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Biology subjects

Bihler, J.

Publications and source records attributed to Bihler, J..

3 recordsLinked to original sources

A targetable dependency on nonsense-mediated decay for proteostasis and immune control in small cell lung cancer

Small cell lung cancer (SCLC) is among the deadliest cancers with excessive somatic alterations, expectedly resulting in immunogenic epitopes. However, patient benefit from immunotherapy is limited. We found abundant frameshift mutations in SCLC, regarded as highly immunogenic, counterbalanced by a hyperactive nonsense-mediated decay (NMD) pathway, responsible for frameshift-mRNA degradation. NMD activity correlated with tumor mutational burden (TMB) across cancers, suggesting that SCLC depends on NMD for cellular homeostasis and immune evasion. NMD inhibition impaired SCLC proliferation and induced ER stress-dependent apoptosis in a TMB-dependent manner, thereby enabling pharmacological inhibition in vivo which effectively controlled tumor growth. By integrating genome and transcriptome sequencing with MHC-I immunopeptidomics and functional in vitro and in vivo assays, we identified that NMD inhibition boosted neoantigen expression and presentation by tumor cells and increased T cell recognition, thus enhancing overall tumor immunogenicity and further improving immunotherapy efficacy in vivo. Our work shows that SCLC - as a TMBhigh cancer - relies on NMD for survival and immune escape, uncovering a novel TMB-dependent tractable vulnerability for this devastating disease.

cancer biology↗

Establishment and Characterization of a CCND1-Rearranged Non-Mantle Cell Lymphoma Cell Line and Patient-Derived Xenograft Model

The pathobiology of aggressive B-cell lymphomas with CCND1 rearrangements, distinct from Mantle Cell Lymphoma (MCL), presents a significant clinical challenge. These lymphomas are often difficult to diagnose and demonstrate resistance to standard immunochemotherapy, underscoring the urgent need for a deeper understanding of their underlying biology to develop more effective treatments. A major impediment to progress has been the lack of robust preclinical models that accurately reflect the complex genomics and clinical behavior of this disease. Here we directly address this critical gap by reporting the establishment and in-depth characterization of the first patient-derived cell line and a corresponding systemic patient-derived xenograft (PDX) model of a CCND1-rearranged, non-MCL lymphoma with a rapidly fatal clinical course. Through a comprehensive multi-omics approach, we demonstrate that these novel in vitro and in vivo models faithfully recapitulate the primary tumors unique immunophenotype, its intricate genetic and transcriptional landscape, and its intrinsic resistance to conventional therapeutic agents.

cancer biology↗

SLX4IP acts in parallel to FANCM to limit BLM-dependent replication stress at ALT telomeres

Alternative Lengthening of Telomeres (ALT) is a telomerase-independent telomere maintenance mechanism that enables cancer cells to gain unlimited replicative capacity. ALT relies on recombination-mediated telomere elongation and is promoted by telomeric replication stress. However, ALT requires strict regulation, as excessive replication stress or recombination are cytotoxic. Central to ALT is the RecQ helicase BLM, which regulates telomeric replication stress and promotes telomere recombination and DNA synthesis. Despite its key role in the ALT pathway, BLM must be tightly regulated to prevent deleterious outcomes. Here, we identify SLX4IP as a key suppressor of BLM-driven replication stress at ALT telomeres. Loss of SLX4IP in ALT-positive cells leads to BLM-dependent telomeric replication stress and impaired replication fork progression. Mechanistically, SLX4IP limits the unwinding of unligated Okazaki fragments by BLM on the lagging strand during telomere replication. This reduces the formation of toxic 5' DNA flaps and prevents hyperactivation of ATR signalling and deleterious recombination levels. We also uncover a synthetic lethal interaction between SLX4IP and FANCM, an ATPase/translocase that is a known regulator of BLM at telomeric replication forks in ALT cells. We demonstrate that SLX4IP and FANCM act in parallel to restrain BLM activity, thereby maintaining the balance of replication stress and recombination that is necessary for productive ALT. These findings reveal a vulnerability in ALT-positive cancers lacking SLX4IP and establish SLX4IP as a potential biomarker for therapeutic strategies targeting FANCM. HIGHLIGHTSO_LISLX4IP depletion activates the replication stress response at ALT telomeres C_LIO_LISLX4IP acts in parallel to FANCM to limit replication stress at ALT telomeres C_LIO_LIThe synthetic lethal interaction between SLX4IP and FANCM is dependent on BLM C_LIO_LISLX4IP depletion causes BLM-dependent lagging-strand replication stress C_LI

cell biology↗