Case Reports in Hematology最新文献_第3页

Refractory Hypereosinophilic Syndrome With Clonal T-Cell Receptor Rearrangement Treated With JAK Inhibition Following Incomplete Response to IL-5 Blockade. IL-5阻断不完全应答后JAK抑制治疗难治性高嗜酸性粒细胞综合征伴克隆t细胞受体重排。

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Case Reports in Hematology Pub Date : 2026-09-04 eCollection Date: 2026-01-01 DOI: 10.1155/crh/9477679

Kevin Wayne McKaughan, Phu V Truong, Gregory Bongers

{"title":"Refractory Hypereosinophilic Syndrome With Clonal T-Cell Receptor Rearrangement Treated With JAK Inhibition Following Incomplete Response to IL-5 Blockade.","authors":"Kevin Wayne McKaughan, Phu V Truong, Gregory Bongers","doi":"10.1155/crh/9477679","DOIUrl":"10.1155/crh/9477679","url":null,"abstract":"Hypereosinophilic syndrome (HES) comprises a heterogeneous group of disorders characterized by persistent eosinophilia and eosinophil-mediated organ involvement. Although interleukin-5 (IL-5) inhibition is an established treatment strategy, some patients experience persistent or recurrent disease despite therapy. Janus kinase (JAK) inhibition has emerged as a potential therapeutic option in refractory cases. We report a 63-year-old White male who presented with a diffuse pruritic erythematous rash, marked leukocytosis with eosinophils comprising approximately 83% of circulating leukocytes, and markedly elevated serum IgE levels. Bone marrow biopsy demonstrated a hypercellular marrow (approximately 89%) with eosinophils accounting for approximately 75% of nucleated cells. Extensive evaluation, including cytogenetic, fluorescence in situ hybridization, and molecular testing for myeloid neoplasms, was unrevealing, while peripheral blood polymerase chain reaction demonstrated a clonal T-cell receptor gene rearrangement. Despite prior hydroxyurea therapy, prolonged treatment with mepolizumab, and repeated corticosteroid courses, the patient developed recurrent disease with an absolute eosinophil count of 2760/μL. Ruxolitinib was initiated while IL-5 inhibition was continued. Following JAK inhibition, leukocyte and eosinophil counts normalized, corticosteroids were discontinued, and disease control was maintained at 3-month follow-up. This case highlights the potential role of JAK inhibition as adjunctive therapy in refractory HES with clonal T-cell receptor rearrangement. Further studies are needed to define the safety, durability, and optimal integration of JAK inhibition in patients with refractory eosinophilic disorders.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"9477679"},"PeriodicalIF":0.7,"publicationDate":"2026-09-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13542987/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148897958","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

Celiac Disease Emerging After ASCT in Multiple Myeloma: A Rare Diagnostic Intersection and Literature Review. 多发性骨髓瘤ASCT后出现乳糜泻：罕见的诊断交叉点和文献综述。

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Case Reports in Hematology Pub Date : 2026-09-02 eCollection Date: 2026-01-01 DOI: 10.1155/crh/8921605

Mais Musleh, Mahmoud Alhamadeh Alswij, Qossay Alhusein

{"title":"Celiac Disease Emerging After ASCT in Multiple Myeloma: A Rare Diagnostic Intersection and Literature Review.","authors":"Mais Musleh, Mahmoud Alhamadeh Alswij, Qossay Alhusein","doi":"10.1155/crh/8921605","DOIUrl":"10.1155/crh/8921605","url":null,"abstract":"Background: Autologous stem cell transplantation (ASCT) is a standard component of therapy for eligible patients with multiple myeloma. Persistent gastrointestinal symptoms after ASCT are commonly attributed to infection, medication-related toxicity, or disease relapse, whereas autoimmune enteropathies such as celiac disease are rarely considered.Case presentation: We report a 53-year-old woman with lambda light-chain multiple myeloma who achieved complete remission after bortezomib, lenalidomide, and dexamethasone induction followed by ASCT and lenalidomide maintenance. Four months after transplantation, she developed persistent diarrhea and a 4-kg unintentional weight loss. Infectious investigations were negative, and imaging and bone marrow assessment showed no evidence of myeloma relapse. Upper gastrointestinal endoscopy demonstrated duodenal villous atrophy, and biopsy findings were consistent with Marsh 3A celiac disease. Serum antitissue transglutaminase IgA was markedly elevated at > 123 U/mL. A strict gluten-free diet resulted in complete resolution of diarrhea within 2 weeks and a subsequent decline in celiac serologic markers. Lenalidomide-associated diarrhea or enteropathy was considered but could not be definitively excluded because treatment withdrawal and rechallenge were not performed. In addition, pre-ASCT celiac serology and HLA-DQ2/DQ8 typing were unavailable.Conclusion: This case highlights celiac disease as a rare but clinically important cause of persistent diarrhea after ASCT. Immune reconstitution following transplantation may unmask previously subclinical autoimmune disease. Clinicians should maintain a broad differential diagnosis for posttransplant gastrointestinal symptoms, as early recognition and appropriate dietary intervention can lead to rapid symptom resolution and prevent unnecessary treatment interruption or morbidity.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8921605"},"PeriodicalIF":0.7,"publicationDate":"2026-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13535857/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148882166","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

