bims-tremyl Biomed News
on Therapy resistance biology in myeloid leukemia
Issue of 2026–06–28
28 papers selected by
Paolo Gallipoli, Barts Cancer Institute, Queen Mary University of London



  1. Haematologica. 2026 Jun 25.
      The European LeukemiaNet (ELN) 2022 classification categorized both t(9;11)(p21.3;q23.3) and isolated del(7q) in acute myeloid leukemia (AML) as intermediate-risk when treated with intensive chemotherapy. However, their prognostic relevance in the context of allogeneic hematopoietic cell transplantation (allo-HCT) needs further validation. This retrospective, registry-based analysis from the EBMT assessed outcomes in adults with AML who underwent allo-HCT in first complete remission between 2010 and 2022. In the first cohort, data from 141 patients with t(9;11) were analyzed, of whom 23% had additional adverse cytogenetic abnormalities (ACA), primarily complex karyotype. Most had de novo AML (72%), had received myeloablative conditioning (57%), and peripheral blood stem cells (88%). After a median follow-up of 3 years, there were no significant differences in 2-year relapse incidence (22% vs. 18.2%, p=0.85), leukemia-free survival (66% vs. 76%, p=0.42), or overall survival (72% vs. 75%, p=0.68) between patients with non-adverse t(9;11) and those with additional ACA. The second cohort included 250 patients: 84 with del(7q), 95 with monosomy 7, and 71 with del(5q), and all without additional ACA. Most had de novo AML (59%) and had received reduced-intensity conditioning (65%). After similar follow-up, survival outcomes did not differ significantly across the groups (2-year leukemia-free survival: 61% vs. 59% vs. 52% for del(7q), monosomy 7 and del(5q), respectively). In conclusion, these findings suggest that the prognostic value of t(9;11) as intermediate-risk remains consistent in the setting of allo-HCT regardless of additional ACA, whereas del(7q), even without additional ACA, confers a risk comparable to monosomy 7 and del(5q).
    DOI:  https://doi.org/10.3324/haematol.2025.289080
  2. Cell Death Dis. 2026 Jun 25.
      MCL-1 (myeloid cell leukemia-1) promotes survival and confers therapeutic resistance in acute myeloid leukemia (AML), particularly in high-risk subtypes harboring KMT2A rearrangements (KMT2A-r). Clinical trials involving patients with hematological malignancies treated with MCL-1 inhibitor monotherapy have been hampered by dose-limiting toxicity and poor response rates. Therefore, we sought to identify combinatorial treatment approaches to enhance the efficacy of MCL-1 inhibitors with the goal of improving response rates and limiting toxicities. Here, we report the inhibition of electron transport chain (ETC) complex I (CI) function as a synthetic lethal partner for MCL-1 inhibition. Co-targeting CI and MCL-1 synergistically reduces the viability in AML cell lines and patient-derived xenograft (PDX) samples in vitro, while significantly prolonging survival in mice bearing PDX AML, indicating the preclinical potential for this combinatorial therapy. These findings provide a mechanistic rationale and preclinical evidence for dual inhibition of MCL-1 and CI as a therapeutic strategy, offering a potential path to overcome resistance to single-agent MCL-1 inhibitors and improve outcomes for patients with high-risk AML. Mechanistically, we reveal that CI inhibition induces the activation of the integrated stress response, resulting in ATF4 activation downstream of the eIF2α kinase, HRI (Heme-regulated inhibitor). HRI activation via CI inhibition is dependent on the mitochondrial stress messenger, DELE1. Together, these results indicate that co-inhibition of MCL-1 and ETC CI function has the potential for improving responses in patients with KMT2A-r AML.
    DOI:  https://doi.org/10.1038/s41419-026-09037-w
  3. Blood. 2026 Jun 26. pii: blood.2026033125. [Epub ahead of print]
      Non‑hematopoietic stromal cells are essential regulators of hematopoiesis; however, their contribution to leukemogenesis and immune dysfunction remains poorly defined. Here, we identified fibroblast‑derived fibroblast growth factor 18 (FGF18) as a novel stromal cytokine that reprograms leukemia-immune interactions. Single-cell RNA sequencing of the bone marrow (BM) niche during acute myeloid leukemia (AML) revealed the upregulation of Fgf18 in stromal fibroblasts. Administration of recombinant FGF18 accelerated AML progression, whereas fibroblast-specific Fgf18 depletion markedly delayed disease development and improved the survival of mice. We performed a pooled CRISPR-Cas9 screen in AML cells and identified FGFR3 signaling as a critical mediator of leukemic fitness in the FGF18‑rich microenvironment. Genetic loss of Fgfr3 in AML cells recapitulated the effects of FGF18 deficiency and limited leukemic expansion in vivo. Mechanistically, FGF18 binds to its receptor, FGFR3, on AML cells, activating the AKT-mTOR signaling pathway and inducing interleukin (IL)-6 production. IL‑6 acts autocrinely to reinforce leukemic signaling and paracrinely to activate fibroblast JAK-STAT3 signaling, thereby amplifying stromal fibroblast FGF18 expression and forming a feed‑forward loop that suppresses CD8⁺ T‑cell effector function and weakens anti‑leukemic immunity. Clinically, elevated FGF18 expression correlates with poor prognosis in AML patients. To therapeutically target this malignant crosstalk, we generated an FGF18‑neutralizing antibody that disrupted the stromal-leukemia feedback loop, restored CD8⁺ T cell effector function, and synergized with anti-PD-1 therapy to elicit durable anti‑leukemic immunity in vivo. Collectively, these findings identify FGF18-dependent stromal-leukemia crosstalk that drives AML progression and immune dysfunction, highlighting FGF18 neutralization as a potential therapeutic strategy.
