Su Young Park, Amrryn Halari, Jessenia Palacio-Meadows, Nehal S Parikh, Danielle Bentsen, Surasak Puvabanditsin
{"title":"A Rare Case of Neonatal Kaposiform Hemangioendothelioma With Kasabach-Merritt Phenomenon Presenting as an Abdominal Mass Without Cutaneous Manifestations.","authors":"Su Young Park, Amrryn Halari, Jessenia Palacio-Meadows, Nehal S Parikh, Danielle Bentsen, Surasak Puvabanditsin","doi":"10.1155/crh/5825111","DOIUrl":"10.1155/crh/5825111","url":null,"abstract":"<p><p>Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm that typically presents in infancy or early childhood, classified as a locally aggressive/borderline vascular tumor. It most commonly involves superficial and deep soft tissues, often demonstrating infiltrative growth into adjacent muscle and bone, whereas retroperitoneal or intraabdominal presentations are less frequent. KHE is frequently complicated by Kasabach-Merritt phenomenon (KMP), a severe consumptive coagulopathy characterized by profound thrombocytopenia, microangiopathic hemolytic anemia, and hypofibrinogenemia with life-threatening sequelae. We report a case of a male neonate who developed KMP secondary to an abdominal KHE without overlying cutaneous involvement. Diagnosis was established through integration of clinical features, imaging findings, hematological evaluation, and confirmatory biopsy. This patient was managed with a multimodal regimen of sirolimus, corticosteroids, and vincristine, resulting in significant clinical improvement. This case underscores both the diagnostic complexity and therapeutic challenges of KHE complicated by KMP, particularly when lesions occur in deep anatomical sites. Given the rarity of this condition, current management strategies rely largely on expert consensus and case-based experience, though recent randomized trial and consensus have established sirolimus-based regimens as highly effective first-line therapy.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5825111"},"PeriodicalIF":0.7,"publicationDate":"2026-06-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13263245/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148253764","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}
Youngrok Park, Seonghan Lee, Jinhang Kim, Jeong-A Kim
{"title":"Allogeneic Stem Cell Transplantation in Patients With Mixed Chimerism Following Double Cord Blood Transplantation: A Case Report.","authors":"Youngrok Park, Seonghan Lee, Jinhang Kim, Jeong-A Kim","doi":"10.1155/crh/2052096","DOIUrl":"10.1155/crh/2052096","url":null,"abstract":"<p><strong>Introduction: </strong>Double-unit cord blood transplantation (dCBT) is an established strategy to enhance progenitor cell dose and accelerate hematopoietic recovery when a matched sibling or unrelated donor is unavailable. While dCBT typically results in the dominance of a single unit, the persistence of donor-donor mixed chimerism is an exceptionally rare clinical phenomenon. This case report describes a unique therapeutic strategy where persistent donor-donor chimerism following dCBT was utilized to redefine the recipient's HLA profile for a subsequent successful allogeneic transplantation.</p><p><strong>Case presentation: </strong>A 38-year-old patient with diffuse large B-cell lymphoma experienced disease relapse following autologous peripheral blood stem cell transplantation. After achieving a second remission through salvage chemotherapy, the patient underwent dCBT due to the absence of a compatible HLA-matched donor. Fifteen months posttransplantation, the disease relapsed again; however, further salvage therapy successfully induced a third remission. Notably, HLA analysis revealed persistent donor-donor mixed chimerism with one unit maintaining clear dominance. Because no suitable matches existed for the patient's native HLA type, the dominant donor's HLA genotype was utilized as the new target for donor selection. This shift facilitated the identification of a matched allogeneic peripheral blood stem cell transplantation (allo-PBSCT) donor.</p><p><strong>Conclusion: </strong>The patient subsequently underwent a second allo-PBSCT based on the dominant donor's HLA profile. The procedure resulted in robust engraftment and complete donor chimerism without the occurrence of graft-versus-host disease. The patient has remained in relapse-free survival for over 4 years. This case demonstrates that in the rare event of persistent donor-donor chimerism post-dCBT, the emergent dominant HLA genotype can serve as a viable and effective target for donor selection, providing a life-saving alternative for patients who lack native HLA-matched donors.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"2052096"},"PeriodicalIF":0.7,"publicationDate":"2026-06-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13263225/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148253731","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}
Neta Sternbach, Shahd Bishara, Morie A Gertz, Iuliana Vaxman
