Case Reports in Hematology最新文献

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Successful Treatment of Anemia With Ringed Sideroblasts Induced by Antidepressants Through Vitamin B6 Supplementation and Discontinuation of Antidepressants. 通过补充维生素B6和停用抗抑郁药物成功治疗抗抑郁药物诱导的环状铁母细胞贫血。
IF 0.7
Case Reports in Hematology Pub Date : 2025-07-01 eCollection Date: 2025-01-01 DOI: 10.1155/crh/7046013
Sanshiro Nakao, Chiaki Nakaseko, Chikako Ohwada, Keisuke Kirito, Asuka Shibamiya, Akane Tanaka, Reiko Watanabe, Naomi Shimizu
{"title":"Successful Treatment of Anemia With Ringed Sideroblasts Induced by Antidepressants Through Vitamin B6 Supplementation and Discontinuation of Antidepressants.","authors":"Sanshiro Nakao, Chiaki Nakaseko, Chikako Ohwada, Keisuke Kirito, Asuka Shibamiya, Akane Tanaka, Reiko Watanabe, Naomi Shimizu","doi":"10.1155/crh/7046013","DOIUrl":"10.1155/crh/7046013","url":null,"abstract":"<p><p>Vitamin B6 (VB6) is a vital coenzyme for δ-aminolevulinic acid synthase (ALAS) in heme biosynthesis. We report a 49-year-old male with severe microcytic anemia and ringed sideroblasts initially diagnosed as myelodysplastic syndrome (MDS). VB6 deficiency, attributed to long-term amoxapine use, was identified. His anemia improved significantly with VB6 supplementation and resolved completely after discontinuing amoxapine. This case highlights the need to consider VB6 deficiency in anemia with ringed sideroblasts.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"7046013"},"PeriodicalIF":0.7,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12255493/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144627325","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}
引用次数: 0
Severe Thrombocytopenia Secondary to Babesiosis: A Case Report. 巴贝斯虫病继发严重血小板减少1例。
IF 0.7
Case Reports in Hematology Pub Date : 2025-07-01 eCollection Date: 2025-01-01 DOI: 10.1155/crh/9918329
Vidyasagar R Cirra, Sharath Kommu, Michael Husak, Christopher Osterbauer
{"title":"Severe Thrombocytopenia Secondary to Babesiosis: A Case Report.","authors":"Vidyasagar R Cirra, Sharath Kommu, Michael Husak, Christopher Osterbauer","doi":"10.1155/crh/9918329","DOIUrl":"10.1155/crh/9918329","url":null,"abstract":"<p><p>A 76-year-old man with a history of atrial fibrillation treated with warfarin, renal calculi with a history of lithotripsy, hypertension, anxiety, and diabetes mellitus with recent tick exposure presented with abdominal pain, fatigue, nausea, and fever with chills. Workup revealed thrombocytopenia and hemolysis. Due to the likelihood of immune thrombocytopenia (ITP) secondary to a viral etiology, the patient was initially started on steroids. The patient subsequently tested positive for babesiosis on peripheral smear and polymerase chain reaction. A peripheral smear showed giant platelets and was positive for immunoglobulin M platelet antibodies. Other etiologies of thrombocytopenia were excluded. The patient was diagnosed with ITP secondary to babesiosis. Antibiotics were initiated to treat babesiosis. The platelet count was nonresponsive to steroids and gradually improved following intravenous immunoglobulin administration and continued antibiotic treatment. This rare case highlights the importance of considering ITP secondary to babesiosis as the etiology of severe thrombocytopenia in babesiosis, as appropriate recognition and early treatment of babesiosis and ITP can prevent serious complications.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"9918329"},"PeriodicalIF":0.7,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12237552/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144592625","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}
引用次数: 0
Life-Threatening Bleeding From Acquired FXI Inhibitors in a Patient With Colorectal Adenocarcinoma. 获得性FXI抑制剂导致结直肠癌患者出血危及生命
IF 0.7
Case Reports in Hematology Pub Date : 2025-06-27 eCollection Date: 2025-01-01 DOI: 10.1155/crh/3821648
Marina Dragičević Jojkić, Amir El Farra, Nebojša Rajić, Ivana Urošević, Aleksandar Savić
