Case Reports in Hematology最新文献

{"title":"Supercharged Hypercoagulability: A Case of Heparin-Induced Thrombocytopenia With Thrombosis in a Patient With Double Heterozygous Factor V Leiden and Prothrombin Mutations.","authors":"Yudai Okabe, Jose Ibarra Rodriguez, Jane Edmunds, Nyembezi L Dhliwayo","doi":"10.1155/crh/3078377","DOIUrl":"https://doi.org/10.1155/crh/3078377","url":null,"abstract":"<p><p>Factor V Leiden (FVL) and the prothrombin 20210A gene mutation are two common genetic predispositions to hypercoagulability. We present a complex case of recurrent venous thromboembolism (VTE) in a 50-year-old woman with double heterozygosity for FVL and prothrombin G20210A, complicated by heparin-induced thrombocytopenia (HIT) and May-Thurner syndrome. Following a recent orthopedic surgery and a sedentary postoperative course, the patient developed extensive bilateral deep vein thrombosis (DVT) and a saddle pulmonary embolism. Initial anticoagulation with heparin was complicated by progressive thrombocytopenia and confirmed HIT, prompting transition to bivalirudin and subsequently argatroban. Despite therapeutic anticoagulation and multiple interventional procedures, the patient experienced repeated thrombotic events. After increasing the therapeutic aPTT goal for argatroban, she ultimately stabilized and was successfully transitioned to oral apixaban. This case highlights the synergistic risk posed by the combination of inherited thrombophilia, structural venous abnormalities, and acquired prothrombotic conditions. It provides insight into the complex nature of proper anticoagulation strategies in these individual cases. Our use of argatroban with higher aPTT goals may provide guidance in future cases of refractory VTE. Further studies are needed to better understand the optimal therapies and management for patients with hereditary and acquired thrombophilia.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"3078377"},"PeriodicalIF":0.7,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12497522/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145245543","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":"POEMS Syndrome Without a Detectable Monoclonal Peak: The Critical Role of VEGF and Bone Marrow Biopsy in Diagnosis.","authors":"Paul J Pecorin, Max Melchioris A, Guy Olson, Emily Flammersfeld, Marwah Al Tekreeti, Patrick Atisha","doi":"10.1155/crh/5530850","DOIUrl":"10.1155/crh/5530850","url":null,"abstract":"<p><p>Polyneuropathy, organomegaly, endocrinopathy, monoclonal plasma cell disorder, skin changes (POEMS) syndrome is a rare disorder that is frequently misdiagnosed due to its heterogeneous presentation and overlap with chronic inflammatory demyelinating polyneuropathy (CIDP). Diagnosis requires the presence of polyneuropathy and a monoclonal plasma cell disorder, along with additional major and minor criteria. We present a 73-year-old woman with progressive weakness, volume overload, and weight loss, initially diagnosed with CIDP. Despite IVIG therapy, her symptoms worsened. Notably, no monoclonal peak was detected on serum protein electrophoresis (SPEP) or immunofixation, complicating the diagnosis. However, markedly elevated vascular endothelial growth factor (VEGF) levels (11.245 pg/mL) and bone marrow biopsy findings of a monoclonal plasma cell disorder confirmed POEMS syndrome. She also developed multiple thromboembolic events, highlighting the syndrome's prothrombotic nature. This case underscores the importance of maintaining high suspicion for POEMS syndrome in the setting of undifferentiated polyneuropathy, even in the absence of a monoclonal peak on SPEP. VEGF measurement and bone marrow biopsy are crucial for diagnosis in such cases. Early recognition and treatment, including plasma cell-directed therapy and anticoagulation, are essential to improving patient outcomes and preventing irreversible complications.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"5530850"},"PeriodicalIF":0.7,"publicationDate":"2025-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12463516/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145187123","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":"Intravascular Lymphoma Presenting as Multiple Endocrine Failure, Transforming Into the Hemophagocytic Variant, and Relapsing as a Thrombotic Microangiopathy.","authors":"Nigel P Murray, Cinthia Escobar, Enrique Ramos, Claudia Perez, Dan Hartman","doi":"10.1155/crh/5560781","DOIUrl":"10.1155/crh/5560781","url":null,"abstract":"<p><p>Intravascular large B-cell lymphoma (IVLCBL) is a rare form of non-Hodgkin's lymphoma and is characterized by the growth of large B-cells within blood vessels and bone marrow sinusoids. A 55-year-old man presented with a multiple endocrine failure which progressed to a pancytopenia. A bone marrow biopsy revealed a diffuse infiltration by large B-cells in the sinusoids consistent with intravascular lymphoma. After 6 cycles of R-CHOP, complete remission was achieved. Six months later, the patient relapsed presenting with a thrombotic microangiopathy which progressed to multiple organ failure and death.