bims-tremyl Biomed News
on Therapy resistance biology in myeloid leukemia
Issue of 2025–06–15
33 papers selected by
Paolo Gallipoli, Barts Cancer Institute, Queen Mary University of London



  1. bioRxiv. 2025 Jun 07. pii: 2025.06.06.657881. [Epub ahead of print]
      Resistance to combination regimens containing the BCL-2 inhibitor venetoclax in acute myeloid leukemia (AML) is a growing clinical challenge for this extensively utilized agent. We previously established the anti-leukemic properties of ceramide, a tumor-suppressive sphingolipid, in AML and demonstrated that upregulated expression of acid ceramidase (AC), a ceramide-neutralizing enzyme, supported leukemic survival and resistance to BH3 mimetics. Here, we report the anti-leukemic efficacy and mechanisms of co-targeting AC and BCL-2 in venetoclax-resistant AML. Analysis of the BeatAML dataset revealed a positive relationship between increased AC gene expression and venetoclax resistance. Targeting AC enhanced single-agent venetoclax cytotoxicity and the venetoclax + cytarabine combination in AML cell lines with primary or acquired venetoclax resistance. SACLAC + venetoclax was equipotent to the combination of venetoclax + cytarabine at reducing cell viability when evaluated ex vivo across a cohort of 71 primary AML patient samples. Mechanistically, SACLAC + venetoclax increased ceramide to levels that trigger a cytotoxic integrated stress response (ISR), ISR-mediated NOXA protein upregulation, mitochondrial dysregulation, and caspase-dependent cell death. Collectively, these data demonstrate the efficacy of co-targeting AC and BCL-2 in AML and rationalize targeting AC as a therapeutic approach to overcome venetoclax resistance.
    DOI:  https://doi.org/10.1101/2025.06.06.657881
  2. J Clin Oncol. 2025 Jun 13. JCO2500640
       PURPOSE: The development of targeted therapeutics has revolutionized treatment for elderly patients with AML. Two doublet regimens are approved in the frontline setting for intensive chemotherapy (IC)-ineligible AML: venetoclax (VEN) in combination with hypomethylating agent (HMA) therapy and azacitidine (AZA) plus ivosidenib (IVO) specifically for IDH1-mutated AML. Although both regimens have improved AML outcomes, most patients will either not respond to frontline therapy or relapse, with dismal salvage outcomes.
    METHODS: We herein report on 60 newly diagnosed IC-ineligible patients treated at our institution with triplet regimens for isocitrate dehydrogenase (IDH)-mutant AML. Patients received either AZA + VEN + IVO on NCT03471260 (IDH1-mutated patients only) or oral decitabine + VEN + IVO/enasidenib on NCT04774393 (arms for IDH1- and IDH2-mutant disease, respectively).
    RESULTS: The triplet regimens were well tolerated with low early mortality (n = 1 [2%] in 60 days) and a similar safety profile to HMA + VEN and isocitrate dehydrogenase inhibitor doublet regimens. The composite complete remission rate (CRc) was 92% (55/60), with an overall response rate of 95% (57/60). With a median follow-up of 27.4 months, the median overall survival (OS) has not yet been reached. The 2-year OS was 69% with a 2-year cumulative incidence of relapse of 24%. Patients with treated-secondary AML (tsAML) experienced inferior outcomes with a CRc of 71% (12/17) and a 2-year OS of 34%; the 2-year OS was 84% in patients without tsAML. Nineteen patients (32%) transitioned to stem cell transplant, and 51% remain on study.
    CONCLUSION: Given the excellent outcomes of IDH-triplet therapy for newly diagnosed, IC-ineligible IDH-mutant AML, further prospective studies comparing IDH-triplet versus IDH-doublet regimens are warranted.
    DOI:  https://doi.org/10.1200/JCO-25-00640
  3. Blood. 2025 Jun 10. pii: blood.2024028033. [Epub ahead of print]
      TET2 is among the most commonly mutated genes in both clonal hematopoiesis and myeloid malignancies, thus, the ability to identify selective dependencies in TET2 deficient cells has broad translational significance. Here, we identify regulators of Tet2 knockout (KO) hematopoietic stem and progenitor cell (HSPC) expansion using an in vivo CRISPR-Cas9 KO screen, in which nucleotide barcoding enabled large-scale clonal tracing of Tet2 deficient HSPCs in a physiological setting. Our screen identified candidate genes, including Ncoa4, that are selectively required for Tet2 KO clonal outgrowth compared to wild-type (WT). Ncoa4 targets ferritin for lysosomal degradation (ferritinophagy), maintaining intracellular iron homeostasis by releasing labile iron (Fe2+) in response to cellular demands. In Tet2-deficient HSPCs, increased mitochondrial ATP production correlates with increased cellular iron requirements, and in turn, promotes Ncoa4-dependent ferritinophagy. Restricting iron availability reduces Tet2 KO stem cell numbers, revealing a dependency in TET2-mutated myeloid neoplasms.
    DOI:  https://doi.org/10.1182/blood.2024028033
  4. Blood. 2025 Jun 11. pii: blood.2025028417. [Epub ahead of print]
      Clonal hematopoiesis of indeterminate potential (CHIP) is associated with increased mortality and malignancy risk, yet the determinants of clonal expansion remain poorly understood. We performed sequencing at >4,000x depth of coverage for CHIP mutations in 6,976 postmenopausal women from the Women's Health Initiative at two timepoints: the WHI baseline exam and approximately 16 years later at the Long Life Study (LLS) visit. Among 3,685 CH mutations detected at baseline (VAF ≥ 0.5%), 24% were not detected at LLS, 26% were micro-CH at LLS (0.5% ≤ VAF < 2%), and 50% were CHIP (VAF ≥ 2%). We confirmed that clonal expansion is highly dependent on initial clone size and CHIP driver gene, with SF3B1 and JAK2 mutations exhibiting the fastest growth rate. We identified germline variants in TERT, IL6R, TCL1A, and MSI2 that modulate clonal expansion rate. Measured baseline leukocyte telomere length showed differential effects on incident CHIP risk, with shorter baseline leukocyte telomere length predisposing to incident PPM1D mutations and longer baseline leukocyte telomere length favoring incident DNMT3A mutations. We discovered that the IL6R missense variant p.Asp358Ala specifically impairs TET2 clonal expansion, supported by direct measurements of soluble interleukin-6 receptor and interleukin-6. Faster clonal growth rate was associated with increased risk of cytopenia, leukemia, and all-cause mortality. Notably, CHIP clonal expansion rate mediated 34.4% and 43.7% of the Clonal Hematopoiesis Risk Score's predictive value for leukemia and all-cause mortality, respectively. These findings reveal key biological determinants of CHIP progression and suggest that incorporating growth rate measurements could enhance risk stratification.