{"title":"Life-Threatening Bleeding From Acquired FXI Inhibitors in a Patient With Colorectal Adenocarcinoma.","authors":"Marina Dragičević Jojkić, Amir El Farra, Nebojša Rajić, Ivana Urošević, Aleksandar Savić","doi":"10.1155/crh/3821648","DOIUrl":"10.1155/crh/3821648","url":null,"abstract":"<p><p>Acquired inhibitors of coagulation factor XI (FXI) are a rare cause of bleeding disorders, typically associated with autoimmune diseases or malignancies. Although uncommon, these inhibitors can lead to severe bleeding, which can be difficult to manage. A limited number of cases have been reported where acquired FXI inhibitors are associated with malignancy. This case report presented a rare occurrence of acquired coagulation FXI inhibitors in a 60-year-old male with sigmoid colon adenocarcinoma. The patient experienced severe postpolypectomy gastrointestinal bleeding and was diagnosed with FXI inhibitors after laboratory tests revealed prolonged activated partial thromboplastin time (aPTT) and reduced activities of factors IX, XI, and XII. The patient underwent surgery, and life-threatening hemorrhagic shock developed. He was reoperated, and treatment with recombinant factor VIIa (rFVIIa), tranexamic acid, and oral corticosteroids was initiated. The therapy successfully controlled the bleeding and resolved the inhibitor. This case highlights the risk of severe bleeding in patients with acquired FXI inhibitors and emphasizes the importance of early diagnosis and personalized treatment. Regular monitoring is essential due to the risk of relapse, particularly in cases associated with malignancy.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"3821648"},"PeriodicalIF":0.7,"publicationDate":"2025-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12255497/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144627324","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}
引用次数: 0
MPRIP::PDGFRB Fusion Gene: A Rare Case Report of Adult Myeloid/Lymphoid Neoplasm With Eosinophilia and Tyrosine Kinase Gene Fusions. MPRIP: PDGFRB融合基因:成人髓/淋巴肿瘤嗜酸性粒细胞增多和酪氨酸激酶基因融合的罕见病例报告。
IF 0.7
Case Reports in Hematology Pub Date : 2025-06-27 eCollection Date: 2025-01-01 DOI: 10.1155/crh/7098722
Taksin Ukkahad, Tanapun Thamgrang
{"title":"<i>MPRIP::PDGFRB</i> Fusion Gene: A Rare Case Report of Adult Myeloid/Lymphoid Neoplasm With Eosinophilia and Tyrosine Kinase Gene Fusions.","authors":"Taksin Ukkahad, Tanapun Thamgrang","doi":"10.1155/crh/7098722","DOIUrl":"10.1155/crh/7098722","url":null,"abstract":"<p><p>Myeloid/lymphoid neoplasms with eosinophilia and tyrosine kinase gene fusions (MLN-TK) represent rare hematological malignancies driven by pathological fusion genes involving tyrosine kinase genes. Among these, rearrangements of the <i>PDGFRB</i> gene, particularly the <i>ETV6::PDGFRB</i> rearrangement, are frequently observed as pathogenic mutations. Conversely, instances of the <i>MPRIP::PDGFRB</i> fusion gene are rarely documented. In this case report, we present a 32-year-old previously healthy Thai male who presented to the hospital with constitutional symptoms and marked splenomegaly. His complete blood count revealed mild anemia, marked leukocytosis with hypereosinophilia, and mild thrombocytopenia. A bone marrow study showed hypercellular marrow with granulocytic hyperplasia extensively involved with eosinophils, without morphological evidence of blasts. Conventional cytogenetics identified a t (5; 17) (q33; p13). Further targeted RNA analysis using next-generation sequencing (NGS) detected a fusion gene involving <i>MPRIP::PDGFRB</i>. The patient was diagnosed with myeloid/lymphoid neoplasms with eosinophilia and <i>MPRIP::PDGFRB</i> rearrangement in the chronic-phase disease and was initiated on oral imatinib at a daily dose of 100 mg. One month after initiating the treatment, the patient achieved a hematological response consistent with complete response (CR) criteria. Imatinib therapy has been well-tolerated without reported adverse events, and a 1-year molecular assessment confirmed the achievement of complete molecular response (CMR).</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"7098722"},"PeriodicalIF":0.7,"publicationDate":"2025-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12227262/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144576610","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}
