Exceptional response to the ATR inhibitor, camonsertib, in a patient with ALT+ metastatic melanoma.

Publication date: Jul 07, 2025

A key hallmark of cancer tumorigenesis is the maintenance of telomere length, which occurs canonically through the reactivation of telomerase. Alternative lengthening of telomeres (ALT) is an atypical, non-canonical telomere maintenance mechanism that uses homologous recombination (HR) to maintain telomere length and is associated with replication stress and defects in genome maintenance. In preclinical models, ALT positivity (ALT+) sensitizes tumor cells to ataxia telangiectasia and Rad3-related (ATR) inhibitors. Camonsertib is a novel potent, and highly selective ATR inhibitor that is synthetic lethal with genomic alterations affecting HR and DNA damage response (DDR). Here we describe a case of confirmed clinical and molecular response to pharmacological ATR inhibition through camonsertib, in a patient with ALT+ metastatic melanoma. To our knowledge, this is the first clinical report of synthetic lethal targeting of a confirmed ALT+ tumor with an ATR inhibitor.

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Concepts Keywords
Genomic Alt
Lethal Alt+
Pharmacological Atr
Recombination Camonsertib
Tumorigenesis Confirmed
Inhibitor
Length
Lethal
Maintenance
Melanoma
Metastatic
Patient
Synthetic
Telomere
Tumor

Semantics

Type Source Name
disease MESH melanoma
pathway KEGG Melanoma
disease MESH cancer
disease MESH tumorigenesis
pathway REACTOME Telomere Maintenance
pathway KEGG Homologous recombination
disease MESH defects
disease MESH ataxia telangiectasia
drug DRUGBANK Honey
drug DRUGBANK Nivolumab
drug DRUGBANK Ipilimumab
drug DRUGBANK Olaparib
drug DRUGBANK Trametinib
pathway REACTOME Methylation
drug DRUGBANK Coenzyme M
disease MESH metastasis
disease MESH anemia
disease MESH leukemia
disease MESH noma
drug DRUGBANK Formaldehyde
drug DRUGBANK Methionine
drug DRUGBANK Ethanol
drug DRUGBANK Sodium Chloride
drug DRUGBANK Edetic Acid
drug DRUGBANK Phosphate ion
drug DRUGBANK Medical air
drug DRUGBANK Sodium Citrate
drug DRUGBANK Tromethamine
drug DRUGBANK Gold
drug DRUGBANK Water
disease MESH microsatellite instability
disease MESH acinar carcinoma
pathway REACTOME Reproduction

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