    DOI:  https://doi.org/10.1182/blood.2025028417
  5. J Clin Oncol. 2025 Jun 12. JCO2500914
       PURPOSE: Azacitidine and venetoclax is a standard frontline treatment regimen for newly diagnosed older adults with AML; however, long-term outcomes remain poor. Revumenib is an oral menin inhibitor with clinical activity in AML patients with nucleophosmin-1 mutation (NPM1m) or lysine methyltransferase 2A rearrangements (KMT2Ar).
    METHODS: We conducted a phase I dose-escalation and expansion study of azacitidine, venetoclax, and revumenib at two dose levels (113 mg or 163 mg orally every 12 hours in combination with strong cytochrome P450 inhibitor azoles) in patients aged 60 years and older newly diagnosed with AML with NPM1m or KMT2Ar (ClinicalTrials.gov identifier: NCT03013998).
    RESULTS: Overall, 43 patients were enrolled and treated. There was no maximal tolerated dose identified. Differentiation syndrome was present in eight (19%) patients and QTc Fridericia prolongation was present in 19 (44%) patients, and neither required permanent discontinuation of revumenib. The overall response rate with an intention-to-treat population was 88.4% (95% CI, 74.9 to 96.1; NPM1m: 85.3%; KMT2Ar: 100%), the rate of composite complete remission (complete remission [CR] + CR with partial or incomplete hematologic recovery) was 81.4% (95% CI, 66.6 to 91.6; NPM1m: 79.4%; KMT2Ar: 88.9%), and the rate of CR was 67.4% (95% CI, 51.5 to 80.9; NPM1m: 65%; KMT2Ar: 78%). No patient had refractory disease after 1-2 cycles of treatment. The median time to first response was 28 days, and 84% of responders achieved remission within the first cycle. All 37 patients evaluated had no evidence of measurable residual disease by a centralized flow cytometry assay.
    CONCLUSION: In older adults newly diagnosed with NPM1m or KMT2Ar AML, the combination of azacitidine, venetoclax, and revumenib was able to be safely administered with high rates of CR and clinical activity.
    DOI:  https://doi.org/10.1200/JCO-25-00914
  6. bioRxiv. 2025 May 28. pii: 2025.05.23.655671. [Epub ahead of print]
      During cancer development, mutations promote gene expression changes that cause transformation. Leukemia is frequently associated with aberrant HOXA expression driven by translocations in nucleoporin genes or KMT2A , and mutations in NPM1 . How disparate mutations converge on this regulatory pathway is not understood. Here we demonstrate that mutant NPM1 (NPM1c) forms nuclear condensates in multiple human cell lines, mouse models, and primary patient samples. We show NPM1c phase separation is necessary and sufficient to coordinate the recruitment of NUP98 and KMT2A to condensates. Through extensive mutagenesis and pharmacological destabilization of phase separation, we find that NPM1c condensates are necessary for regulating gene expression, promoting in vivo expansion, and maintaining the undifferentiated leukemic state. Finally, we show that nucleoporin and KMT2A fusion proteins form condensates that are biophysically indistinguishable from NPM1c condensates. Together, these data define a new condensate underlying leukemias that we term coordinating bodies (C-bodies), and propose C-bodies as a therapeutic vulnerability.
    DOI:  https://doi.org/10.1101/2025.05.23.655671
  7. EMBO Rep. 2025 Jun 11.
      Hematopoietic stem and progenitor cells (HSPCs) polarize in contact with the bone marrow stromal cells constituting their niche. Given the role of cell polarity in protection against tumorigenesis and the importance of the niche in the progression of acute myeloid leukemias (AMLs), we investigated the polarization capacities of leukemic blasts. Using engineered micro-niches and centrosome position with respect to the contact site with stromal cells as a proxy for cell polarization, we show that AML cell lines and primary cells from AML patient blasts are unable to polarize in contact with healthy stromal cells. Exposure to AML patient-derived stromal cells compromises the polarization of healthy adult HSPCs and AML blasts from patients. When cultured in "bone-marrow-on-a-chip", stromal cells from a leukemic niche stimulate the migration of healthy HSPCs and AML blast. These results reveal the detrimental influences of both intrinsic transformation and extrinsic contact with transformed stromal cells on the polarization of AML blasts.
    Keywords:  Acute Myeloid Leukemia (AML); Artificial Niche; Bone-marrow-on-a-Chip (BMoC); Hematopoietic Stem and Progenitor Cells (HSPCs); Microwell
    DOI:  https://doi.org/10.1038/s44319-025-00466-w
  8. Haematologica. 2025 Jun 12. 0
      To investigate the clinical features and outcomes of measurable residual disease recurrence (MRD-R) by multiparameter flow cytometric in acute myeloid leukemia (AML). We retrospectively analyzed clinical characteristic, residual disease status and outcomes of 767 newly diagnosed AML patients achieving complete remission within two cycles of induction at our center. Totally, 171 (22.3%) patients experienced MRD-R during follow-up. Patients with MRD-R had inferior outcomes compared to those without MRD-R, with 3-year cumulative incidence of morphologic relapse (CIR), relapse-free survival (RFS) and overall survival (OS) at 63.6% vs. 30.6% (P.
    DOI:  https://doi.org/10.3324/haematol.2024.287119
  9. Haematologica. 2025 Jun 12. 0
      Acute myeloid leukemia (AML) patients with high PRDM16 expression frequently experience induction failure and have a poor prognosis. However, the molecular mechanisms underlying these clinical features remain elusive. We found that murine AML cells transformed by MLL::AF9 fusion and oncogenic short-isoform Prdm16 overexpression (hereafter, MF9/sPrdm16) exhibited resistance to cytarabine (AraC), but not to anthracycline, both in vitro and in vivo. Intriguingly, MF9/sPrdm16 cells displayed a gene expression signature of high oxidative phosphorylation (OxPHOS) and increased mitochondrial respiration. The inhibition of mitochondrial respiration with metformin or tigecycline abrogated AraC resistance in MF9/sPrdm16 cells via an energetic shift toward low OxPHOS status. Furthermore, sPrdm16 upregulated Myc and the glutamine transporter Slc1a5, activating TCA cycle and glutaminolysis. Of note, both OxPHOS and MYC-target gene signatures were significantly enriched in AML patient samples with high PRDM16 expression. Together, we showed that PRDM16 overexpression activates mitochondrial respiration through metabolic reprogramming via MYC-SLC1A5-Glutaminolysis axis, thereby conferring AraC resistance on AML cells. These results suggest that targeting mitochondrial respiration might be a novel treatment strategy to overcome chemoresistance in AML patients with high PRDM16 expression.