引用次数: 0
Liver Transplantation for Isolated Langerhans Cell Histiocytosis in an Adult. 成人孤立朗格汉斯细胞组织细胞增多症的肝移植治疗。
IF 0.7
Case Reports in Hematology Pub Date : 2025-06-25 eCollection Date: 2025-01-01 DOI: 10.1155/crh/1179811
Ruiyang Huang
{"title":"Liver Transplantation for Isolated Langerhans Cell Histiocytosis in an Adult.","authors":"Ruiyang Huang","doi":"10.1155/crh/1179811","DOIUrl":"10.1155/crh/1179811","url":null,"abstract":"<p><p>Langerhans cell histiocytosis (LCH) is a rare disease of proliferation of histiocytic disorder composed of histologically bland Langerhans cells mixed with reactive mononuclear and granulocytic cells, and often accompanied by eosinophils. These cells are characterized by expression of CD1a, S-100 and Langerin proteins. The clinical presentation ranges from indolent to aggressive, depending on the anatomic site involved which can be unifocal, multifocal, unisystemic, or multifocal and multisystemic disease. Cases involving LCH disease and treatment involving the liver are rare, especially in adult patients. Herein, we discuss a case of a 56-year-old male patient who presented with jaundice, acute abdominal pain, and a history of elevated liver function tests assumed to be caused by fatty liver disease. However, a computed tomography (CT) scan revealed a cholangiocarcinoma with associated biliary dilatation and cirrhosis. Pathological examination revealed Langerhans cell involvement. Negative bone marrow biopsy and bone scan indicated that the patient was indeed suffering from unisystemic LCH with isolated liver involvement causing cirrhosis. Patient underwent orthotopic liver transplantation (LT) and has since shown stable liver function without external therapy for 3 years.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"1179811"},"PeriodicalIF":0.7,"publicationDate":"2025-06-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12221540/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144555231","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}
引用次数: 0
Acute Myeloid Leukemia With NPM1 Mutation Presenting With Rapidly Progressing Hypereosinophilia. 急性髓性白血病伴NPM1突变,表现为快速进展的嗜酸性粒细胞增多症。
IF 0.7
Case Reports in Hematology Pub Date : 2025-06-18 eCollection Date: 2025-01-01 DOI: 10.1155/crh/5125740
S Einarsdottir, G Orrsjö, L von Bahr, A Staffas, L Fogelstrand
{"title":"Acute Myeloid Leukemia With <i>NPM1</i> Mutation Presenting With Rapidly Progressing Hypereosinophilia.","authors":"S Einarsdottir, G Orrsjö, L von Bahr, A Staffas, L Fogelstrand","doi":"10.1155/crh/5125740","DOIUrl":"10.1155/crh/5125740","url":null,"abstract":"<p><p>Hypereosinophilia presents a significant clinical challenge. We describe a case of severe, rapidly progressing hypereosinophilia, with the white blood cell count increasing from 40,000/μL to over 130,000/μL within days, and 70% eosinophils on differential count. The patient initially presented with diffuse symptoms but developed eosinophilic myocarditis during hospitalization. Targeted next-generation sequencing identified a mutation in <i>NPM1</i> and according to the WHO 5<sup>th</sup> edition criteria, the patient was diagnosed with acute myeloid leukemia (AML) with <i>NPM1</i> mutation. Whole genome and transcriptome sequencing revealed a concurrent fusion <i>ETV6</i>::<i>ACSL6</i>. This fusion has been previously described in myeloid diseases with eosinophilia. Despite initial deep response to AML treatment, reaching MRD-negativity for <i>NPM1</i>, the patient relapsed shortly after stem cell transplantation and died.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"5125740"},"PeriodicalIF":0.7,"publicationDate":"2025-06-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12197511/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144498351","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}