Cutaneous Clue to Clonal Evolution: Sweet's Syndrome Heralding Hematologic Progression in Essential Thrombocythemia. 克隆进化的皮肤线索：Sweet综合征预示着原发性血小板增多症的血液学进展。

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Case Reports in Hematology Pub Date : 2026-09-02 eCollection Date: 2026-01-01 DOI: 10.1155/crh/9919558

Kavya Ronanki, Bibhant Shah, Prisla Maria Dalton, Nikhil Nagpal, Kartik Saini, Sushantika, Nilotpal Chowdhury, Uttam Kumar Nath

{"title":"Cutaneous Clue to Clonal Evolution: Sweet's Syndrome Heralding Hematologic Progression in Essential Thrombocythemia.","authors":"Kavya Ronanki, Bibhant Shah, Prisla Maria Dalton, Nikhil Nagpal, Kartik Saini, Sushantika, Nilotpal Chowdhury, Uttam Kumar Nath","doi":"10.1155/crh/9919558","DOIUrl":"10.1155/crh/9919558","url":null,"abstract":"Sweet's syndrome, or acute febrile neutrophilic dermatosis, is a rare inflammatory condition that may occur idiopathically or in association with malignancy, drugs, or systemic disease. Among malignancy-associated cases, hematologic neoplasms predominate. Its bullous variant is uncommon and is more frequently linked to underlying hematologic disorders, where it may act as a paraneoplastic manifestation or an early marker of disease progression. We report a 54-year-old woman with long-standing, low-risk essential thrombocythemia, diagnosed in 2011 and maintained on hydroxyurea and aspirin with stable blood counts for over a decade. She presented with an acute onset of painful erythematous swellings progressing to tense bullae over the hands and feet, accompanied by intermittent fever. Histopathological examination of a skin biopsy demonstrated dense neutrophilic infiltration of the dermis without vasculitis, consistent with Sweet's syndrome. Systemic corticosteroid therapy led to rapid clinical improvement, and hydroxyurea was discontinued. Given the known association between Sweet's syndrome and hematologic disease progression, further evaluation was undertaken. Bone marrow examination and cytogenetic analysis revealed a complex karyotype, while next-generation sequencing identified an acquired TP53 mutation along with a CALR mutation, indicating clonal evolution and progression to a high-risk disease state. This case highlights bullous Sweet's syndrome as a rare but important cutaneous marker of clonal evolution in essential thrombocythemia. New-onset neutrophilic dermatoses in patients with previously indolent myeloproliferative neoplasms should prompt dermatologic evaluation and comprehensive hematologic reassessment, as early recognition may facilitate timely diagnosis of disease progression and guide management.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"9919558"},"PeriodicalIF":0.7,"publicationDate":"2026-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13535846/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148882246","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

Congenital Dyserythropoietic Anemia Type II Caused by a New SEC23B Sequence Variant. 由一种新的SEC23B序列变异引起的先天性II型促红细胞增生性贫血。

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Case Reports in Hematology Pub Date : 2026-09-02 eCollection Date: 2026-01-01 DOI: 10.1155/crh/7158943

Geir Erland Tjønnfjord, Arne Loraas, Signe Spetalen, Liv Toril Nygård Osnes, Kristian Tveten