    DOI:  https://doi.org/10.3324/haematol.2024.287265
  10. Leuk Res. 2025 Jun 02. pii: S0145-2126(25)00084-0. [Epub ahead of print]155 107724
       BACKGROUND: Acute Myeloid Leukemia (AML) with FLT3-ITD mutations is associated with high post-transplant relapse rates. FLT3 inhibitor (FLT3i) maintenance therapy following allogeneic hematopoietic stem cell transplantation (allo-HCT) has emerged as a promising strategy to improve outcomes in this high-risk population.
    METHODS: We conducted a systematic review and meta-analysis of randomized controlled trials (RCTs) evaluating FLT3i maintenance therapy versus standard of care (SOC) after allo-HCT in patients with FLT3-ITD-mutated acute myeloid leukemia (AML). A comprehensive search of PubMed, Embase, CENTRAL, and ClinicalTrials.gov was performed in accordance with PRISMA guidelines. Primary outcomes included relapse-free survival (RFS), overall survival (OS), and FLT3i-related adverse events. Pooled hazard ratios (HRs) and relative risks (RRs) were calculated using the "meta" package in R (version 4.4.0).
    RESULTS: Four RCTs including 701 patients (ages 18-78) met inclusion criteria. FLT3i maintenance significantly reduced relapse (HR 0.50; 95 % CI: 0.34-0.74) and mortality (HR 0.63; 95 % CI: 0.44-0.91) compared to SOC. Hematologic toxicity (RR 2.12; 95 % CI: 1.67-2.70) and chronic GVHD (RR 1.18; 95 % CI: 1.00-1.41) were more frequent in the FLT3i group. Rates of acute GVHD (RR 1.05; 95 % CI: 0.78-1.41) and hepatotoxicity (RR 1.09; 95 % CI: 0.72-1.66) were comparable. Interestingly, skin toxicity was lower with FLT3i (RR 0.36; 95 % CI: 0.16-0.84).
    CONCLUSION: FLT3i maintenance significantly improves RFS and OS in FLT3-ITD-mutated AML post-allo-HCT, though at the cost of increased hematologic toxicity and chronic GVHD. Further studies are needed to define optimal agents, duration, and patient selection to balance efficacy with tolerability.
    Keywords:  Acute myeloid leukemia (AML); FLT3 inhibitors; Post-transplant maintenance therapy
    DOI:  https://doi.org/10.1016/j.leukres.2025.107724
  11. Leuk Res. 2025 Jun 05. pii: S0145-2126(25)00094-3. [Epub ahead of print]155 107734
      SETBP1 gene, located on 18q12.3, is a major oncogene in myeloid neoplasms. GATA2 gene, located on 3q21, is one of the six GATA transcription factors regulating gene expression via two conserved zinc finger domains (ZF). Previous data suggested that in patients with germline GATA2 mutation (m), acquisition of a somatic SETBP1 mutation (m) was associated with leukemic transformation among patients with AML, excess blast MDS and CMML. We hypothesize that the co-occurrence of SETBP1m and GATA2m have a unique impact on the clinical and molecular characteristics of myeloid neoplasms. After IRB approval, we retrospectively reviewed the charts of patients who had myeloid NGS panel results between 2016 and 2023. All patients with myeloid neoplasms who had either SETBP1m or GATA2m were included. One hundred sixty-eight patients had either SETBP1m and/or GATA2m; 105 patients had SETBP1m, 54 had GATA2m and 9 had both SETBP1m and GATA2m. Majority (66.1 %) were males with a median age of 71.3 years. At time of NGS, MDS/MPN was the most common diagnosis (32.1 %), followed by MDS (30 %) and AML (20.2 %). In SETBP1m/GATA2m patients, 7 GATA2m clustered in zinc finger 2 (ZF2) (77.8 %); higher than the ZF2 mutated cases among SETBP1wt/GATA2m (46.3 %, p = 0.1). Among SETBP1m/GATA2m, 77.8 % of patients had SRSF2 co-mutation, compared to 44.8 % among SETBP1m/GATA2wt (p = 0.08), and 27.8 % among SETBP1wt/GATA2m (p = 0.006). AML progression frequency for non-AML cases did not significantly differ between the 3 groups. Survival of SETBP1m/GATA2m patients was not worse compared to SETBP1wt/GATA2m or SETBP1m/GATA2wt.
    Keywords:  GATA2; Leukemic transformation; Myeloid neoplasms; SETBP1
    DOI:  https://doi.org/10.1016/j.leukres.2025.107734
  12. bioRxiv. 2025 May 28. pii: 2025.05.25.655566. [Epub ahead of print]
      One of the strongest signatures of aging is an accumulation of mutant mitochondrial DNA (mtDNA) heteroplasmy. Here we investigate the mechanism underlying this phenomenon by calling mtDNA sequence, abundance, and heteroplasmic variation in human blood using whole genome sequences from ∼750,000 individuals. Our analyses reveal a simple, two-step mechanism: first, individual cells randomly accumulate low levels of "cryptic" mtDNA mutations; then, when a cell clone proliferates, the cryptic mtDNA variants are carried as passenger mutations and become detectable in whole blood. Four lines of evidence support this model: (1) the mutational spectrum of age-accumulating mtDNA variants is consistent with a well-established model of mtDNA replication errors, (2) these mutations are found primarily at low levels of heteroplasmy and do not show evidence of positive selection, (3) high mtDNA mutation burden tends to co-occur in samples harboring somatic driver mutations for clonal hematopoiesis (CH), and (4) nuclear GWAS reveals that germline variants predisposing to CH (such as those near TERT , TCL1A , and SMC4 ) also increase mtDNA mutation burden. We propose that the high copy number and high mutation rate of mtDNA make it a particularly sensitive blood-based marker of CH. Importantly, our work helps to mechanistically unify three prominent signatures of aging: common germline variants in TERT , clonal hematopoiesis, and observed mtDNA mutation accrual.