    DOI:  https://doi.org/10.1182/blood.2026033125
  4. Blood. 2026 Jun 23. pii: blood.2025032681. [Epub ahead of print]
      We assessed the addition of the tyrosine kinase inhibitor Quizartinib, following intensive chemotherapy and as maintenance, in patients aged >60 years with AML or high-risk MDS, regardless of FLT3 mutation status. 463 patients (median age 68yrs) were randomised (1:1) to receive Quizartinib 40mg or not for 14 days immediately following chemotherapy courses 2 and 3, plus 28 additional days; those allocated Quizartinib were further randomised (1:1) to either 12 additional 28-day maintenance courses (long Quizartinib), or no further treatment (short Quizartinib). Median follow-up was 76 months. 314 patients were FLT3 wild type (WT); 116 had FLT3 mutations. The primary endpoint, overall survival (OS) unselected by FLT3 status, showed no significant difference (HR 0.99, 95% CI 0.79-1.24, p=0.937) and there was an increase in non-relapse mortality with Quizartinib (HR 1.64, 95% CI 1.04-2.59, p=0.032). In a pre-planned subgroup analysis, FLT3-mutated patients who received Quizartinib had significantly improved OS (HR 0.59, 95% CI 0.37-0.93, p=0.024) due to reduced relapse risk (HR 0.57, 95% CI 0.35-0.91, p=0.017) with greater benefit in the short Quizartinib group (HR 0.49, 95% CI 0.24-1.02, p=0.055). In FLT3-WT patients there was no survival benefit and no reduction in relapse risk. No significant differences were seen in time to hematologic count recovery or in the duration of hospitalisation. The most observed grade 3/4 adverse events were febrile neutropenia. In conclusion, the addition of Quizartinib to intensive chemotherapy, delayed until chemotherapy course 2, prolonged OS in older patients with FLT3-mutated AML but did not improve OS in non-FLT3 selected patients. ISRCTN-31682779, EudraCR-2013-002730-21.
    DOI:  https://doi.org/10.1182/blood.2025032681
  5. Clin Cancer Res. 2026 Jun 22.
       PURPOSE: Outcomes in patients with relapsed/refractory (R/R) acute myeloid leukemia (AML) remain discouraging despite rapid expansion of treatment paradigms over the past decade. Clinicians must often decide between conventional salvage treatment regimens or enrollment on clinical trials.
    EXPERIMENTAL DESIGN: We retrospectively identified 657 patients with AML in second treatment salvage and compared the outcomes of those who received investigational agents (N=354) versus conventional salvage regimens (N=303).
    RESULTS: Conventional salvage was associated with higher response rates (CR/CRi rate: 22% vs 12%, P=0.0008) and higher early mortality rates (60-day mortality: 17% vs 7%, P=0.0002). There was no difference in rates of bridging to allogeneic stem cell transplant (7% vs 10%, P=0.2), event free survival (EFS; median: 1.5 months vs 2.0 months, P=0.9) or overall survival (OS; median: 4.1 months vs 4.5 months, P=0.8). A subgroup analysis of conventional regimens showed improved EFS (median EFS: 3.6 months vs 1.4 months, P=0.004) and a trend towards improved OS (median OS: 7.5 months vs 3.8 months, P=0.06) with the addition of venetoclax in venetoclax-naïve patients.
    CONCLUSION: Enrollment of R/R patients on clinical trials is a suitable alternative to conventional salvage regimens, offering novel therapeutic approaches to improve upon the historically dismal outcomes in this setting.
    DOI:  https://doi.org/10.1158/1078-0432.CCR-26-0951
  6. J Hematol Oncol. 2026 Jun 26.
       BACKGROUND: Systemic mastocytosis (SM) is a spectrum of hematologic disorders characterized by accumulation of atypical mast cells (MCs) in extracutaneous organs. SM with an associated hematologic neoplasm (SM-AHN), the most frequent subtype of advanced SM, is predominantly associated with myeloid neoplasms, consistent with shared clonal architecture. Because of its rarity and heterogeneity, robust outcome data aligned with contemporary classifications are needed to inform risk stratification.
    METHODS: We analyzed the 10th data wave of the European Competence Network on Mastocytosis registry (34 European centers and 1 US center). SM and AHN diagnoses followed the 2022 World Health Organization classification. Baseline characteristics and overall survival (OS) were compared between patients with myeloid SM-AHN and SM without AHN (SM-no-AHN). Within SM-AHN, outcomes were analyzed by SM component (advanced: aggressive SM [ASM] or MC leukemia [MCL] vs. non-advanced: bone marrow mastocytosis, indolent SM, or smoldering SM) and AHN subtype.
    RESULTS: Among 3,925 patients with SM, 467 (11.9%) had myeloid SM-AHN. Myelodysplastic/myeloproliferative neoplasms (MDS/MPN) were the most frequent AHN category (41.1%), with chronic myelomonocytic leukemia as the most common subtype (29.6%). Compared with SM-no-AHN, SM-AHN patients were older, more often male, and less frequently had skin involvement. The SM component was advanced in 55.0% of SM-AHN versus 5.8% of SM-no-AHN (p < 0.001). OS was shorter in SM-AHN than SM-no-AHN (median 36.1 vs. 340.9 months; p < 0.001) and was reduced across SM subtypes, including ASM (31.0 vs. 81.1 months) and MCL (7.1 vs. 23.2 months). Within SM-AHN, advanced SM-AHN had shorter OS than non-advanced SM-AHN (28.3 vs. 70.9 months; p < 0.001). Median OS differed by AHN subtype (57.0 months in SM-MPN, 35.7 in SM-MDS, 34.2 in SM-MDS/MPN, and 14.7 in SM associated with acute myeloid leukemia; p < 0.001). In multivariable analysis, non-advanced SM (vs. advanced) remained independently associated with improved OS (hazard ratio = 0.44; p < 0.001).