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"5560781"},"PeriodicalIF":0.7,"publicationDate":"2025-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12460004/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145151297","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":"Intravascular Large B-Cell Lymphoma Presenting as Systemic Capillary Leak Syndrome With Immunological Phenomena: A Case Report.","authors":"Helena M van Dongen, Bregje M Koomen, Jordy Jurgens, Helen L Leavis","doi":"10.1155/crh/9919411","DOIUrl":"10.1155/crh/9919411","url":null,"abstract":"<p><p>Intravascular large B-cell lymphoma (IVLBCL) has a high mortality rate, partly due to its heterogeneous presentation and rarity. We present a case of a 73-year-old woman who came into the emergency room in need of fluid resuscitation, interpreted as septic shock. However, broad-spectrum antibiotics gave no resolution, and no causative agent was found. Further physical examination showed proximal muscle weakness, Raynaud's phenomenon, and calcinosis cutis. During 3 weeks of admission, vasopressor support was required continuously due to a capillary leak syndrome. The patient passed away. The underlying malignancy was only revealed at autopsy. To the best of our knowledge, this is the first case of IVLBCL with hypovolemic shock due to systemic capillary leak syndrome in combination with a wide range of immunological phenomena.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"9919411"},"PeriodicalIF":0.7,"publicationDate":"2025-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12446591/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145114624","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":"Adalimumab-Induced Thrombocytopenia: A Case Report of Drug-Induced Thrombocytopenia.","authors":"Shelby E Jones, Joshua Sellers, Margaret C Wheless, Danielle Fishman, Robert K McKenzie","doi":"10.1155/crh/4414589","DOIUrl":"10.1155/crh/4414589","url":null,"abstract":"<p><p>In this case report, we present a patient with severe thrombocytopenia induced by adalimumab after 4 weeks of treatment for rheumatoid arthritis. This case adds to present literature regarding antitumor necrosis factor-alpha (TNF-α)-induced thrombocytopenia and explores the use of thrombopoietin receptor agonist medications in refractory drug-induced immune thrombocytopenia (DITP). A 57-year-old female with a history of rheumatoid arthritis presented to the emergency department for progressive petechial rash 4 days after her third dose of adalimumab. It was determined that the patient had immune thrombocytopenia caused by adalimumab and was initially treated with steroids and intravenous immunoglobulin (IVIG). Platelets remained undetectable; hence, romiplostim was initiated, after which platelets trended up to normal levels a week later. Anti-TNF-α agents carry the risk of severe side effects, including thrombocytopenia, which should be monitored closely in the first few months of therapy. The treatment of DITP is challenging, especially when the causative agent has a prolonged half-life.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"4414589"},"PeriodicalIF":0.7,"publicationDate":"2025-09-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12435997/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145076339","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":"POEMS Syndrome Masquerading as Metastatic Prostate Cancer Based on PSMA Avid Lesions.","authors":"Pierre Rodriguez Alarcon, Dawid Mehlich, Fidai Shiraz, Matias Sanchez","doi":"10.1155/crh/4556395","DOIUrl":"10.1155/crh/4556395","url":null,"abstract":"<p><p>Polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin change (POEMS) syndrome is a multisystem disorder, and it is often misdiagnosed with other entities including chronic inflammatory demyelinating polyneuropathy (CIDP). Here, we present a case of a patient with presumed metastatic prostate cancer due to prostate-specific membrane antigen (PSMA) avid lesions and a history of neuropathy not responding to conventional treatment for CIDP. His physical exam findings, in addition to an appropriate workup, led to a diagnosis of POEMS syndrome. This case highlights the importance of a high index of clinical suspicion, even when imaging suggests otherwise.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"4556395"},"PeriodicalIF":0.7,"publicationDate":"2025-09-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12422860/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145041749","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":"Successful Treatment of Leukemic Synovitis Complicated With Chronic Myelomonocytic Leukemia.","authors":"Hiroki Tsutsumi, Masahisa Kudo, Nobuo Maseki, Machiko Kawamura, Kazuhiko Kobayashi, Yasunobu Sekiguchi, Yuka Harada, Daichi Sadato, Hirofumi Kobayashi","doi":"10.1155/crh/3159757","DOIUrl":"10.1155/crh/3159757","url":null,"abstract":"<p><p>Chronic myelomonocytic leukemia (CMML) is a myeloproliferative disease characterized by monocyte-predominant hematopoiesis. It is sometimes complicated with cutaneous involvement known as leukemic cutis, which is associated with a poor prognosis. We report a patient with CMML who developed fever with knee joint swelling and pain. The patient was considered to have leukemic synovitis, and treatment with azacitidine improved her symptoms. Our case suggested that leukemic synovitis might indicate the indication for treatment, and arthrocentesis should be performed in patients with leukemia who present with joint swelling.