引用次数: 0
Primary Cutaneous Gamma-Delta T-Cell Lymphoma Presenting With Hemophagocytic Lymphohistiocytosis in a Young Polynesian Male. 原发性皮肤γ - δ t细胞淋巴瘤一例年轻波利尼西亚男性表现为噬血细胞性淋巴组织细胞增多症。
IF 0.7
Case Reports in Hematology Pub Date : 2025-05-23 eCollection Date: 2025-01-01 DOI: 10.1155/crh/8582804
Saeed Arabi, Victoria Vardell, Timothy Hanley, Scott Florell, Ahmad Halwani, Ming Lim
{"title":"Primary Cutaneous Gamma-Delta T-Cell Lymphoma Presenting With Hemophagocytic Lymphohistiocytosis in a Young Polynesian Male.","authors":"Saeed Arabi, Victoria Vardell, Timothy Hanley, Scott Florell, Ahmad Halwani, Ming Lim","doi":"10.1155/crh/8582804","DOIUrl":"10.1155/crh/8582804","url":null,"abstract":"<p><p>Primary cutaneous gamma-delta T-cell lymphoma (PCGD-TCL) is a very rare subtype of cutaneous T-cell lymphoma. We report the case of a young Polynesian male who presented with fever and an abdominal wall rash and highlight the workup leading to the diagnosis of PCGD-TCL. As PCGD-TCL is rare and mimics other medical conditions, its diagnosis requires a high index of suspicion and can be challenging. Hemophagocytic lymphohistiocytosis (HLH) occurs with PCGD-TCL and can be a marker of more invasive disease. There are no well-defined treatment guidelines, but the most common treatment approach is anthracycline-based multiagent chemotherapy followed by allogeneic stem cell transplant. Targeted therapies are being increasingly used as well. Prognosis remains poor and 5-year survival is < 20%, particularly in more invasive disease. We highlight how this patient's demographic varies from the published literature and discuss some unique particulars of the diagnostic evaluation and treatment, especially in the presence of concurrent HLH.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"8582804"},"PeriodicalIF":0.7,"publicationDate":"2025-05-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12124927/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144200413","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}
引用次数: 0
Unusual Presentation of Childhood Leukemia With Vaginal Bleeding: A Case Report. 儿童白血病伴阴道出血的罕见表现:1例报告。
IF 0.7
Case Reports in Hematology Pub Date : 2025-05-20 eCollection Date: 2025-01-01 DOI: 10.1155/crh/9949125
Şule Çalışkan Kamış
{"title":"Unusual Presentation of Childhood Leukemia With Vaginal Bleeding: A Case Report.","authors":"Şule Çalışkan Kamış","doi":"10.1155/crh/9949125","DOIUrl":"10.1155/crh/9949125","url":null,"abstract":"<p><p><b>Introduction:</b> This case report describes a 17-year-old female patient who initially presented with vaginal bleeding, weight loss, and nonspecific symptoms, which led to the diagnosis of B-cell acute lymphoblastic leukemia (B-ALL). This unusual presentation highlights the importance of considering hematological malignancies in patients with atypical symptoms. <b>Case Presentation:</b> The patient, married for 2 weeks, experienced vaginal bleeding following her first sexual intercourse, which did not resolve spontaneously. She also reported a 6 kg weight loss over the past 3-4 months, hair loss, and a history of dysmenorrhea and an ovarian cyst detected 2 years prior. Laboratory investigations revealed leukocytosis (WBC: 16,500/μL), anemia (Hb: 11.4 g/dL), and thrombocytopenia (Plt: 44,000/μL). Bone marrow aspiration (BMA) and flow cytometry confirmed the diagnosis of B-ALL, revealing a high percentage of atypical lymphoid cells. <b>Discussion:</b> This case underscores the rarity of diagnosing hematological malignancies in patients with vaginal bleeding. The patient's symptoms, including weight loss and thrombocytopenia, should have prompted a more comprehensive evaluation. Early recognition of B-ALL is crucial as prompt treatment significantly improves outcomes. The patient was started on prednisolone and alkalinized fluids, and her condition was closely monitored. <b>Conclusion:</b> Vaginal bleeding in young patients should not be dismissed as a minor issue, especially when accompanied by other systemic symptoms like weight loss and thrombocytopenia. Early diagnosis of B-ALL in such cases can lead to better management and prognosis.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"9949125"},"PeriodicalIF":0.7,"publicationDate":"2025-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12116196/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144162748","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}