    CONCLUSIONS: SM-AHN is associated with reduced survival compared with SM without AHN across SM subtypes. Outcomes in SM-AHN are driven primarily by the aggressiveness of the mastocytosis component, supporting recognition and classification of SM in patients with concomitant myeloid neoplasms, given approved KIT-targeted tyrosine kinase inhibitors for advanced SM.
    Keywords:  Acute myeloid leukemia; Associated myeloid neoplasm; Chronic myelomonocytic leukemia; KIT D816V; Overall survival; SM-AHN; Systemic mastocytosis
    DOI:  https://doi.org/10.1186/s13045-026-01819-1
  7. Cell Commun Signal. 2026 Jun 23. pii: 370. [Epub ahead of print]24(1):
       BACKGROUND: Myeloproliferative neoplasms (MPN) comprise a heterogenous group of hematological malignancies that include polycythemia vera (PV), essential thrombocythemia (ET), and primary myelofibrosis (PMF). Current therapeutic strategies rely on cytoreductive approaches that mitigate disease burden and thromboembolic risk but are not curative. Allogeneic stem cell transplantation remains the only curative option, underscoring the need for novel therapeutic strategies. We previously identified hypoxia-inducible factor 1 (HIF-‍‍1) as a selective vulnerability in JAK2V617F-positive cells, but the underlying metabolic mechanisms remain incompletely defined.
    METHODS: In vitro studies utilized 32D cells transduced with an empty vector control, Jak2WT, or Jak2V617F. To evaluate metabolic dependencies, CRISPR-Cas9 was used to generate Slc2a1 (GLUT1) and Slc2a3 (GLUT3) knockout clones, which were subsequently characterized via RNA sequencing, extracellular flux analysis, and cellular fitness assays (proliferation, viability, and apoptosis). Pharmacological targeted inhibition of GLUT1/3 was evaluated in human JAK2V617F-mutated post-MPN AML cell lines (SET-2, HEL), primary patient-derived cells and a Jak2V617F knock-in mouse model. Combinatorial efficacy was assessed using the JAK1/2 inhibitor ruxolitinib.
    RESULTS: JAK2V617F induced HIF-1-dependent metabolic reprogramming, characterized by increased glycolytic flux and oxidative metabolism. Complete abrogation of glucose uptake occurred only upon combined loss of GLUT1 and GLUT3 in Jak2V617F cells, revealing functional redundancy between these transporters that sustains enhanced glycolysis. Disruption of glucose uptake selectively induced stress-associated transcriptional programs and replication stress, triggering an S-phase arrest that culminated in apoptosis and impaired viability, specifically in Jak2V617F cells. In vivo, pharmacological inhibition of HIF-1 or GLUT induced a reorganization of erythropoiesis to the spleen but did not ameliorate core disease features. In contrast, in vitro GLUT inhibition robustly reduced cell viability in human SET-2 and HEL cell lines and impaired proliferation, viability, and colony formation in patient-derived PBMCs.
    CONCLUSIONS: Collectively, these findings establish HIF-1-driven glucose metabolism as a metabolic vulnerability in JAK2V617F-positive MPN. The selective exhaustion of patient-derived clones defines the HIF-1-GLUT1/3 axis as a central, targetable bottleneck. These data provide a mechanistic rationale for further investigation of HIF-1 or GLUT inhibitors, suggesting that targeting this fundamental requirement may help overcome clinical limitations to achieve disease modification and eradicate the malignant clone.
    Keywords:  CALR; GLUT1; GLUT3; Glucose transport; HIF-1; JAK2V617F; MPL; MPN; Metabolic reprogramming
    DOI:  https://doi.org/10.1186/s12964-026-03018-4
  8. Blood. 2026 Jun 23. pii: blood.2025032631. [Epub ahead of print]
      Dysregulation of galectins and global protein glycosylation have been reported in various cancers, but their role in myeloproliferative neoplasms (MPNs) have remained incompletely understood. We performed single-cell RNA sequencing (scRNA-seq) which revealed significant enrichment of galectin genes in MPN monocytes. Cell-cell communication analysis predicted monocytes as a pivotal mediator of cell interactions and galectin signaling as a robust input/output pathway for monocytes. We identified elevated expression of galectin-1 (Gal-1, LGALS1) in monocytes from both human MPN samples and mouse models. Mass cytometry (CyTOF) profiling of MPN blood samples demonstrated that recombinant galectin-1 (rGal-1) significantly increased levels of multiple inflammatory cytokines in monocytes without affecting other cell types. Incubation of CD14+ monocytes from MPN patients with rGal-1 led to markedly increased transcription and secretion of inflammatory cytokines. Mechanistically, we uncovered crosstalk between the TLR4 and Gal-1 signaling pathways, as evidenced by protein 3D modeling and co-immunoprecipitation. Notably, TLR4 inhibition abrogated Gal-1 mediated proinflammatory effects in monocytes. We further identified NF-κB-dependent signaling as a key downstream effector of Gal-1, as reporter assays demonstrated rGal-1 mediated activation of NF-κB signaling in a TLR4-dependent manner. We corroborated these findings in vivo in a murine model driven by MPLW515L in which genetic abrogation of Lgals1 ameliorated key MPN disease features, including leukocytosis and splenomegaly. Additionally, Gal-1 inhibition suppressed carrageenan-induced thrombosis and inflammation in vivo. In summary, we identify Gal-1 enrichment in MPN monocytes as a driver of monocyte-mediated inflammation through TLR4 and NF-κB activation and uncover a novel therapeutic avenue for MPNs.
