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13189451/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147989455","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":"Diffuse Large B-Cell Lymphoma Presenting in a Background of Rosai-Dorfman Disease.","authors":"Chao Wu, Anders Meyer, Aung M Tun","doi":"10.1155/crh/6560267","DOIUrl":"10.1155/crh/6560267","url":null,"abstract":"<p><p>Rosai-Dorfman disease (RDD) can present with lymphadenopathy. However, it is important to note that RDD can also coexist with hematologic malignancies, including non-Hodgkin lymphomas. We present a case of an older male patient with generalized lymphadenopathy who was first diagnosed with RDD. High-dose steroid therapy was rendered without a satisfactory response. Thus, the tissue specimens were re-examined, and the patient was found to have diffuse large B-cell lymphoma (DLBCL) in the background of RDD. The laboratory tests are notable for a slightly elevated serum lactate dehydrogenase (LDH) level. A positron emission tomography-computed tomography scan revealed generalized hypermetabolic lymphadenopathy above and below the diaphragm. A bone marrow biopsy showed normocellular marrow without evidence of DLBCL. The international prognostic index score was 3 (age > 60 years, elevated serum LDH level, and Stage III disease), indicating a high-intermediate risk disease. The patient was treated with six cycles of golcadomide in combination with R-Pola-CHP (rituximab, polatuzumab vedotin, cyclophosphamide, doxorubicin, and prednisone) for the underlying DLBCL, with resolution of both DLBCL and RDD. The disease remained in remission at the follow-up visit 9 months after completing the therapy. This case highlights the importance of recognizing underlying DLBCL in patients with RDD for optimal patient outcomes. <b>Trial Registration:</b> ClinicalTrials.gov identifier: NCT04884035.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"6560267"},"PeriodicalIF":0.7,"publicationDate":"2026-05-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13180685/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147976312","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":"The First Reported Case of VEXAS Syndrome in Lebanon: Efficacy of Azacitidine as a Therapeutic Option-Case Report.","authors":"Roudy Issa, Bassem Akiki, Sophie Georgin-Lavialle, Olivier Kosmider, Danielle Hammond, Marcel Massoud, Vicky Najjar, Elissar Dagher","doi":"10.1155/crh/3903470","DOIUrl":"10.1155/crh/3903470","url":null,"abstract":"<p><strong>Background: </strong>Vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome is a rare, adult-onset autoinflammatory disease that has only been described since late 2020. Given the rarity of the disease and the absence of established treatment guidelines, management remains challenging and largely based on clinical experience and case reports.</p><p><strong>Case presentation: </strong>This paper reports the first documented case of VEXAS syndrome in Lebanon. A 73-year-old man presented with fever, severe asthenia, erythematous skin lesions, and anemia, initially diagnosed as giant cell arteritis. Following a diagnosis of VEXAS syndrome, he was treated with corticosteroids and methotrexate but developed refractory anemia and required erythropoietin therapy. Tocilizumab was introduced to manage inflammation, but the patient's condition remained challenging due to corticosteroid dependence and myelodysplastic syndrome (MDS). Given the patient's comorbidities and intermediate-risk MDS, azacitidine was initiated as a therapeutic option. Despite initial neutropenia and infections, adjustments to the azacitidine regimen led to significant clinical and hematologic improvements. The patient achieved complete remission, became transfusion-independent, and maintained stable hemoglobin levels.</p><p><strong>Conclusion: </strong>This case highlights the efficacy of azacitidine in managing VEXAS syndrome with MDS, particularly among patients ineligible for hematopoietic stem cell transplantation, offering a potential pathway to sustained remission and reduced corticosteroid dependence.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"3903470"},"PeriodicalIF":0.7,"publicationDate":"2026-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13168939/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147942862","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":"A Rare Presentation of Mixed Warm and Cold Autoimmune Hemolytic Anemia.","authors":"Onyekachi Anya, Oluchi Idenyi, Ogbonna Chikere, Joseph J Shatzel","doi":"10.1155/crh/8805562","DOIUrl":"10.1155/crh/8805562","url":null,"abstract":"<p><p>Mixed autoimmune hemolytic anemia (mAIHA) is a rare clinical condition where both warm and cold antibodies lead to autoimmune red cell destruction and progressive anemia. Herein, we report a rare case of combined warm and cold agglutinin-mediated autoimmune hemolytic anemia in a 61-year-old male who initially presented with incidental macrocytic anemia during a preoperative evaluation for pterygium surgery. Investigations showed the presence of both warm (IgG) and cold (IgM) autoantibodies, evidenced by a positive direct Coombs test (both anti-IgG and anti-C3), and elevated serum IgM levels. Type and cross-isolate revealed two distinct autoantibodies with different thermal amplitudes. Further workup did not reveal a definitive underlying etiology for the hemolysis. Mycoplasma pneumoniae and Epstein-Barr virus antibody panels were not suggestive of active infection. He was treated with corticosteroids and rituximab, resulting in an improvement in his hemoglobin levels, a reduction in splenomegaly, and ultimately remission of his autoimmune hemolytic anemia. This case emphasizes the need for a thorough workup to identify the cause of anemia, even in the context of seemingly unrelated surgical procedures.