{"title":"Congenital Dyserythropoietic Anemia Type II Caused by a New SEC23B Sequence Variant.","authors":"Geir Erland Tjønnfjord, Arne Loraas, Signe Spetalen, Liv Toril Nygård Osnes, Kristian Tveten","doi":"10.1155/crh/7158943","DOIUrl":"10.1155/crh/7158943","url":null,"abstract":"Congenital dyserythropoietic anemias (CDAs) include a heterogeneous group of conditions characterized by insufficient erythropoiesis giving rise to monolinear cytopenia. Based on the morphological features of erythroblasts, three major subtypes have been established: CDA Type I, CDA Type II, and CDA Type III. CDA Type II is the most common type of CDAs, and this disease is characterized by a normocytic anemia with a normal or slightly increased reticulocyte count. The bone marrow is hypercellular with erythroid hyperplasia and binucleated erythroblasts with two nuclei at the same maturation stage. The inheritance is autosomal recessive, and the disease is caused by biallelic mutations in the SEC28B gene. More than 100 pathogenetic variants have been identified. Here, we report on three patients from Somalia with CDA Type II caused by a new SEC23B sequence variant. They came to attention and were diagnosed in early adulthood. Cases 1 and 2 had mild anemia with signs of hemolysis, and Case 3 had mild anemia with reticulocytosis. The eosin-5-maleimide binding test showed reduced binding in all three patients. Molecular genetics disclosed homozygosity for the sequence variant NM_006363.6:c.1580T > C p.(Leu527Ser) in the SEC23B gene. A bone marrow aspirate was performed from Case 2, and the smear disclosed morphological abnormalities typical for CDA Type II.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"7158943"},"PeriodicalIF":0.7,"publicationDate":"2026-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13536906/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148882189","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

Double Heterozygous CDAN1 Variants of Uncertain Significance Associated With a Phenotype Consistent With Congenital Dyserythropoietic Anemia Type 1. 与1型先天性促红细胞增生性贫血表型一致的双杂合CDAN1变异的不确定意义

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Case Reports in Hematology Pub Date : 2026-08-31 eCollection Date: 2026-01-01 DOI: 10.1155/crh/8073716

Kevin G Zablonski, Neil A Lachant, Archibald Perkins, William J Archibald

引用次数: 0

Patient Care of Pregnant Women With Chronic Myeloid Leukemia in a Resource Limited Setting-Case Reports From Ghana. 在资源有限的情况下，慢性髓性白血病孕妇的患者护理——来自加纳的病例报告。

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Case Reports in Hematology Pub Date : 2026-08-29 eCollection Date: 2026-01-01 DOI: 10.1155/crh/8111388

Amma Benneh-Akwasi Kuma, Leticia Lokko Hanson, Nii Boi-Doku Pepra-Ameyaw, George Dennis Obeng, Marcell Csanádi

{"title":"Patient Care of Pregnant Women With Chronic Myeloid Leukemia in a Resource Limited Setting-Case Reports From Ghana.","authors":"Amma Benneh-Akwasi Kuma, Leticia Lokko Hanson, Nii Boi-Doku Pepra-Ameyaw, George Dennis Obeng, Marcell Csanádi","doi":"10.1155/crh/8111388","DOIUrl":"10.1155/crh/8111388","url":null,"abstract":"Although life expectancy and survival have improved for CML patients due to the advent of tyrosine kinase inhibitor (TKI) therapies, the quality of life and family planning for women still hold challenges, especially in a resource-limited setting where routine molecular monitoring and pregnancy-compatible therapeutic options are limited. Due to their teratogenicity, TKI therapies are generally considered to be contraindicated during pregnancy. Here, we present case reports of three patients from the Korle-Bu Teaching Hospital in Accra, Ghana, who were diagnosed with CML prior to pregnancy. A 29-year-old, 19-year-old and 33-year-old female were diagnosed with CML at the clinic. All patients initiated imatinib, which was stopped after confirming pregnancy. Patients were subsequently monitored with a monthly full blood count (FBC). While one of the patients was lost to follow-up, the other two patients delivered without adverse events or fetal abnormalities and were able to restart imatinib therapy within 6 months. Based on the presented cases, a pragmatic framework emerges: (1) embed comprehensive counselling at diagnosis; (2) encourage planned conception only after sustained molecular remission; (3) stop TKIs upon pregnancy confirmation; (4) monitor with clear, communicated thresholds for intervention; (5) introduce cytoreduction after the first trimester only when clinically indicated and consent given; and (6) align postpartum feeding plans with timely TKI re-initiation. As a conclusion, in contexts where social pressure to bear children is strong and pregnancy-safe treatments are limited, transparent counselling, a remission-first strategy, and tightly coordinated hematology-obstetric care offer the best path to balancing reproductive autonomy with maternal and fetal safety.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8111388"},"PeriodicalIF":0.7,"publicationDate":"2026-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13525543/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148857654","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

Sustained Erythroid Response to Eltrombopag in Low-Risk Myelodysplastic Neoplasms With Severe Anemia and Thrombocytopenia: A Case Report. 低危骨髓增生异常肿瘤伴严重贫血和血小板减少的持续红系反应：1例报告。