    DOI:  https://doi.org/10.1182/blood.2025032631
  9. NPJ Precis Oncol. 2026 Jun 20.
      FLT3-ITD mutations are among the most frequent mutations in acute myeloid leukemia (AML) and are associated with poor prognosis and high relapse rates. Despite extensive use of FLT3 inhibitors, resistance caused by secondary TKD mutations remains a major challenge. Therefore, developing next-generation FLT3 inhibitors with broad-spectrum activity against drug-resistant mutations is of considerable clinical value. We systematically evaluated the antileukemic activity of a novel type I FLT3 inhibitor, SGI-7079, in the context of FLT3-ITD and secondary resistance mutations. SGI-7079 inhibited the proliferation of AML cell lines harboring FLT3-ITD and multiple resistant mutations, including D835Y/V/F, Y842C, and F691L, in vitro. Mechanistically, SGI-7079 bound stably to FLT3, inhibited FLT3 phosphorylation and downstream STAT5, AKT, and ERK signaling, and induced G1-phase arrest and apoptosis. In FLT3-ITD leukemia mouse models, including Ba/F3-FLT3-ITD and MOLM13 cell-derived xenografts, SGI-7079 reduced leukemia burden and splenic infiltration without significant toxicity. Notably, it overcame gilteritinib resistance in FLT3-ITD-F691L and quizartinib resistance in FLT3-ITD-D835Y in vivo. SGI-7079 also exhibited potent activity in primary AML cells from FLT3-ITD-positive patients, outperforming gilteritinib and quizartinib. These findings demonstrate that SGI-7079 provides broad-spectrum inhibition across diverse FLT3 mutation contexts, with distinct advantages against drug-resistant mutations, particularly F691L, highlighting its potential for clinical translation.
    DOI:  https://doi.org/10.1038/s41698-026-01550-7
  10. Blood. 2026 Jun 23. pii: blood.2025032484. [Epub ahead of print]
      The transition of hematopoietic stem cells (HSCs) from quiescence to lineage commitment requires precise post-transcriptional control, yet the contribution of mRNA isoform regulation remains poorly defined. Here, we identify a translationally controlled splicing program that contributes to HSC fate decisions. Using activity-based signatures of 305 splicing regulators, we uncover widespread post-transcriptional modulation of the spliceosome in stem and progenitor cells. The branch-point recognition factor Sf1 emerges as a key node, regulated by a conserved structured 5' UTR that cooperates with the RNA-binding protein Igf2bp2 to control its translation. Disrupting this cis-trans module reduces Sf1 protein synthesis and skews differentiation toward stem and erythroid programs. Mechanistically, Sf1-dependent alternative splicing remodels 5' UTRs of hematopoietic and DNA damage response genes, altering their translation and modulating DNA damage resolution. Together, these findings reveal an unrecognized translational layer controlling spliceosome activity and link RNA regulons, alternative splicing, and HSC fate determination.
    DOI:  https://doi.org/10.1182/blood.2025032484
  11. Sci Transl Med. 2026 Jun 24. 18(855): eadu0167
      Acute myeloid leukemia (AML) is an aggressive leukemia with high rates of chemoresistance and relapse. Patients with AML undergoing induction chemotherapy often have delayed erythropoietic recovery and febrile neutropenia. Infection is a leading cause of mortality in this population. There is an unmet need to improve disease-specific outcomes in patients with AML undergoing cytotoxic chemotherapy. AML, at diagnosis, is characterized by increased levels of circulating iron due to erythroid block and cell death, which is further aggravated upon intensive chemotherapy. We hypothesized that iron, particularly toxic non-transferrin-bound iron (NTBI), can be redistributed away from AML cells and bacteria into nonmalignant transferrin receptor (CD71)-expressing cells by administering exogenous iron-free apotransferrin (apoTF). Using mouse models of AML, we show that mice treated with human apoTF had decreased NTBI and increased bone marrow erythropoiesis and B cell responses. ApoTF treatment resulted in normalization of bone marrow blood vessels and reduction of lipid peroxidation in endothelial cells. Crucially, apoTF combined with chemotherapy resulted in a reduction of AML cells and in improved survival, which was dependent on adaptive immunity. We established a murine model of Escherichia coli sepsis in leukemic mice receiving chemotherapy. We show that apoTF administration increased the survival of E. coli-infected mice. Mechanistically, apoTF treatment decreased the levels of circulating C-C motif chemokine ligand 2 (CCL2) and interleukin-6 through reduced expression of CCL2 in lipopolysaccharide-polarized macrophages. Our results demonstrate an overall benefit of iron redistribution induced by transferrin in combination with cytotoxic chemotherapy in AML.