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"3159757"},"PeriodicalIF":0.7,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12419918/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145041762","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":"Subgaleal Hematoma in a Female With Normal Coagulation Tests.","authors":"Sajjad Ataei-Azimi, Mettine H A Bos, Hossein Rahimi, Hassan Mansouritorghabeh","doi":"10.1155/crh/5481806","DOIUrl":"10.1155/crh/5481806","url":null,"abstract":"<p><p>Factor XIII (FXIII) deficiency is a rare coagulopathy with an estimated prevalence of approximately 1 in 1 to 2 million, affecting males and females with equal frequency. FXIII plays a critical role in hemostasis by stabilizing fibrin clots through covalent cross-linking of fibrin monomers, thereby conferring mechanical resistance and durability to the clot structure. Clinically, FXIII deficiency presents with a spectrum of hemorrhagic manifestations including bleeding from the umbilical cord, intracranial hemorrhage, recurrent miscarriages, menorrhagia, epistaxis, gingival bleeding, and poor wound healing. Despite significant bleeding symptoms, routine primary hemostasis screening tests are typically within normal limits since FXIII acts downstream of clot formation. The clot solubility in 5-molar urea is widely used, especially in resource-limited settings. An 11-year-old female patient presented with symptoms including vomiting, lethargy, severe headache, and a subgaleal hematoma. Neurosurgical intervention confirmed intracranial hemorrhage. Her medical history was notable for neonatal umbilical cord bleeding, hematomas, and postdental extraction bleeding. Despite these clinical features, multiple clot solubility tests yielded normal results. Subsequent quantitative assessment of FXIII by chromogenic assay performed on the CS-5100 system revealed a markedly decreased FXIII activity level of 12.4%. This discrepancy highlights the limited insensitivity of the clot solubility test in detecting FXIII deficiency. Therefore, accurate diagnosis of FXIII deficiency necessitates a combined diagnostic approach incorporating both clot solubility testing and specific quantitative FXIII activity measurement. The clot stability test, despite its limitations in detecting FXIII deficiency, is frequently employed in developing countries for screening reduced FXIII levels due to its simplicity. However, the current findings indicate that in patients suspected of FXIII deficiency, accurate diagnosis necessitates the performance of both a clot stability test (5 M urea test) and a specific FXIII activity assay. A comprehensive medical and family history is fundamental to the clinical and laboratory approach to patients presenting with bleeding tendencies. Notably, a subset of patients exhibiting bleeding symptoms may exhibit normal findings on initial first-line hemostasis screening assays.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"5481806"},"PeriodicalIF":0.7,"publicationDate":"2025-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12413938/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145013390","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 Case of Renal Failure Caused by T-Cell Prolymphocytic Leukaemia Infiltration.","authors":"Stuart K Gibson, Thomas Davis, Melisa Vazquez, Swe Htet, Eric Wong","doi":"10.1155/crh/1402078","DOIUrl":"10.1155/crh/1402078","url":null,"abstract":"<p><p>T-cell prolymphocytic leukaemia (T-PLL) is an aggressive and rare post-thymic T cell malignancy, highly refractory to conventional cytotoxic chemotherapeutics. While extranodal involvement is common, solid organ invasion is rare. We present the case of a 76-year-old man who developed acute renal failure secondary to T-PLL renal infiltration. On day four of his admission, prior to commencing alemtuzumab, his creatinine rose from 133 μmol/L to 390 μmol/L, with anuria. Renal biopsy demonstrated an infiltrate of monomorphic, mononuclear cells positive for a STAT5B mutation, consistent with T-PLL infiltration. He required haemodialysis, but was treated with pulsed methylprednisolone and alemtuzumab, with excellent renal recovery, although remission was not achieved. This case demonstrates that renal leukaemic infiltration must be considered in T-PLL patients with rapidly progressive renal failure, and that solid organ invasion should not contraindicate timely commencement of T-PLL-directed therapy with alemtuzumab.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"1402078"},"PeriodicalIF":0.7,"publicationDate":"2025-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12413279/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145013384","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":"Coexistence of Essential Thrombocythemia and Waldenström Macroglobulinemia: A Case Report.","authors":"Meghan Wallace, Bruce Mathey, Cecilia C S Yeung, Jacob S Appelbaum, Mark Wallace","doi":"10.1155/crh/3390770","DOIUrl":"10.1155/crh/3390770","url":null,"abstract":"<p><p>Waldenström macroglobulinemia (WM) and essential thrombocythemia (ET) are distinct hematologic malignancies that have only been reported to co-occur in one previous patient. We present a 64-year-old man with a significant family history for WM who was found to have both ET and WM. He had symptomatic ET, diagnosed by elevated platelets and a positive JAK2 V617F mutation, and asymptomatic WM was found on serum electrophoresis done for screening due to family history. Genomic evaluation of the myeloid and lymphoid cells suggested independent neoplastic transformation. This is the second reported case of a patient with both WM and ET. There was no evidence for a shared mechanism in these dual malignancies.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"3390770"},"PeriodicalIF":0.7,"publicationDate":"2025-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12411022/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145013429","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}