{"title":"Waldenström Macroglobulinemia Presenting With Hepatic Lesions: Case Report and Literature Review.","authors":"Neta Sternbach, Shahd Bishara, Morie A Gertz, Iuliana Vaxman","doi":"10.1155/crh/9908044","DOIUrl":"10.1155/crh/9908044","url":null,"abstract":"<p><p>Waldenström macroglobulinemia (WM) is a rare lymphoplasmacytic lymphoma characterized by bone marrow (BM) infiltration and monoclonal IgM secretion. Clinical manifestations most often reflect BM involvement, lymphadenopathy, or splenomegaly, while extramedullary disease is uncommon. Hepatic infiltration usually presents as diffuse hepatomegaly; focal hepatic lesions are exceedingly rare, with only two cases previously reported. We describe a 63-year-old woman with a history of celiac disease who presented with fatigue and pancytopenia. Laboratory testing showed elevated IgM with an IgM-λ monoclonal protein. Imaging revealed multiple hypodense hepatic lesions, splenomegaly, and small-volume lymphadenopathy. BM biopsy demonstrated lymphoplasmacytic lymphoma with <i>λ</i>-restricted plasma cells, and molecular studies were negative for MYD88 L265P and CXCR4 mutations. Liver biopsy showed granulomatous inflammation with necrosis, accompanied by IgM-positive, <i>λ</i>-restricted B cells clonally related to the BM infiltrate, consistent with WM involvement. Sarcoidosis and infectious causes were excluded. The patient received six cycles of bendamustine and rituximab (BR), leading to a reduction in hepatic lesions and IgM levels, though treatment was complicated by persistent cytopenias. This case represents the first reported instance of WM presenting with focal hepatic lesions associated with IgM paraproteinemia and a sarcoid-like granulomatous reaction. Recognition of such atypical presentations is essential, as they may mimic primary hepatic lymphoma, sarcoidosis, or infectious granulomatous disease. Comprehensive histopathological and molecular evaluation is critical for accurate diagnosis and appropriate therapy. Our report expands the spectrum of extramedullary WM and highlights the potential for favorable response to BR despite unusual presentation.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"9908044"},"PeriodicalIF":0.7,"publicationDate":"2026-06-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13250251/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148228429","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}
Philip Papayanis, Kara Conroy, Melissa Larson, Sunita Nathan, Celalettin Ustun
{"title":"Exploring the Origin of Cells in the Progression of Primary Myelofibrosis to Acute Myeloid Leukemia Case Report.","authors":"Philip Papayanis, Kara Conroy, Melissa Larson, Sunita Nathan, Celalettin Ustun","doi":"10.1155/crh/5472609","DOIUrl":"10.1155/crh/5472609","url":null,"abstract":"<p><p>Primary myelofibrosis (PMF) is the clonal proliferation of common myeloid progenitor (CMP) cells and can transform into acute myeloid leukemia (AML). The exact transformation process and cell lines involved are not well-understood. A patient with PMF received an allogenic hematopoietic stem cell transplant (alloHCT) from his major ABO incompatible sibling and developed pure red cell aplasia that resolved when his blood type switched to that of his donor's. He relapsed with AML 10 months after alloHCT; interestingly, his blood type remained his donor's type. This supports that AML in this patient with PMF stemmed from more committed progenitors (myeloblasts) than CMP.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5472609"},"PeriodicalIF":0.7,"publicationDate":"2026-06-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13249797/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148228465","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}
Mehdi Loukhnati, Benjamin Hebraud, Marine Cazaux, Cécile Borel, Pierre Bories, Jean Alain Martignoles, Miguel Granell, Pierre Cougoul, Jeremie Dion, Lucie Oberic, Anne Huynh, Illias Tazi, Aurore Perrot
{"title":"Outpatient Rescue With Stem Cells Boost for Refractory Hematotoxicity Following anti-BCMA CAR T-Cell Therapy: A Case Report.","authors":"Mehdi Loukhnati, Benjamin Hebraud, Marine Cazaux, Cécile Borel, Pierre Bories, Jean Alain Martignoles, Miguel Granell, Pierre Cougoul, Jeremie Dion, Lucie Oberic, Anne Huynh, Illias Tazi, Aurore Perrot","doi":"10.1155/crh/1558475","DOIUrl":"10.1155/crh/1558475","url":null,"abstract":"<p><p>Prolonged and refractory hematotoxicity is a severe complication of anti-BCMA CAR-T cell therapy, associated with substantial morbidity. When conventional supportive measures (transfusions and hematopoietic growth factors) fail, alternative strategies are urgently needed. We report the case of a 55-year-old man who developed persistent cytopenias after idecabtagene vicleucel. Reinfusion of cryopreserved autologous stem cells (stem cell boost), administered without conditioning chemotherapy, resulted in rapid and complete hematopoietic recovery after 14 days. This case highlights the potential of stem cell boost as a salvage approach for prolonged post-CAR-T hematotoxicity and underscores the need for prospective multicenter studies to standardize indications, timing, and procedures for this intervention.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"1558475"},"PeriodicalIF":0.7,"publicationDate":"2026-06-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13238238/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148200431","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}
