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Ceralasertib

AZD6738

ATR kinase inhibitor

Evidence Score

33

preclinical
Mechanism of Action

Selective, orally bioavailable inhibitor of ATR (ataxia telangiectasia and Rad3-related) serine/threonine kinase. The mechanistic rationale is synthetic lethality with ATRX loss: in SDHB-driven metastatic pheochromocytoma and paraganglioma, ATRX co-mutations occur in ~30–40% of cases (confirmed by multi-omics analysis, PMID 42230482) and are the strongest genomic predictor of malignancy in this subtype. ATRX loss triggers the Alternative Lengthening of Telomeres (ALT) pathway, creating constitutive telomeric replication stress — G-quadruplex accumulation, R-loops, fragile telomeres — that generates an absolute dependency on ATR kinase for stalled-fork resolution and survival. Flynn et al. (Science 2015, PMID 25614623) established that ATRX-loss/ALT-positive cells are 10–30× more sensitive to ATR inhibition than ALT-negative controls across multiple cancer types, a synthetic lethality that is absent from ATRX-wild-type cells. Ceralasertib is in Phase 1/2 clinical development; the OLAPCO Phase 2 trial (NCT03787680) tests ceralasertib + olaparib in DNA-damage-response-deficient tumors including ATRX-loss contexts. Key limitation: no dedicated PPGL or SDH-deficient-specific trial data exists; the ~30–40% ATRX co-mutation prevalence means this strategy applies only to the ATRX-loss subgroup, requiring prospective ATRX genotyping to identify eligible patients.

Pathway Connections
ATRX Loss / ALT Replication Stress

In SDHB-driven metastatic pheochromocytoma and paraganglioma, ATRX co-mutations occur in ~30–40% of cases and are among the strongest genomic predictors of malignancy (confirmed by multi-omics profiling: PMID 42230482). ATRX loss activates the Alternative Lengthening of Telomeres (ALT) pathway — a recombination-based telomere maintenance mechanism — which creates constitutive replication stress at telomeric sequences through G-quadruplex DNA accumulation, R-loop formation, and fragile telomeres. ALT-positive cells are rendered hypersensitive to ATR kinase inhibition: Flynn et al. (Science 2015, PMID 25614623) demonstrated that ATRX-loss/ALT-positive cancer cells are 10–30× more sensitive to ATR inhibitors than ALT-negative cells across multiple cancer types, establishing a synthetic lethality that is absent in ATRX-wild-type tumors.

Upstream event:

SDH loss (particularly SDHB mutation) → epigenetic instability → ATRX co-mutation → ALT pathway activation → constitutive telomeric replication stress → ATR dependency

Downstream effects:

G-quadruplex DNA accumulation at telomeresR-loop formation and replication fork stallingConstitutive ATR kinase activation at stalled forksSynthetic lethality with ATR inhibition (10–30× sensitization vs. ALT-negative cells)C-circles as an extrachromosomal DNA biomarker of ALT activityHigh metastatic potential in SDHB-driven PPGL
Molecular Targets

ATR

Ataxia telangiectasia and Rad3-related protein kinase

synthetic_lethal

The primary replication stress checkpoint kinase. Phosphorylates and activates CHK1, stabilizes stalled replication forks, and coordinates origin firing suppression under genotoxic stress. In ATRX-loss/ALT cells, constitutive telomeric G-quadruplex formation and R-loop accumulation generate persistent stalled replication forks that require ATR signaling for resolution; ALT cells are therefore dependent on ATR for survival. Flynn et al. (Science 2015, PMID 25614623) established that ATR inhibition is synthetically lethal with ALT in multiple cancer types with ATRX or DAXX loss, showing 10–30× greater sensitivity in ALT-positive versus ALT-negative cells.

UniProt: Q13535

Quick Facts
Not FDA Approved
Evidence

Evidence from PubMed, OpenTargets, and ChEMBL will appear here once external data integration is enabled.

Coming in Phase 3

For research exploration only — not medical advice. Consult your doctor before acting on any information.

AI Analysis

Have Claude analyze this drug's repurposing potential for SDH-deficient diseases.