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Case Reports in Hematology Pub Date : 2026-08-26 eCollection Date: 2026-01-01 DOI: 10.1155/crh/1385595

Ambra Di Veroli, Cinzia De Gregoris, Roberta Laureana, Matteo Caridi, Valentina Panichi, Monica Poscente, Esther Natalie Oliva, Roberto Latagliata

{"title":"Sustained Erythroid Response to Eltrombopag in Low-Risk Myelodysplastic Neoplasms With Severe Anemia and Thrombocytopenia: A Case Report.","authors":"Ambra Di Veroli, Cinzia De Gregoris, Roberta Laureana, Matteo Caridi, Valentina Panichi, Monica Poscente, Esther Natalie Oliva, Roberto Latagliata","doi":"10.1155/crh/1385595","DOIUrl":"10.1155/crh/1385595","url":null,"abstract":"Eltrombopag has been already employed for thrombocytopenia in myelodysplastic neoplasms (MDS), but its role and efficacy in improving other cytopenias are still unknown. In October 2020, a 58-year-old low-risk MDS male with transfusion-dependent/erythropoietin-resistant anemia and severe thrombocytopenia (platelets [PLTs]: 19 × 109/L) started eltrombopag in the EQOL-MDS trial, showing an unexpected increase in Hb values up to 12.0 g/dL at Week 24, with discontinuation of the transfusion requirement. However, PLT levels remained unchanged and, as per protocol, eltrombopag was stopped in May 2021. After eltrombopag discontinuation, Hb levels worsened again with reappearance of transfusion requirement from July 2021. High-dose erythropoietin and deferasirox (ferritin: 1140 ng/mL) were restarted in September 2021, without efficacy. Thus, in December 2022, eltrombopag was reinitiated on a compassionate basis, and a new progressive increase in Hb (also in PLT values) was observed. After 12 months of 2nd eltrombopag treatment, Hb levels became normal, while PLT count maintained stable values of > 50 × 109/L. At the last follow-up, the patient was still in complete erythroid response (Hb > 12 g/dL) lasting > 29 months and in complete PLT response (> 100 × 109/L) lasting > 18 months. Eltrombopag could be evaluated as a possible option for treatment of low-risk MDS with anemia and thrombocytopenia.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"1385595"},"PeriodicalIF":0.7,"publicationDate":"2026-08-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13507826/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148833257","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

Engraftment Syndrome After Second Autologous Stem Cell Transplantation for Myeloma: A Case Report. 第二次自体干细胞移植治疗骨髓瘤后移植综合征1例报告。

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Case Reports in Hematology Pub Date : 2026-08-25 eCollection Date: 2026-01-01 DOI: 10.1155/crh/2925166

Konstantina Salveridou, Theodoros Tzamalis, Maika Klaiber-Hakimi, Sabine Haase, Stefan Diederich, Aristoteles Giagounidis

{"title":"Engraftment Syndrome After Second Autologous Stem Cell Transplantation for Myeloma: A Case Report.","authors":"Konstantina Salveridou, Theodoros Tzamalis, Maika Klaiber-Hakimi, Sabine Haase, Stefan Diederich, Aristoteles Giagounidis","doi":"10.1155/crh/2925166","DOIUrl":"10.1155/crh/2925166","url":null,"abstract":"Engraftment syndrome (ES) is a rare but challenging posttransplant complication, presenting during or after absolute neutrophil recovery. It may easily be confused with febrile infectious complications since its main clinical features include fever, extensive skin rash, diarrhea, and atypical pulmonary infiltrates. Its pathophysiology and potential trigger factors remain not well understood. An immunosuppressive therapy with corticosteroids represents the standard treatment, and most cases present a rapid resolution of symptoms after treatment initiation. Still, in cases of severe presentation including periengraftment respiratory distress syndrome (PERDS), a mortality rate up to 21% has been described. In this case report, we report the case of a male patient after second autologous stem cell transplantation (SCT) with recurrent febrile episodes with no response to usual antibiotic regimens. Our patient developed diarrhea from Day 9 post-SC, escalating to a full-body rash and atypical pneumonia on and after Day 16. Our diagnosis was based on the Maiolino criteria, and a corticosteroid treatment was instituted. The symptoms ceased 2 days later, while we continued the treatment for a week. Physicians should be aware of this rare clinical entity, which can mimic other common posttransplant complications, hindering prompt diagnosis and treatment.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"2925166"},"PeriodicalIF":0.7,"publicationDate":"2026-08-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13504669/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148819863","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