Jordan S Nunnelee, Garrett A Weber, Nicholas P Bergeron, Katherine E Talbott, Andrew J Layman, David Dingli, Richard C Godby
{"title":"Paroxysmal Nocturnal Hemoglobinuria Presenting With Progressive Biventricular Thrombi.","authors":"Jordan S Nunnelee, Garrett A Weber, Nicholas P Bergeron, Katherine E Talbott, Andrew J Layman, David Dingli, Richard C Godby","doi":"10.1155/crh/5349577","DOIUrl":"10.1155/crh/5349577","url":null,"abstract":"<p><p>A 34-year-old previously healthy woman presented with right-sided, pleuritic chest pain three weeks after a viral respiratory infection. Labs revealed hemoglobin of 10.2 g/dL (nadir 7.0), platelets of 109 × 10<sup>9</sup>/L, LDH of 1129 U/L (Ref: 122-222 U/L), undetectable haptoglobin, indirect hyperbilirubinemia, and elevated reticulocytes. Troponins were elevated. CRP was 184 mg/L (Ref: ≤ 5 mg/L). Peripheral smear was without schistocytes. Direct antiglobulin testing was negative. Urinalysis showed microscopic hematuria and positive hemosiderin. Echocardiogram revealed normal left ventricular (LV) function with left lateral wall hypokinesis and a 2.5 × 3 cm LV thrombus. Cardiac MRI was concerning for myopericarditis and new right ventricular (RV) thrombus, later verified by biopsy. There was also an infarction of the RV endomyocardium. Anticoagulation was initiated. Bone marrow biopsy showed erythroid hyperplasia without clonal myeloid features. Flow cytometry showed 57% CD59 deficient red blood cells, 88% CD157 deficient monocytes, and 91% CD157 deficient neutrophils, consistent with a new diagnosis of paroxysmal nocturnal hemoglobinuria (PNH). She was urgently vaccinated against encapsulated organisms, treated with eculizumab, and given antibacterial prophylaxis. Follow-up imaging showed near resolution of thrombi; hemoglobin and hemolysis markers normalized. PNH is a clonal hematopoietic disorder causing complement-mediated hemolysis and thrombosis. While thrombosis typically affects atypical venous sites, intracardiac thrombi are rare. PNH was the patient's only identified prothrombotic factor. This is the first reported case of intracardiac thrombosis in a previously healthy patient with new-onset PNH. Early recognition and complement inhibition are critical to reduce hemolysis, thrombosis, and mortality in PNH.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"5349577"},"PeriodicalIF":0.7,"publicationDate":"2026-06-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13239058/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148200477","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}
Yvette von Aarburg, Astrid Beerlage, Darius Juskevicius, Cyrill Rütsche, Jan Dirks, Rainer Grobholz, Britta Hartmann, Roby Mathew, Katharina Leonards, Sabine Gerull
{"title":"Very Late Extramedullary Relapse of Acute Myeloid Leukemia 13 Years After Allogeneic Transplant: A Case Report.","authors":"Yvette von Aarburg, Astrid Beerlage, Darius Juskevicius, Cyrill Rütsche, Jan Dirks, Rainer Grobholz, Britta Hartmann, Roby Mathew, Katharina Leonards, Sabine Gerull","doi":"10.1155/crh/8840484","DOIUrl":"10.1155/crh/8840484","url":null,"abstract":"<p><p>Relapse of acute myeloid leukemia (AML) typically occurs within the first 3 years following treatment, with very late relapses being rare and often presenting extramedullary. We report a unique case of extramedullary AML relapse occurring 13 years after allogeneic hematopoietic stem cell transplantation. The patient developed a myelosarcoma, prompting molecular and histological investigation to determine the origin and mutational landscape of the disease. Next-generation sequencing (NGS) was performed on archived bone marrow samples from the initial AML diagnosis in 2007 and on the relapsed tumor tissue from 2021. The 2007 sample contained nine somatic variants, while the 2021 myelosarcoma harbored four variants. Only a single somatic mutation, a frameshift alteration in the <i>HNRNPK</i> gene, located on 9q21, was shared across both timepoints, suggesting a potential founder event and supporting the hypothesis of clonal evolution. Chimerism analysis and histopathological findings confirmed that the relapse originated from the patient rather than from donor-derived hematopoiesis. This case underscores the importance of long-term vigilance in AML survivors, especially in those receiving chronic immunosuppression, as impaired immune surveillance may allow for reemergence of malignant clones. The persistent <i>HNRNPK</i> mutation across both disease phases may point to a rare but critical driver of leukemogenesis and relapse. Our findings highlight the utility of comprehensive molecular profiling in elucidating disease dynamics and informing personalized monitoring strategies.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8840484"},"PeriodicalIF":0.7,"publicationDate":"2026-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13239328/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148200400","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}