    DOI:  https://doi.org/10.1101/2025.05.25.655566
  13. Haematologica. 2025 Jun 12.
      Erythropoiesis-stimulating agents (ESAs) achieve hematological improvement-erythroid (HIE) in only 30% of ESA-naïve lower risk myelodysplastic syndrome (LR-MDS) patients with anemia, highlighting the need for developing novel drugs or new treatment strategies to improve the outcome of these patients. We conducted this multicenter, single-arm trial to investigate the efficacy and safety of a triple regimen consisting of recombinant human erythropoietin (rhEPO), all-trans retinoic acid (ATRA) and testosterone undecanoate in patients with anemia due to lower-risk MDS based on Revised International Prognostic Scoring System. Eligible patients received rhEPO 10000 IU/day, oral ATRA 25 mg/m2/day and oral testosterone undecanoate 80 mg twice daily for 12 weeks. The primary endpoint was the proportion of patients achieving HI-E during 12 weeks of treatment. Of 52 eligible patients, 32 (61.5%, 95%CI 48.0%-73.5%) achieved HI-E, meeting the primary endpoint. Fifteen patients (65.2% [15/23]) with baseline serum erythropoietin ≤500 IU/L had HI-E versus 58.6% of those (17/29) with baseline serum erythropoietin >500 IU/L. More patients with very low or low risk had HI-E than those with intermediate risk (73.3% vs. 45.5%, P = 0.041) and fewer patients with mutated ASXL1 had HI-E than those with wildtype ASXL1 (33.3% vs. 70.0%, P = 0.040). The regimen had an acceptable safety profile compatible with individual agents. In conclusion, the triple regimen of rhEPO combined with ATRA and testosterone undecanoate attained HI-E in approximately 61.5% of patients regardless of baseline serum EPO levels, supporting further development of this regimen for LR-MDS patients with anemia. This study was registered at CHICTR.ORG.CN as ChiCTR2000032845.
    DOI:  https://doi.org/10.3324/haematol.2024.287055
  14. Leuk Lymphoma. 2025 Jun 13. 1-12
      First-line venetoclax (VEN) treatment for acute myeloid leukemia (AML) has high relapse rates, with limited evidence guiding subsequent therapy sequencing. This study evaluated the effectiveness of olutasidenib (OLU) versus ivosidenib (IVO) for patients withIDH1 relapsed/refractory (R/R) AML previously treated with VEN based therapy. Outcomes were compared between a subcohort of OLU-treated patients from the 2102-HEM-101 trial and an external control arm of IVO-treated patients from the Loopback Analytics electronic health record database. Entropy balancing was applied to align key prognostic variables. Risk differences (RD) for response/TI were estimated via logistic regression, and hazard ratios (HR) for OS via Cox regression. Following weighting, treatment with OLU versus IVO was associated with significantly higher rates of complete response (RD: 0.25; 95%CI: 0.01, 0.49), transfusion independence (RD: 0.27; 95%CI: 0.01, 0.53), and OS (HR: 0.33; 95%CI: 0.11, 0.94). Results suggest favorable effectiveness of OLU versus IVO in this population.
    Keywords:  Acute myeloid leukemia; ivosidenib; olutasidenib; real-world
    DOI:  https://doi.org/10.1080/10428194.2025.2514894
  15. bioRxiv. 2025 May 29. pii: 2025.05.28.656712. [Epub ahead of print]
      Hematopoietic stem and progenitor cells (HSPC) respond to infections, inflammation, and regenerative challenges using a collection of cellular and molecular mechanisms termed emergency myelopoiesis (EM) pathways. However, it remains unclear how various EM inducers regulate HSPCs using shared or distinct molecular mechanisms. Here, we generate a comprehensive and generalizable cell annotation method (HemaScribe) and a refined quantitative model of hematopoietic differentiation (HemaScape) using single cell RNA sequencing (scRNA-seq) of HSPCs, which we apply to a broad range of EM modalities. We uncover multiple strategies to enhance myelopoiesis acting at different levels of the HSPC hierarchy, which are associated with both unique and shared transcriptional response modules. In particular, we identify a myeloid progenitor-based module of EM engagement across diverse inflammatory challenges, which informs outcome in adult and pediatric human acute myeloid leukemia. Collectively, our work illuminates fundamental regulatory mechanisms in hematopoietic regeneration that have direct translational applications in disease contexts.
    HIGHLIGHTS: New HemaScribe method for hematopoietic progenitor annotation in scRNA-seq datasetsDifferent emergency myelopoiesis (EM) inducers act at distinct hematopoiesis levelsUnique and shared transcriptional response modules enacted by different EM inducersA myeloid progenitor EM module informs outcome in acute myeloid leukemia.
    eTOC BLURB: Swann et al. conduct comparative analysis of single cell RNA sequencing data from multiple emergency myelopoiesis models, finding that different perturbations act at various levels of the hematopoietic hierarchy and recruit distinct sets of molecular mechanisms to enhance myelopoiesis. In particular, they identify a conserved myeloid progenitor-based activation module across multiple disease conditions, which informs outcome in human acute myeloid leukemia.
    GRAPHICAL ABSTRACT:
    DOI:  https://doi.org/10.1101/2025.05.28.656712
  16. Nat Genet. 2025 Jun;57(6): 1478-1492
      Chromothripsis, the chaotic shattering and repair of chromosomes, is common in cancer. Whether chromothripsis generates actionable therapeutic targets remains an open question. In a cohort of 64 patients in blast phase of a myeloproliferative neoplasm (BP-MPN), we describe recurrent amplification of a region of chromosome 21q ('chr. 21amp') in 25%, driven by chromothripsis in a third of these cases. We report that chr. 21amp BP-MPN has a particularly aggressive and treatment-resistant phenotype. DYRK1A, a serine threonine kinase, is the only gene in the 2.7-megabase minimally amplified region that showed both increased expression and chromatin accessibility compared with non-chr. 21amp BP-MPN controls. DYRK1A is a central node at the nexus of multiple cellular functions critical for BP-MPN development and is essential for BP-MPN cell proliferation in vitro and in vivo, and represents a druggable axis. Collectively, these findings define chr. 21amp as a prognostic biomarker in BP-MPN, and link chromothripsis to a therapeutic target.