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8805562"},"PeriodicalIF":0.7,"publicationDate":"2026-05-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13161755/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147934407","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":"A Case of Acute Myelogenous Leukemia in Long-Term Remission After Severe Infection Following Only One Course of Chemotherapy.","authors":"Motoharu Shibusawa","doi":"10.1155/crh/5567757","DOIUrl":"10.1155/crh/5567757","url":null,"abstract":"<p><p>Acute myelogenous leukemia (AML) is characterized by the uncontrolled growth of tumor cells in the bone marrow. Standard treatment includes induction remission therapy followed by several courses of consolidation chemotherapy. The 5-year disease-free survival rate is only 39%. A 61-year-old man with AML achieved remission after standard induction therapy but experienced severe myelosuppression and a serious infection, leaving him too exhausted for further chemotherapy. Remarkably, he remained in remission for over 5 years. This case suggests that both the initial chemotherapy and the immune response triggered by severe infection may have contributed to long-term remission.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5567757"},"PeriodicalIF":0.7,"publicationDate":"2026-05-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13161994/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147934391","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":"Patient With MGRS/PGNMID Without Detection of a Peripheral Clone: Case Report and Literature Review.","authors":"Agatha Larrazábal, Ana Ávila-Rodriguez, Matías Sánchez","doi":"10.1155/crh/8043845","DOIUrl":"10.1155/crh/8043845","url":null,"abstract":"<p><p>Monoclonal gammopathy of renal significance (MGRS) is a clonal cell proliferative disorder, characterized by the production of monoclonal immunoglobulins in patients that do not meet hematological criteria for a specific malignancy. It can be present in B cell and plasma cell clonal proliferative diseases and accounts for 10% of monoclonal gammopathy of undetermined significance (MGUS) cases. We present a case of a patient presenting with acute kidney injury, hematuria, and nephrotic syndrome, who after renal biopsy was diagnosed with a proliferative glomerulonephritis with monoclonal immunoglobulin deposits (PGNMIDs), a subtype of MGRS, and treated with chemoimmunotherapy (daratumumab, cyclophosphamide, bortezomib, and dexamethasone), with complete renal function recovery.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8043845"},"PeriodicalIF":0.7,"publicationDate":"2026-05-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13158363/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147934447","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":"Leukemia Cutis at Initial Diagnosis of Acute Myeloid Leukemia Consistent With Therapy-Related Disease After Lung Cancer Chemotherapy: A Case Report.","authors":"Adam Bowen, Nicholas DiLoreto, Maha Bayya, Robert McDonald, Daniel Isaac","doi":"10.1155/crh/1198642","DOIUrl":"10.1155/crh/1198642","url":null,"abstract":"<p><strong>Introduction: </strong>Leukemia cutis (LC) is characterized by dermal infiltration of leukemic blasts and typically occurs in patients with known leukemia. Rarely, LC is identified at the time of initial diagnosis of systemic disease, especially acute myeloid leukemia (AML). We describe a case of LC identified at the initial diagnosis of AML consistent with therapy-related disease in a patient with metastatic non-small-cell lung cancer (NSCLC) previously treated with cytotoxic chemotherapy and immunotherapy.</p><p><strong>Case presentation: </strong>A 78-year-old woman with Stage IV NSCLC (adenocarcinoma, no actionable mutations, PD-L1 negative) initially received carboplatin, pemetrexed, and pembrolizumab, complicated by neutropenic fever. She later enrolled in the SHERLOC trial (MM-121 + docetaxel), discontinued due to colitis and neutropenia. Maintenance therapy with pembrolizumab was continued intermittently alongside stereotactic body radiation and cryoablation. After several years of disease control, follow-up PET/CT demonstrated new adrenal and iliac bone lesions concerning for progression. Shortly thereafter, she developed multiple pruritic, erythematous plaques on her extremities, accompanied by fatigue, weight loss, and cytopenias. Laboratory evaluation showed leukocytosis, macrocytic anemia, and severe thrombocytopenia, with peripheral smear revealing 72% circulating blasts. Punch biopsy confirmed LC, demonstrating dermal infiltration by myeloid blasts (CD4, CD43, CD68, and MPO positive). Flow cytometry and bone marrow biopsy established AML with > 75% myeloblasts. Next-generation sequencing identified DNMT3A, U2AF1, and PPM1D mutations. Conventional cytogenetic analysis was noninformative because no metaphase cells were obtained, AML FISH was normal, and PML-RARA FISH was negative, findings consistent with therapy-related AML.</p><p><strong>Interventions and outcomes: </strong>The patient received hydroxyurea, transfusion support, and tumor lysis prophylaxis. Given her age, ECOG 1, adverse-risk mutations, and personal preference, she declined further chemotherapy and transitioned to hospice.</p><p><strong>Conclusion: </strong>LC at the time of initial AML diagnosis is uncommon and portends poor prognosis. Clinicians should remain vigilant for new skin lesions and cytopenias in cancer survivors previously exposed to chemotherapy, as early recognition of extramedullary AML may expedite timely diagnosis and care planning.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"1198642"},"PeriodicalIF":0.7,"publicationDate":"2026-05-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13157302/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147869898","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}