    DOI:  https://doi.org/10.1126/scitranslmed.adu0167
  12. Blood. 2026 Jun 22. pii: blood.2025030831. [Epub ahead of print]
      The distinctive milieu of the bone marrow (BM), known as the BM niche, supports hematopoietic stem cells (HSCs) and serves as a foundation for hematopoietic regeneration. Myeloablative stress disrupts not only hematopoietic stem and progenitor cells but also essential BM niche components, including endothelial cells (ECs) and mesenchymal stromal cells (MSCs); disruption of the latter impairs efficient hematopoietic recovery. However, therapeutic strategies targeting niche restoration remain largely underdeveloped. Here, we demonstrate that the Hippo pathway effectors YAP/TAZ are critical for enabling ECs and MSCs to respond to BM injury, and that YAP/TAZ activation accelerates BM niche recovery, thereby promoting hematopoietic regeneration. We found that YAP/TAZ are rapidly activated in both MSCs and ECs following myeloablative stress, maintaining MSC multipotency and orchestrating vascular remodeling. Mechanistically, YAP/TAZ function as transcriptional hubs in MSCs, regulating key transcriptional factors such as Ebf1 and Ebf3. This regulation preserves MSC identity by preventing osteogenic and fibrogenic differentiation while promoting the expression of hematopoietic factors such as Cxcl12 and angiogenic factors. In addition, YAP/TAZ signaling in MSCs and ECs appeared to coordinately remodel sinusoidal vessels following BM injury. These YAP/TAZ-mediated niche responses are essential for HSC retention and hematopoietic regeneration following diverse myelosuppressive therapies. Notably, pharmacological activation of YAP/TAZ enhances BM niche reorganization and augments hematopoietic regeneration following myeloablative therapies. These findings establish YAP/TAZ as central regulators of BM niche resilience, providing a rationale for niche-targeted therapeutic strategies to enhance hematopoietic regeneration.
    DOI:  https://doi.org/10.1182/blood.2025030831
  13. Leukemia. 2026 Jun 25.
      Bone marrow endothelial cells (ECs) form specialized vascular niches that support hematopoietic stem and progenitor cells (HSPC), yet the molecular regulators of this function remain incompletely defined. Here, we identify the Rho GTPase-activating protein Dlc1 as an essential regulator of developmental vasculogenesis, and adult bone marrow vascular niche integrity. Endothelial-specific deletion of Dlc1 caused embryonic lethality with severe vascular defects. In adult mice, inducible EC-specific Dlc1 deletion disrupted the bone marrow vascular architecture, associated with a significant reduction of multipotent hematopoietic progenitors and myeloid-lineage cells. Single-cell RNA sequencing revealed transcriptional reprogramming of Dlc1-deficient ECs, with the emergence of cell subsets displaying altered transcriptional profiles and disrupted expression of key niche signals, including Kitl, Cxcl12, and Pdgfb. Ligand-receptor interactome analysis demonstrated impaired EC-to-HSPC communication, and the hematopoietic cells exhibited transcriptional features of metabolic stress and reduced biosynthetic activity. These findings position Dlc1 as a central regulator of developmental vasculogenesis, adult vascular architecture in the bone marrow, and EC-derived hematopoietic cell support. This work uncovers a previously unrecognized role for Dlc1 in coupling vessel function to hematopoietic cell output, with implications for understanding bone marrow failure syndromes and targeting endothelial dysfunction in hematologic diseases.
    DOI:  https://doi.org/10.1038/s41375-026-03013-1
  14. J Clin Oncol. 2026 Jun 25. JCO2600113
    HLALOSS Consortium
       PURPOSE: For patients with hematologic malignancies, relapse is the leading cause of death after allogeneic hematopoietic cell transplantation (allo-HCT). Frequently, relapses are explained by immune evasion through alterations of human leukocyte antigens (HLAs), but determinants and clinical consequences remain poorly defined.
    METHODS: We analyzed 533 relapses of hematologic cancers after allo-HCT from different donor types, conducted at 27 centers worldwide. Genomic loss of mismatched HLA (HLA loss) was assessed using a newly developed next-generation sequencing pipeline. Clinical and immunogenetic factors associated with HLA loss were evaluated. Using HLA data from approximately 5 million individuals, a web-based tool to infer HLA incompatibility phasing was developed.
    RESULTS: HLA loss occurred in 15.6% of relapses, with significant variation according to donor type (28.7% haploidentical family, 7.2% unrelated adult, 2.7% cord blood, P < .0001). The distribution of HLA mismatches across the patient's haplotypes, predicted through the phasing tool, was strongly associated with HLA loss, with an incidence of 27.6% when HLA mismatches were in the same haplotype, compared with 5.4% if present on different haplotypes (P < .0001). HLA loss affected postrelapse outcomes, abrogating the efficacy of original donor lymphocyte infusions, with significant survival advantage by second allo-HCT from a different donor.
    CONCLUSION: The likelihood of HLA loss varies significantly according to the number and positioning of HLA mismatches between patient and donor. A newly developed phasing tool enables reliable prediction of its risk, supporting informed donor selection. Routine assessment of HLA loss at relapse is warranted, as it critically affects the success of immunologic salvage therapies.
    DOI:  https://doi.org/10.1200/JCO-26-00113
  15. Leukemia. 2026 Jun 25.
      Chronic myeloid leukemia (CML) is defined by the reciprocal translocation between chromosomes 9 and 22 [t(9;22)], resulting in the constitutively active BCR::ABL1 fusion oncogene in hematopoietic stem cells (HSC). CML is mostly diagnosed in older adults. However, it can also occur in children, adolescents, and young adults. Therapeutic strategies for chronic phase-CML (CML-CP) have drastically improved overall survival and disease relapse, including the development of small molecular tyrosine kinase inhibitors (TKI). However, therapeutic approaches in CML still face challenges and potential downfalls, including resistance, intolerance, persistence of leukemic cells (LCs) that risk the progression to blast phase-CML (CML-BP), and other challenges in effectiveness. Furthermore, standardized treatment by pediatric oncologists follows guidelines designed for adult CML patients and does not consider differences in host or disease biology in pediatric patients. Although some patients can achieve treatment-free remission (TFR), most patients require lifelong TKI therapy. Thus, an attractive goal in CML, particularly in children with CML, who may need several decades of TKI treatment, is to identify and target immunoregulatory pathways and restoration of immune surveillance mechanisms to promote strong immune responses and TFR success. In this review, we discuss the immunological mechanisms that contribute to the development, progression and control of CML, including supporting evidence of currently approved or investigated therapeutic approaches.