Prolymphocytic Progression in Chronic Lymphocytic Leukemia Without Richter Transformation: An Eighteen-Year Clinical-Diagnostic Correlation. 无里氏转化的慢性淋巴细胞白血病的前淋巴细胞进展：18年临床诊断相关性

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Case Reports in Hematology Pub Date : 2026-08-21 eCollection Date: 2026-01-01 DOI: 10.1155/crh/9707876

Harini Venkatraman Ravisankar, Kavita Umrau, Liang Cheng, Magdalena Czader, Rohit Gulati

{"title":"Prolymphocytic Progression in Chronic Lymphocytic Leukemia Without Richter Transformation: An Eighteen-Year Clinical-Diagnostic Correlation.","authors":"Harini Venkatraman Ravisankar, Kavita Umrau, Liang Cheng, Magdalena Czader, Rohit Gulati","doi":"10.1155/crh/9707876","DOIUrl":"10.1155/crh/9707876","url":null,"abstract":"Chronic lymphocytic leukemia (CLL) is an indolent neoplasm of small, mature B lymphocytes characterized by clumped chromatin and scant cytoplasm. A minor subset of neoplastic cells may exhibit prolymphocytic morphology, with prominent nucleoli and moderate cytoplasm. An increased proportion of prolymphocytes in peripheral blood often correlates with an accelerated or aggressive disease course, distinct from Richter transformation (RT), which more commonly manifests as diffuse large B-cell lymphoma and requires intensified therapy. We report an 18-year clinical course of CLL in a 57-year-old man that evolved to prolymphocytic progression without transformation to large-cell lymphoma. Serial morphological assessments revealed a steady increase in prolymphocytes compatible with prolymphocytic progression in CLL and excluded RT. At the final assessment, 70%-80% of circulating lymphocytes exhibited prolymphocytic morphology, fulfilling criteria for prolymphocytic progression and reclassification as accelerated CLL. Accompanied immunophenotypic and IgH gene rearrangement findings confirmed CLL clonality. This case underscores the diagnostic and prognostic importance of recognizing prolymphocytic progression and integrating morphology with immunophenotypic and molecular data in the longitudinal management of CLL.","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"9707876"},"PeriodicalIF":0.7,"publicationDate":"2026-08-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13495556/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148799273","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

Reversible Neutrophilia Driven by Monoclonal Gammopathy of Clinical Significance Simulating Chronic Neutrophilic Leukemia: A Case Report. 临床意义模拟慢性中性粒细胞白血病的单克隆γ病驱动的可逆性中性粒细胞：1例报告。

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Case Reports in Hematology Pub Date : 2026-08-20 eCollection Date: 2026-01-01 DOI: 10.1155/crh/7405586

Éliott Larin, Lambert Busque, Genevieve Chabot-Roy, Sylvie Lesage, Gaspard Jadot, Richard LeBlanc, Jean Roy, Jean-Sébastien Claveau, Natasha Szuber

{"title":"Reversible Neutrophilia Driven by Monoclonal Gammopathy of Clinical Significance Simulating Chronic Neutrophilic Leukemia: A Case Report.","authors":"Éliott Larin, Lambert Busque, Genevieve Chabot-Roy, Sylvie Lesage, Gaspard Jadot, Richard LeBlanc, Jean Roy, Jean-Sébastien Claveau, Natasha Szuber","doi":"10.1155/crh/7405586","DOIUrl":"10.1155/crh/7405586","url":null,"abstract":"We encountered a 51-year-old male with a family history of acute myeloid leukemia who presented with persistent neutrophilia and splenomegaly progressing over eight years. Despite extensive workup, no myeloid driver mutation was identified and repeated bone marrow biopsies revealed no clonal myeloid disorder. However, an IgA kappa plasma cell clone was detected, consistent with monoclonal gammopathy of clinical significance (MGCS). Reactive neutrophilia secondary to the abnormal plasma cell clone was suspected, hypothesized to be mediated by paraneoplastic granulocyte colony-stimulating factor (G-CSF) secretion. G-CSF levels were elevated before treatment, supporting the hypothesis that it might induce neutrophilia. The patient received antimyeloma therapy and exhibited significant hematologic improvement: reduction in neutrophil count, normalization of G-CSF levels, and improvement in splenomegaly. This is the first report to describe improvement of neutrophilia following antimyeloma therapy, providing novel evidence that MGCS could simulate chronic neutrophilic leukemia (CNL).","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"7405586"},"PeriodicalIF":0.7,"publicationDate":"2026-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13494517/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148799250","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