引用次数: 0
Severe Thrombocytopenia Secondary to Severe Iron Deficiency Anemia due to Menorrhagia. 月经过多引起的严重缺铁性贫血继发的严重血小板减少症。
IF 0.7
Case Reports in Hematology Pub Date : 2025-05-18 eCollection Date: 2025-01-01 DOI: 10.1155/crh/3894943
Tyler E Russeth, Amanda Luong, Mandi Liu, Mihir Shah, Nicole Desai
{"title":"Severe Thrombocytopenia Secondary to Severe Iron Deficiency Anemia due to Menorrhagia.","authors":"Tyler E Russeth, Amanda Luong, Mandi Liu, Mihir Shah, Nicole Desai","doi":"10.1155/crh/3894943","DOIUrl":"10.1155/crh/3894943","url":null,"abstract":"<p><p>Thrombocytosis is commonly seen in patients with iron deficiency anemia and often normalizes following iron supplementation. Thrombocytopenia with iron deficiency anemia is a less common occurrence that can be seen in severe cases. This phenomenon is well documented in the pediatric population secondary to nutritional deficiency but is underreported in the adult population. Similarly, thrombocytopenia resolves following iron supplementation but the mechanism behind this and why select patients are affected is not well understood. This case report describes a young woman with menorrhagia who was found to have iron deficiency anemia and severe thrombocytopenia with resolution following intravenous iron.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"3894943"},"PeriodicalIF":0.7,"publicationDate":"2025-05-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12103951/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144152349","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}
引用次数: 0
Unusual Long-Term Survival in an Adult Patient With Langerhans Cell Histiocytosis and Central Nervous System Involvement: A Case Report. 朗格汉斯细胞组织细胞增多症并累及中枢神经系统的成人患者异常的长期生存率:1例报告。
IF 0.7
Case Reports in Hematology Pub Date : 2025-05-16 eCollection Date: 2025-01-01 DOI: 10.1155/crh/6031427
María José Miranda Pallarés, Tomás Álvaro Naranjo, Anna Vidal Rodriguez, Francisca Martínez Madueño
{"title":"Unusual Long-Term Survival in an Adult Patient With Langerhans Cell Histiocytosis and Central Nervous System Involvement: A Case Report.","authors":"María José Miranda Pallarés, Tomás Álvaro Naranjo, Anna Vidal Rodriguez, Francisca Martínez Madueño","doi":"10.1155/crh/6031427","DOIUrl":"10.1155/crh/6031427","url":null,"abstract":"<p><p>Langerhans cell histiocytosis (LCH) in adults is a rare condition. The disease presents with focal or multifocal involvement of a single organ/system or focal or disseminated multisystem disease. Involvement of the central nervous system (CNS) is also infrequent, with diabetes insipidus as a common manifestation of posterior pituitary lesions. Biopsy-proven diagnosis with the observation of characteristic Langerhans cells infiltrate with positive immunohistochemistry of S100 protein, CD1a, and CD68 cells. The patient presented generalized lymphadenopathy and was diagnosed with low-risk single-system LCH-based distinctive pathological findings in a lymph node biopsy. During the disease, CNS involvement was documented and the patient received different sequential therapeutic schemes, achieving complete remission that has been maintained for 8 years. The prolonged duration of remission with disappearance of lymphadenopathy and CNS lesions in an adult patient with LCH is an unusual clinical observation.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2025 ","pages":"6031427"},"PeriodicalIF":0.7,"publicationDate":"2025-05-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12101904/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144143931","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}
引用次数: 0
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