    DOI:  https://doi.org/10.1038/s41375-026-03006-0
  16. Leukemia. 2026 Jun 26.
      NPM1 mutation (NPM1c)-driven acute myeloid leukemia (AML) is characterized by the sequestration of nuclear proteins and chromatin hijacking. Current treatment strategies targeting NPM1c AML are often indirect, which might lead to toxicity and resistance. In this study, we developed an allele-specific siRNA that selectively silences NPM1c while preserving the function of wild-type NPM1. This approach inhibited proliferation and promoted myeloid differentiation in NPM1-mutated AML cells in vitro. Notably, the systemic delivery of chemically optimized siNPM1c via lipid nanoparticles (LNPs) significantly reduced leukemogenesis, and enhanced the therapeutic efficacy of the menin inhibitor revumenib and overcame its resistance in vivo. Mechanistically, NPM1c recruits KAT7 and p300, driving leukemogenic transcription and establishing a pathogenic acetylome and open chromatin state. KAT7 recruitment is crucial for the retention of this complex on chromatin. Targeting NPM1c with siRNA disrupts this interaction, reverses the oncogenic epigenetic landscape, and suppresses transcription. Our findings demonstrate that silencing NPM1c effectively suppresses AML by dismantling a pathogenic KAT7/p300-dependent acetylome, highlighting the potential of LNP‑delivered siNPM1c as a promising therapeutic strategy, either as a monotherapy or in combination with menin inhibition.
    DOI:  https://doi.org/10.1038/s41375-026-03014-0
  17. Hemasphere. 2026 Jun;10(6): e70406
    An international study on behalf of GenoMed4all, Synthema, and icMDS consortia
      Myelodysplastic syndromes (MDS) are clinically and biologically diverse disorders, emphasizing the need for personalized treatment approaches. The International Working Group for Prognostication of MDS (IWG_PM) recently introduced a molecular classification, referred to as the MDS taxonomy, that categorizes patients into 16 subgroups based on 21 gene mutations, 6 cytogenetic abnormalities, and loss of heterozygosity (LOH) at TP53 and TET2 loci. This study sought to validate and enhance the clinical relevance of the MDS taxonomy by analyzing a large retrospective cohort (n = 5136) and transcriptomic data from a prospective cohort (n = 477). The taxonomy successfully identified subgroups with distinct clinical characteristics and disease progression patterns. However, incorporating gene interactions from taxonomy subgroups did not improve the prognostic performance of the Molecular International Prognostic Scoring System (IPSS-M). We further assessed whether the taxonomy could guide management in patients receiving disease-modifying therapies. Except for the "TP53-complex" subgroup, taxonomy classifications were not predictive of hypomethylating agent response or transplant outcomes. Nonetheless, they correlated with overall survival, suggesting that while both IPSS-M and the taxonomy capture disease biology, other non-genetic factors may influence treatment response. RNA sequencing confirmed the biological distinctiveness of the taxonomy groups. Transcriptomic profiling of CD34+ bone marrow cells revealed unique, homogeneous gene expression patterns, particularly within the AML-like, biTET2, SF3B1, and TP53-complex subgroups. Further integration of multi-omics data may refine MDS classification, improving clinical decision-making and guiding the development of targeted therapies.
    DOI:  https://doi.org/10.1002/hem3.70406
  18. J Clin Invest. 2026 Jun 23. pii: e202856. [Epub ahead of print]
      Obesity is increasingly implicated in hematopoietic malignancies, yet its role in mutation-driven myeloid leukemias remains unclear. Using UK Biobank data from over 440,000 individuals, we found obesity traits including elevated BMI and waist-to-hip ratio were associated with type 2 diabetes, increased plasma IL-17A (interleukin-17A), reduced GLP-1R (glucagon like peptide 1 receptor) expression, and heightened risk of myeloid malignancies. Transplantation of protein tyrosine phosphatase non-receptor type 11, PTPN11 (Shp2E76K/+) mutant hematopoietic stem/progenitors into obese mice demonstrated that metabolic inflammation accelerates leukemogenesis via myeloid cell expansion, lipid metabolic rewiring, IL-17A activation, and accumulation of M2-like tumor-associated macrophages (TAMs), accompanied by T-cell exhaustion and impaired antigen presentation. Notably, dual therapy with an anti-IL-17A antibody and a GLP-1R agonist reversed these effects, by reducing M2-like TAMs, restoring Ciita-dependent antigen presentation, Tyk2-mediated IFNγ signaling, reactivated T-cell responses, and reducing leukemic burden. These findings establish IL-17A driven, metabolism-coupled immunosuppression as a mechanistic link between obesity and SHP2-mutant myeloid leukemias, highlighting a tractable therapeutic strategy for high-risk obese patients.