    DOI:  https://doi.org/10.1038/s41588-025-02190-6
  17. bioRxiv. 2025 May 27. pii: 2025.05.22.654334. [Epub ahead of print]
      Analysis of neither Diamond Blackfan anemia syndrome (DBAS) cohorts nor animal models has revealed a potential mechanism for the variable anemia phenotype, a key feature of this disease. Here, we utilized an established Rpl5 Skax23-Jus/+ murine DBAS model in order to study this dynamic erythropoiesis deficiency. These haploinsufficient mice exhibit variably penetrant craniofacial and cardiac defects mimicking the phenotypes of DBAS patients bearing RPL5 mutations. We additionally discovered that this specific heterozygous splicing mutation is pathogenic and leads to partial intron retention. By examining the transcriptome of fetal liver erythroid progenitors at E12.5, we demonstrate that the downregulation of erythroid differentiation pathways is consistent with the DBAS phenotype. We also identified dysregulated transcription of lipid metabolism genes with significant reduction in the abundance of Scd1 in a subset of E12.5 mutant embryos at risk for erythroid failure. SCD1, a key enzyme that converts saturated to monounsaturated fatty acids, has not been previously linked to erythropoiesis or DBAS. When anemia was induced in adult mice, pretreatment with an SCD1 inhibitor resulted in improved erythropoiesis. This analysis suggests a key role of lipid metabolism in the variable anemia penetrance in DBAS and highlights a previously unappreciated pathway that may serve as a potential target for drug development.
    Key Points: The variable anemia in Rpl5 Skax23-Jus/+ mice is triggered by intrinsic/extrinsic stress Rpl5 haploinsufficient murine and human erythroid progenitors exhibit a lipid metabolism signature with downregulation of Scd1 / SCD .
    DOI:  https://doi.org/10.1101/2025.05.22.654334
  18. Nature. 2025 Jun 11.
      Lipids are essential components of cancer cells due to their structural and signalling roles1. To meet metabolic demands, many cancers take up extracellular lipids2-5; however, how these lipids contribute to cancer growth and progression remains poorly understood. Here, using functional genetic screens, we identify uptake of lipoproteins-the primary mechanism for lipid transport in circulation-as a key determinant of ferroptosis sensitivity in cancer. Lipoprotein supplementation robustly inhibits ferroptosis across diverse cancer types, primarily through the delivery of α-tocopherol (α-toc), the most abundant form of vitamin E in human lipoproteins. Mechanistically, cancer cells take up lipoproteins through a pathway dependent on sulfated glycosaminoglycans (GAGs) linked to cell-surface proteoglycans. Disrupting GAG biosynthesis or acutely degrading surface GAGs reduces lipoprotein uptake, sensitizes cancer cells to ferroptosis and impairs tumour growth in mice. Notably, human clear cell renal cell carcinomas-a lipid-rich malignancy-exhibit elevated levels of chondroitin sulfate and increased lipoprotein-derived α-toc compared with normal kidney tissue. Together, our study establishes lipoprotein uptake as a critical anti-ferroptotic mechanism in cancer and implicates GAG biosynthesis as a therapeutic target.
    DOI:  https://doi.org/10.1038/s41586-025-09162-0
  19. Br J Haematol. 2025 Jun 12.
      Anti-thymocyte globulin in multi-refractory immune-mediated cytopenia is an effective treatment, with response rate as high as 65%. Younger patients, presenting with thrombocytopenia or neutropenia, and those with LGL and STAT3 mutations appear to be more likely responsive.
    Keywords:  amegakaryocytic thrombocytopenia; anti‐thymocyte globulin; immune cytopenia; immune neutropenia; pure red cell aplasia
    DOI:  https://doi.org/10.1111/bjh.20165
  20. N Engl J Med. 2025 Jun 13.
    Australasian Leukaemia and Lymphoma Group
       BACKGROUND: Allogeneic peripheral-blood stem-cell transplantation (SCT) from a matched related donor after myeloablative conditioning is the preferred curative treatment for patients with high-risk blood cancers. The combination of a calcineurin inhibitor and an antimetabolite remains standard care for graft-versus-host disease (GVHD) prophylaxis in these patients. Data from two randomized trials have suggested that post-transplantation cyclophosphamide can reduce the risk of GVHD after SCT from a matched donor when it is added to or replaces the antimetabolite. However, the effects of post-transplantation cyclophosphamide specifically after SCT from a matched related donor remain uncertain, and effects in the context of myeloablative conditioning are unclear.
    METHODS: We randomly assigned adults who were undergoing SCT from a matched related donor after myeloablative or reduced-intensity conditioning to receive either post-transplantation cyclophosphamide-cyclosporin (experimental prophylaxis) or cyclosporin-methotrexate (standard prophylaxis). The primary end point was GVHD-free, relapse-free survival.
    RESULTS: Among 134 patients who underwent randomization, 66 were assigned to receive experimental prophylaxis and 68 to receive standard prophylaxis. GVHD-free, relapse-free survival was significantly longer with experimental prophylaxis (median, 26.2 months; 95% confidence interval [CI], 9.1 to not reached) than with standard prophylaxis (median, 6.4 months; 95% CI, 5.6 to 8.3; P<0.001 by a log-rank test). GVHD-free, relapse-free survival at 3 years was 49% (95% CI, 36 to 61) with experimental prophylaxis and 14% (95% CI, 6 to 25) with standard prophylaxis (hazard ratio for GVHD, relapse, or death, 0.42; 95% CI, 0.27 to 0.66). The cumulative incidence of grade III to IV acute GVHD at 3 months was 3% (95% CI, 1 to 10) in the experimental-prophylaxis group and 10% (95% CI, 4 to 19) in the standard-prophylaxis group. At 2 years, overall survival was 83% and 71%, respectively (hazard ratio for death, 0.59; 95% CI, 0.29 to 1.19). The incidence of serious adverse events was similar in the two groups in the first 100 days after SCT.
    CONCLUSIONS: The combination of post-transplantation cyclophosphamide and a calcineurin inhibitor led to longer GVHD-free, relapse-free survival than standard prophylaxis after transplantation from a matched related donor with either reduced-intensity or myeloablative conditioning in patients with blood cancers. (Funded by the Australian Government Medical Research Future Fund and others; ALLG BM12 CAST Australian-New Zealand Clinical Trials Registry number, ACTRN12618000505202.).