Joseph Brandon Parker, Claire Wilson, Ricardo Parrondo, Muhamad Alhaj Moustafa
{"title":"Wandering Lymphoma: A Case of Wandering Spleen Secondary to Splenic Marginal Zone Lymphoma.","authors":"Joseph Brandon Parker, Claire Wilson, Ricardo Parrondo, Muhamad Alhaj Moustafa","doi":"10.1155/crh/8172177","DOIUrl":"10.1155/crh/8172177","url":null,"abstract":"<p><strong>Background: </strong>Splenic marginal zone lymphoma (SMZL) is a rare subtype of non-Hodgkin lymphoma. It often follows an indolent course presenting with splenomegaly. Treatment varies from clinically monitoring to splenectomy, chemotherapy, and rituximab treatments. Wandering spleen is an even rarer condition with less than 500 cases reported in the literature, often resulting from laxity in the suspensory ligaments supporting the spleen. Wandering spleen most commonly affects women of childbearing age and children. Treatment for this can be either nonsurgical or surgical with splenopexy or splenectomy.</p><p><strong>Case presentation: </strong>A 75-year-old female initially presented from Mexico for evaluation of splenomegaly and abdominal distension. Computer tomography (CT) scan of the abdomen and pelvis showed a pelvic mass, and labs showed leukocytosis with lymphocytic predominance. The pelvic mass was later confirmed as a wandering spleen. Flow cytometry on peripheral blood was diagnostic for SMZL. The patient was diagnosed with wandering spleen secondary to SMZL and treated with splenectomy.</p><p><strong>Conclusion: </strong>Wandering spleen is very rare and is typically associated with pregnancy, splenomegaly, or inborn errors of the ligaments supporting the spleen. This is only the fifth case reported in the literature of wandering spleen secondary to a lymphoma and the first reported with SMZL. This case offers a simultaneous presentation of two very rare conditions presenting in an atypical fashion. SMZL commonly presents in this age group; however, wandering spleen is rarely seen at this age. Additionally, this patient's first-line treatment for her SMZL was nontraditional with only a splenectomy with no rituximab administered due to the risk of torsion and infarction of the wandering spleen.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"8172177"},"PeriodicalIF":0.7,"publicationDate":"2026-05-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13215980/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148139297","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":"Sickle Cell Hepatic Vaso-Occlusive Crisis: A Case Report.","authors":"Bhimla Rambaran","doi":"10.1155/crh/7991112","DOIUrl":"10.1155/crh/7991112","url":null,"abstract":"<p><p>Sickle cell disease (SCD) is an inherited hemoglobinopathy caused by a point mutation in the β-globin gene, resulting in hemoglobin S polymerization under hypoxic conditions. This process leads to erythrocyte dehydration, impaired deformability, hemolysis, and recurrent vaso-occlusion, which may culminate in acute painful crises and progressive organ damage. Hepatic involvement is a recognized but underreported complication of SCD and can range from mild biochemical abnormalities to life-threatening hepatic vaso-occlusive crisis (VOC). This case report presents a 25-year-old woman, with homozygous sickle cell anemia who developed a rapidly progressive hepatic VOC following a mild respiratory illness. Her clinical course was marked by acute abdominal pain, severe transaminitis, cholestasis, escalating transfusion requirements, and radiologic evidence of hepatomegaly. Clinical and biochemical improvement was observed following a multimodal management approach that included supportive care, corticosteroids, blood transfusion, and the addition of intravenous metronidazole. This case highlights hepatic VOC as a serious manifestation of SCD and explores the potential role of metronidazole as an adjunctive therapy, given its immunomodulatory and anti-inflammatory properties. Further investigation is warranted to clarify its therapeutic role in sickle cell-related vaso-occlusive complications.</p>","PeriodicalId":46307,"journal":{"name":"Case Reports in Hematology","volume":"2026 ","pages":"7991112"},"PeriodicalIF":0.7,"publicationDate":"2026-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13201256/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148044142","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}
Reihaneh Sedghi, Ahmad Nazar Zadeh, Mohammadreza Bordbar, Omid Reza Zekavat, Shirin Parand
{"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}