    Keywords:  Hematology; Hematopoietic stem cells; Inflammation; Leukemias; Obesity; Oncology
    DOI:  https://doi.org/10.1172/JCI202856
  19. Blood Adv. 2026 Jun 25. pii: bloodadvances.2026019960. [Epub ahead of print]
      Acute myeloid leukemia (AML) is characterized by recurrent chromosomal abnormalities that form the basis of the European LeukemiaNet (ELN) risk classification and serve as essential determinants of prognosis and therapeutic decision-making. Conventional metaphase karyotyping remains the diagnostic gold standard for detecting these abnormalities; however, its utility is limited by longer turnaround times, often delaying critical clinical management. Here, we present a long-read sequencing-based (LRS) low-coverage whole genome sequencing (lcWGS) approach using Oxford Nanopore Technology as a rapid and scalable alternative for cytogenetic profiling. A total of 100 diagnostic AML samples were analyzed, comprising 50 retrospectively selected cases with known adverse-risk cytogenetics and 50 prospectively enrolled patients with clinically defined de novo AML. LcWGS demonstrated robust analytical performance, identifying chromosomal aberrations with 93% sensitivity, specificity, and overall accuracy, respectively. Complex karyotypes were reliably detected, with an area under the curve (AUC) of 0.971. Reproducibility was validated through replicate sequencing at two independent laboratories (R=0.99). LcWGS-derived estimates of clone size showed moderate correlation with conventional cytogenetic assessments (R=0.54). Patients with complex karyotypes identified by lcWGS exhibited significantly shorter overall and relapse-free survival, closely mirroring outcomes defined by conventional karyotyping and underscoring the value of lcWGS for risk stratification. Median turnaround time from sample receipt to bioinformatics interpretation was approximately 34 hours, enabling delivery of actionable karyotype results within 72 hours. These findings establish lcWGS as a rapid, reproducible, and accurate platform for detecting clinically relevant chromosomal abnormalities, addressing a critical need for timely risk stratification and treatment initiation in AML.
    DOI:  https://doi.org/10.1182/bloodadvances.2026019960
  20. Nat Cancer. 2026 Jun;7(6): 944-963
      De novo purine synthesis is required to maintain tumor growth; however, its impact on therapy resistance remains unclear. Here, through a dynamic BH3-priming-based CRISPR screen, we found that deletion of ADSS2, which encodes the adenylosuccinate synthase 2 enzyme essential for adenosine monophosphate (AMP) synthesis, re-sensitizes drug-resistant acute myeloid leukemia cells to venetoclax and a myeloid cell leukemia-1 (MCL1) inhibitor. Single-cell sequencing analysis of patient-derived xenograft samples revealed a positive association of high ADSS2 activity in TP53-mutant cells with poor responsiveness to venetoclax. We developed an ADSS2 antagonist, which synergized with BH3 mimetics to promote apoptosis in preclinical models. Mechanistically, sensitization mediated by ADSS2 targeting correlated with downregulated AMP-activated protein kinase activity, which in resistant cells promotes mitophagy to eliminate damaged mitochondria after BH3 mimetic treatment. These data show that AMP synthesis promotes BH3 mimetic resistance and that combining ADSS2 targeting with BH3 mimetics represents a promising anti-cancer approach.
    DOI:  https://doi.org/10.1038/s43018-026-01184-5
  21. Leukemia. 2026 Jun 26.
      Relapsed acute leukemia can be difficult to salvage. An uncommon but increasingly recognized and aggressive mechanism of relapse involves lineage switch. In lineage switch, the immunophenotype of the leukemia at relapse differs from the immunophenotype at initial diagnosis, with the underlying genetic driver(s) conserved, confirming a clonal relationship. Lineage switch is most common-and was first recognized-in B-cell acute lymphoblastic leukemia with KMT2A rearrangement, which often relapses as acute myeloid leukemia. In an era where antigen-targeted therapies, including chimeric antigen receptor T-cells and bispecific T-cell engagers, are increasingly utilized and thus apply selective antigen pressure, this may increase the incidence of lineage switch across different leukemia subtypes. Patients with lineage switch have dismal outcomes and optimal therapies remain unknown, thus there is a large unmet need to better understand the biology, define the diagnosis, and determine the therapeutic approaches to lineage switch. Here, we address these needs providing a review of the current biology of lineage switch, the relationship to different genetic subtypes and present definitions and recommendations for immunophenotypic and molecular monitoring.
    DOI:  https://doi.org/10.1038/s41375-026-03020-2
  22. Nucleic Acids Res. 2026 Jun 22. pii: gkag613. [Epub ahead of print]54(12):
      Erythropoiesis is a highly coordinated process governed by lineage-defining transcription factors. While several master regulators have been characterized, many additional factors expressed in erythroid progenitors remain poorly understood. Here, we identify the epithelial-to-mesenchymal transition (EMT)-associated transcription factor ZEB1 as a previously unrecognized regulator of erythroid maturation. ZEB1 is expressed in murine and human erythroid progenitors, and its loss impairs proliferation and disrupts the erythroid transcriptional network. Mechanistically, ZEB1 interacts with KLF1 and governs both its expression dynamics and nuclear localization. In ZEB1-deficient cells, GATA2 silencing is delayed due to failure of KLF1 to properly accumulate in the nucleus, impairing KLF1-mediated repression of GATA2 and perturbing the GATA2-KLF1 regulatory switch that drives late-stage erythropoiesis. Consistent with these findings, ZEB1 downregulation hampers maturation of primary human CD34⁺-derived erythroid cells. Together, our findings establish KLF1 as a repressor of GATA2, and ZEB1 as a critical enforcer of erythroid transcriptional circuitry, and demonstrate that erythroid cells recruit EMT-associated factors to ensure lineage completion.
    DOI:  https://doi.org/10.1093/nar/gkag613
  23. NEJM Evid. 2026 Jul;5(7): EVIDra2600012
      AbstractRecent discoveries have uncovered roles for telomere length, at both short and long extremes, as a driver of inherited disease risk in children and adults. For short telomere length the predominant phenotype is degenerative, with immunodeficiency, bone marrow failure, and pulmonary disease being most common. Short telomere syndrome genetics inform clinical decisions and have transformed understanding of the etiology, natural history, and treatment of common diseases such as idiopathic pulmonary fibrosis. At the other extreme, ultra-long telomere length predisposes to neoplasia including lympho- and myeloproliferative disease. Individuals with mutations that lengthen telomeres may show features of youthfulness such as delayed hair graying but paradoxically are at risk for benign and malignant neoplasia, which are associated with aging. Their earliest events are traceable in the blood as premature onset of clonal hematopoiesis which shows complete penetrance with aging. Here, we review the genetic basis, pathophysiology, and contrasting phenotypes of Mendelian short and long telomere syndromes, emphasizing how they inform clinical decisions as well as our understanding of the fundamentals of aging and cancer.