    DOI:  https://doi.org/10.1056/NEJMoa2503189
  21. Br J Haematol. 2025 Jun 10.
      Blinatumomab consolidation was recently approved for patients with acute lymphoblastic leukaemia (ALL) who achieve measurable residual disease (MRD) negative remission based on the survival benefit yielded in the E1910 trial. The CALGB 10403 (C10403) is the most frequently used paediatric inspired regimen for young adults treated in the United States; however, data and guidance on how to best incorporate blinatumomab consolidation into the C10403 regimen are lacking. Here, we describe our experience of adding blinatumomab consolidation to the C10403 regimen per our institutional consensus. Thirty-one adult patients met inclusion criteria. The median age was 31 years and the majority were males (64.5%) and Hispanic (83.9%). The most common ALL subtype was Philadelphia-like (45.2%). The median follow-up was 13.7 months and the median overall survival was not reached (NR) (95% confidence interval [CI] NR-NR) with one death occurring post allogenic transplant and one relapse (3.2%). All-grade cytokine release syndrome and treatment-related neurological/psychiatric adverse event rates were 9.7% and 19.4%, respectively, and none of the patients permanently discontinued blinatumomab due to toxicity. The majority (84.6%) of patients were able to receive over half of their planned pegaspargase doses. The incorporation of blinatumomab consolidation to C10403 is safe and feasible in adults with ALL.
    Keywords:  CALGB 10403; MRD negative; acute lymphoblastic leukaemia; blinatumomab; consolidation
    DOI:  https://doi.org/10.1111/bjh.20200
  22. iScience. 2025 Jun 20. 28(6): 112646
      Hematopoietic stem/progenitor cell (HSPC) trafficking in and out of the bone marrow (BM) is essential for immune surveillance and hematopoietic balance. We previously identified Oncostatin M (OSM), primarily from myeloid cells, as a key regulator of HSPC traffic. Here, we show that neutrophils highly express and secrete OSM, especially when senesced. However, OSM is not required for neutrophil-mediated modulation of steady-state or circadian HSPC levels. Aged neutrophils returning to the BM reduce HSPC levels in peripheral blood (PB) independently of OSM, suggesting additional mechanisms beyond CXCL12/CXCR4 axis. While neutrophil transfer modulated HSPC kinetics in wild-type mice, OSM-secreting neutrophils failed to normalize elevated PB-HSPC levels in Osm -/- mice, though recombinant OSM successfully did. Macrophage depletion-induced HSPC egress was OSM-dependent, but neutrophil depletion elevated PB-HSPCs regardless of OSM. These findings reveal that neutrophils regulate HSPC migration via largely OSM-independent pathways, emphasizing the importance of cell-specific and context-dependent cues within the BM niche.
    Keywords:  Components of the immune system; Immunology
    DOI:  https://doi.org/10.1016/j.isci.2025.112646
  23. Nucleic Acids Res. 2025 Jun 06. pii: gkaf512. [Epub ahead of print]53(11):
      Mutation, deletion, or silencing of genes encoding cellular metabolism factors occurs frequently in human malignancies. Neomorphic mutations in isocitrate dehydrogenases 1 and 2 (IDH1/2) promoting the production of R-2-hydroxyglutarate (R-2HG) instead of α-ketoglutarate (αKG) are recurrent in human brain cancers and constitute an early event in low-grade gliomagenesis. Due to its structural similarity with αKG, R-2HG acts as an inhibitor of αKG-dependent enzymes. These include the JUMONJI family of lysine demethylases, among which KDM4A is particularly sensitive to R-2HG-mediated inhibition. However, the precise molecular mechanism through which inhibition of αKG-dependent enzymes by R-2HG promotes gliomagenesis remains poorly understood. Here, we show that treatment with R-2HG induces cellular senescence in a p53-dependent manner. Furthermore, expression of mutated IDH1R132H or exposure to R-2HG, which leads to KDM4A inhibition, causes telomeric dysfunction. We demonstrate that KDM4A localizes to telomeric repeats and regulates abundance of H3K9(me3) at telomeres. We show that R-2HG caused reduced replication fork progression, and that depletion of SMARCAL1, a helicase involved in replication fork reversal, rescues telomeric defects caused by R-2HG or KDM4A depletion. These results establish a model whereby IDH1/2 mutations cause R-2HG-mediated inhibition of KDM4A, leading to telomeric DNA replication defects, telomere dysfunction, and associated genomic instability.
    DOI:  https://doi.org/10.1093/nar/gkaf512
  24. Nature. 2025 Jun 11.
      Although cell-fate specification is generally attributed to transcriptional regulation, emerging data also indicate a role for molecules linked with intermediary metabolism. For example, α-ketoglutarate (αKG), which fuels energy production and biosynthetic pathways in the tricarboxylic acid (TCA) cycle, is also a co-factor for chromatin-modifying enzymes1-3. Nevertheless, whether TCA-cycle metabolites regulate cell fate during tissue homeostasis and regeneration remains unclear. Here we show that TCA-cycle enzymes are expressed in the intestine in a heterogeneous manner, with components of the αKG dehydrogenase complex4-6 upregulated in the absorptive lineage and downregulated in the secretory lineage. Using genetically modified mouse models and organoids, we reveal that 2-oxoglutarate dehydrogenase (OGDH), the enzymatic subunit of the αKG dehydrogenase complex, has a dual, lineage-specific role. In the absorptive lineage, OGDH is upregulated by HNF4 transcription factors to maintain the bioenergetic and biosynthetic needs of enterocytes. In the secretory lineage, OGDH is downregulated through a process that, when modelled, increases the levels of αKG and stimulates the differentiation of secretory cells. Consistent with this, in mouse models of colitis with impaired differentiation and maturation of secretory cells, inhibition of OGDH or supplementation with αKG reversed these impairments and promoted tissue healing. Hence, OGDH dependency is lineage-specific, and its regulation helps to direct cell fate, offering insights for targeted therapies in regenerative medicine.