    DOI:  https://doi.org/10.1056/EVIDra2600012
  24. J Biol Chem. 2026 Jun 20. pii: S0021-9258(26)02156-3. [Epub ahead of print] 113284
      While all-trans retinoic acid (ATRA) is successfully used to treat acute promyelocytic leukemia (APL), therapeutic resistance remains a major clinical challenge, highlighting an urgent need to identify the regulators of ATRA efficiency. In the present study, we analyzed paired bone marrow samples of APL patients and found that miR34a isoforms (-3p and -5p) were significantly elevated at complete remission compared to initial diagnosis. Overexpression of pre-miR34a enhanced ATRA-induced myeloid differentiation in APL cells as well as in xenograft models. Mechanistically, we found that miR34a promoted G1/S cell cycle arrest and reduced the protein level of CDK6, a validated target of miR34a-5p. Furthermore, bioinformatic analysis predicted ISOC1 was a target of miR34a-3p, and dual-luciferase reporter assays validated the direct binding of miR34a-3p to the 3' UTR of ISOC1. Gene set enrichment analysis (GSEA) illustrated "hematopoietic cell lineage" pathway was significantly enriched in acute myeloid leukemia (AML) patients with low levels of ISOC1. Knockdown of ISOC1 enhanced ATRA-induced myeloid differentiation, and rescue experiments confirmed that miR34a regulated ATRA efficiency by targeting ISOC1. Moreover, ERK inhibition blocked the ability of miR34a to enhance ATRA-induced myeloid differentiation. Consistently, ERK inactivation significantly reduced miR34a-3p levels while increased ISOC1 levels in the presence of ATRA. Taken together, Our findings demonstrated that miR34a enhanced ATRA-induced myeloid differentiation by targeting CDK6 via miR34a-5p and ISOC1 via miR34a-3p, ERK signaling promoted ATRA-induced myeloid differentiation through miR34a-3p/ISOC1 axis. Targeting this regulatory axis may provide a novel combinatorial strategy to improve the therapeutic efficacy of ATRA in APL.
    Keywords:  CDK6; ERK; ISOC1; acute promyelocytic leukemia; all trans retinoic acid; miR34a
    DOI:  https://doi.org/10.1016/j.jbc.2026.113284
  25. Blood Red Cells Iron. 2026 Jun;2(2): None
      HFE encodes a nonclassical major histocompatibility complex class I molecule involved in iron homeostasis. Among the known HFE variants, p.Cys282Tyr in homozygosity accounts for >90% of cases of hemochromatosis (HFE-HC), a genetic disorder characterized by systemic iron overload. HFE plays a well-established role in systemic iron control through hepatic regulation of hepcidin expression. However, its putative role within the hematopoietic system remains underexplored. Prior studies have demonstrated that Hfe is expressed in erythroid progenitors, influencing iron uptake and erythropoiesis. HFE has also been identified as a negative regulator of CD8+ T-cell activation and, in mice, its loss contributes to dysplastic hematopoiesis under oxidative stress conditions. Here, we investigated the role of Hfe/HFE in mouse and human hematopoietic stem and progenitor cell (HSPC) function. We demonstrate that loss of intrinsic hematopoietic Hfe/HFE leads to reduced numbers and function of HSPC, likely through enhanced cellular iron uptake and differentiation. Consistently, patients with HFE-HC had reduced peripheral blood clonogenic activity and changes in peripheral blood counts that may partially reflect ineffective hematopoiesis. Analysis of data from the UK Biobank revealed that women carrying p.Cys282Tyr variant in homozygosity are at increased risk of myelodysplastic syndrome (odds ratio, 3.50; 95% confidence interval, 1.64-7.49). Altogether, our data demonstrate that intrinsic Hfe/HFE affects HSPC function and supports future studies exploring its liver-independent role in hematopoiesis and hematologic malignancies.
    DOI:  https://doi.org/10.1016/j.brci.2026.100061
  26. Cancer Res. 2026 Jun 22.
      Single-cell RNA-sequencing (scRNA-seq) has revolutionized our understanding of cancer. However, identifying meaningful disease states from single-cell data remains challenging due to the complex continuum of transcriptional alterations, which may obscure clear phenotype boundaries and complicate biological interpretation and clinical relevance. Here, we systematically explored the chronic myeloid leukemia (CML) specific information content encoded in scRNA-seq versus bulk transcriptomics to resolve this paradox and clarify how discrete disease-defining states emerge from inherently noisy single-cell data. While CML single-cell transcriptomes existed along continuous transcriptional micro-states, clinically relevant leukemia phenotypes clearly manifested only at the pseudobulk (macro-state) level. State-transition theory was leveraged to reveal how disease phenotype state-transitions are governed by cell type specific contributions. Together, these results establish a theoretical framework explaining why discrete disease phenotypes remain hidden at the single-cell scale but emerge clearly at the aggregated macro-state level, enabling previously inaccessible biological insights into leukemia evolution. By resolving how single-cell variation aggregates into macroscopic disease states, this framework provides insights into CML progression and offers a broadly applicable strategy for exploring disease dynamics across cancers and other complex conditions.
    DOI:  https://doi.org/10.1158/0008-5472.CAN-25-4371