    DOI:  https://doi.org/10.1038/s41586-025-09097-6
  25. Blood Adv. 2025 Jun 12. pii: bloodadvances.2024015626. [Epub ahead of print]
      The bone marrow represents the tumor microenvironment for many hematologic malignancies and a potentially critical site for alloimmunity following hematopoietic transplantation. Despite the importance of immune reconstitution (IR) post-transplant, marrow IR data are limited, and insights are largely derived from studies of peripheral blood (PB). We investigated lymphocyte IR longitudinally in marrow (n=110) and PB (n=115) samples from adults undergoing allogeneic transplantation for hematologic malignancies (n=33). This transplant cohort included a diverse representation of graft sources (mobilized peripheral blood, CD34-selected grafts, and umbilical cord blood) and degrees of HLA mismatch. Natural killer (NK) cells quickly expanded within the first 30 days post-transplant in both marrow and PB, but were then outnumbered by T cells in PB after day 100. In contrast, NK cells remained dominant in the marrow at day 100 (p<0.01, paired Wilcoxon signed-rank test), and thereafter marrow T and NK cell frequencies were similar throughout year-one. Tissue-specific features post-transplant included fewer regulatory T cells, more innate lymphoid cells, and increased CD69 expression on lymphocytes in marrow compared to PB. Furthermore, day 100 PD1 expression on marrow T cells was greater in non-relapsing patients than those who subsequently relapsed. These findings reveal persistent NK dominance of the marrow early post-transplant and suggest correlations between marrow immunity and clinical transplant outcomes.
    DOI:  https://doi.org/10.1182/bloodadvances.2024015626
  26. Cell Metab. 2025 Jun 09. pii: S1550-4131(25)00265-7. [Epub ahead of print]
      Metabolites are essential substrates for epigenetic modifications. Although nuclear acetyl-coenzyme A (CoA) constitutes a small fraction of the whole-cell pool, it regulates cell fate by locally providing histone acetylation substrate. Here, we report a nucleus-specific acetyl-CoA regulatory mechanism that can be modulated to achieve therapeutic cancer cell reprogramming. Combining phenotypic chemical screen, genome-wide CRISPR screen, and proteomics, we identified that the nucleus-localized pyruvate dehydrogenase complex (nPDC) is constitutively inhibited by the nuclear protein ELMSAN1 through direct interaction. Pharmacologic inhibition of the ELMSAN1-nPDC interaction derepressed nPDC activity, enhancing nuclear acetyl-CoA generation and reprogramming cancer cells to a postmitotic state with diminished cell-of-origin signatures. Reprogramming was synergistically enhanced by histone deacetylase 1/2 inhibition, resulting in inhibited tumor growth, durably suppressed tumor-initiating ability, and improved survival in multiple cancer types in vivo, including therapy-resistant sarcoma patient-derived xenografts and carcinoma cell line xenografts. Our findings highlight the potential of targeting ELMSAN1-nPDC as an epigenetic cancer therapy.
    Keywords:  ELMSAN1; HDAC; ISX9; acetyl-CoA metabolism; cancer therapy; compartmentalized metabolism; epigenetic reprogramming; nuclear metabolism; pyruvate dehydrogenase complex; therapeutic reprogramming
    DOI:  https://doi.org/10.1016/j.cmet.2025.05.009
  27. Blood. 2025 Jun 12. pii: blood.2024027239. [Epub ahead of print]
      Rapid CD137 upregulation on alloreactive T-cells upon allogeneic stimulation suggests that their selective elimination could prevent acute graft-versus-host disease (aGVHD) after allogeneic hematopoietic stem cell transplantation (HCT). Here, we developed a novel aGVHD prophylactic regimen consisting of a single dose of an anti-CD137 antibody-drug conjugate (CD137-ADC) administered on the day of transplant without additional immunosuppression. The CD137-ADC depleted both human and non-human primate (NHP) activated T-cells and proved highly effective in preventing xenogeneic aGVHD in mice receiving human peripheral blood mononuclear cells (PBMC), as well as in NHP undergoing MHC-haploidentical HCT. Flow cytometry analysis of NHP T-cells indicated specific depletion of activated PD-1+ CD4 and CD8 T-cells, while sparing naïve and PD-1-OX40+ memory T-cell subsets during the first week after HCT. CD137-ADC-treated NHP recipients demonstrated robust hematopoietic and immune reconstitution. Hallmarks of T-cell recovery after CD137-ADC, which were associated with long-term aGVHD-free survival, included reconstitution of CD4 memory T-cells expressing TRAIL, terminally-differentiated CD8 T-cells expressing CX3CR1, and CD4 FoxP3+ Tregs - cell types not expected to be involved in aGVHD pathogenesis. CD137-ADC-treated recipients demonstrated a higher risk of reactivation of rhLCV (the rhesus macaque EBV analogue), which was associated with reconstitution of follicular helper T-cells, interferon signaling-associated memory, and gamma-delta T-cell subsets. This reactivation was controllable with rituximab administration. These results document effective depletion of alloreactive T-cells and prevention of aGVHD following a single dose of CD137-ADC, suggesting that clinical translation should be carefully explored.
    DOI:  https://doi.org/10.1182/blood.2024027239
  28. Br J Clin Pharmacol. 2025 Jun 09.
       AIMS: Fedratinib is a potent, oral, Janus kinase inhibitor for the treatment of myelofibrosis (MF). This report describes exposure-response (E-R) analyses of fedratinib based on pooled data from phase 2/3 studies in patients with intermediate-2 or high-risk MF, with or without prior ruxolitinib exposure.
    METHODS: Pharmacokinetic (PK) exposures were derived from the population PK analysis. Efficacy endpoints included spleen volume reduction ≥35% (SVR35) and total symptom score reduction ≥50% (TSS response). Safety endpoints included grade ≥3 anaemia or thrombocytopenia, any-grade nausea/vomiting and diarrhoea. The E-R models were developed using logistic regression analyses.
    RESULTS: Fedratinib exposure was positively associated with SVR35 (odds ratio [OR], 38.2; 95% confidence interval [CI], 12.4-118; P < 0.001) and TSS response (OR, 20.8; 95% CI, 6.27-69.2; P < 0.001), after adjusting for covariates. Baseline spleen volume was inversely associated with SVR35 (P = 0.029). Prior ruxolitinib exposure was not associated with SVR35 (P = 0.090) or TSS response (P = 0.326). Although numerically higher incidence of adverse events was observed in patients with higher fedratinib exposure, there was no statistically significant association between fedratinib exposure and any safety related endpoints. Prior ruxolitinib exposure was associated with experiencing grade ≥3 thrombocytopenia (P = 0.004). Lower baseline haemoglobin level (<10 g/dL) and platelet count (<100 × 109/L) were associated with experiencing grade ≥3 anaemia (P < 0.001) and thrombocytopenia (P < 0.001), respectively. Antiemetic prophylaxis was associated with lower rates of nausea/vomiting (P < 0.001).
    CONCLUSIONS: Fedratinib exposure was positively associated with spleen volume reduction and TSS responses, without having significant impact on safety. Fedratinib 400 mg once daily is an appropriate dose for patients with MF regardless of ruxolitinib exposure.
    Keywords:  dose; exposure–response; fedratinib; myelofibrosis; prophylaxis
    DOI:  https://doi.org/10.1002/bcp.70118
  29. Blood. 2025 Jun 13. pii: blood.2025029352. [Epub ahead of print]
      Anemia of inflammation (AI) is the second most common form of anemia and is prevalent in patients with chronic inflammatory states, such as infection, autoimmunity, and cancer. Interleukin 6 (IL6) is well-known to induce the iron-sequestering hormone hepcidin, which results in iron-restricted anemia. The contributions of other pro-inflammatory cytokines such as tumor necrosis factor-α (TNFα) and interferon-γ (IFNγ) are less understood in the pathophysiology of AI. This study investigated the role of TNFα in a mouse model of AI by administering heat-killed Brucella Abortus (HKBA) to germline TNFα knockout (KO) mice. We hypothesized that TNFα possessed an important role in restoring steady-state erythropoiesis after inflammatory insult. TNFαKO injected with HKBA displayed a chronic anemia, with elevated pro-inflammatory IL12p40 and IFNγ cytokines that did not resolve. However, IFNγKO and TNFαKO/FNγKO double knock-out (DKO) mice showed reduced inflammation and anemia following HKBA administration. Since IFNγKO displayed normal serum TNFα and IL12p40 levels, we hypothesized that the persistent anemia was IFNγ-induced and TNFα was necessary for AI cessation. However, treatment with recombinant TNFα (rTNFα) accelerated death, while reducing IFNγ using an anti-IFNγ antibody (Ab) only briefly improved anemia. Only the combination of both the Ab and rTNFα together reversed the hyper-inflammatory phenotype, restored erythropoiesis, and prevented death of TNFαKO+HKBA mice. Our data provides compelling evidence for an anti-inflammatory role of TNFα that is necessary for the restoration of erythropoiesis and mitigation of pro-inflammatory IFNγ action in a mouse model of AI.
    DOI:  https://doi.org/10.1182/blood.2025029352
  30. bioRxiv. 2025 May 28. pii: 2025.05.24.655962. [Epub ahead of print]
      Allogeneic hematopoietic cell transplantation (alloHCT) is curative for various hematologic diseases but often leads to acute graft-versus-host disease (GVHD), a potentially life-threatening complication. We leverage GVHD as a uniquely tractable disease model to dissect complex T-cell-mediated pathology in 27 alloHCT recipients. We integrate pre-transplant identification of alloreactive T-cells with longitudinal tracking across blood and gut, using mixed lymphocyte reaction-based clonal "fingerprinting", TCR clonotyping, single-cell RNA/TCR sequencing, and spatial transcriptomics. Using DecompTCR, a novel computational tool for longitudinal TCR analysis, we uncover clonal expansion programs linked to GVHD severity and TCR features. Multi-omics profiling of gut biopsies reveals enrichment and clonal expansion of CD8⁺ effector and ZNF683(Hobit)⁺ resident memory T-cells, cytolytic remodeling of regulatory and unconventional T-cells, and localization of CD8⁺ effector T-cells near intestinal stem cells in crypt loss regions. This framework defines dynamic immune circuit rewiring and phenotypic plasticity with implications for biomarkers and therapies.
    Highlights: Persistent expansion of diverse alloreactive T cell clones is a hallmark of severe GVHDDecompTCR reveals dynamic clonal expansion programs linked to GVHD severity and clinical outcome CD8+ T cell clones exhibit phenotypic plasticity in vivo across intestinal tissue compartments in GVHD High-resolution spatial profiling shows CD8+ effector T cells localize near intestinal stem cell niches and drive epithelial injury in GVHD.
    DOI:  https://doi.org/10.1101/2025.05.24.655962
  31. iScience. 2025 Jun 20. 28(6): 112634
      In silico analysis revealed an elevated expression of cytohesin-4 (CYTH4) in acute myeloid leukemia (AML) cells, correlating with a poorer prognosis for AML patients. However, its role in AML is not fully understood. Our study using loss-of-function assays identified CYTH4 as an oncogene promoting leukemogenesis. Silencing CYTH4 in MV4-11 and THP-1 cells reduced cell proliferation and colony formation, and induced apoptosis and cell-cycle arrest at G0/G1, whereas overexpression had no significant impact. CYTH4 silencing also increased chemosensitivity to cytarabine. In a THP-1 xenograft model, CYTH4 silencing slowed AML progression and reduced leukemic cell homing and infiltration. Mechanistically, CYTH4 silencing inhibited PI3K/AKT pathway by lowering PIK3R5 and decreased ARF6-GTP levels, as confirmed by pull-down assays. Overexpression of PIK3R5 and AKT activation via SC-79 successfully countered the cellular dysfunctions from CYTH4 silencing. Thus, CYTH4 may play a role in AML progression, and targeting its pathway could be a promising anti-leukemic treatment strategy.
    Keywords:  Cancer; Cell biology
    DOI:  https://doi.org/10.1016/j.isci.2025.112634
  32. Blood. 2025 Jun 09. pii: blood.2025028617. [Epub ahead of print]
      Although allogeneic HCT is a leading treatment approach for myeloid malignancies, challenges in its immune biology and in treatment approaches remain. In the past decade major advances in the knowledge of mechanisms of graft-versus host disease (GvHD) has allowed development of new treatments both for GvHD prophylaxis and treatment. However, although successes did occur, failure did as well. Reasons for failure can be linked either to incomplete understanding of the pathophysiology of GVHD, or, in some cases, to errors in the design of clinical trials. Better GVHD prophylaxes and disease control have likely led to decreased non relapse mortality (NRM). However, while NRM rates have decreased, rates of relapse of the original malignancy have not significantly improved. Our current understanding of the biology of the graft-versus leukemia effect (GvL) still lag beyond that of GvHD, and treatment approaches to manipulate the GvL effect remain limited. The reasons for such a lag are numerous, but improved knowledge of the biology of hematological malignancies open the gate to new developments, providing that we can better understand the interplay between the immune system with leukemic clones. From a therapeutical perspective, much attention has been paid to the results from randomized clinical trials and from a biological perspective on recent discoveries, especially in the human setting. The objective of this perspective is to analyze what are the current challenges in the biology and treatment of GvHD and GvL and to provide a personal view on how some biological and therapeutic issues could be approached.
    DOI:  https://doi.org/10.1182/blood.2025028617