Background:We previously reported that interstitial injection of bleomycin (BLM) reduces the size of early-stage extracranial arteriovenous malformation (AVM).Here,we sought to investigate the potential mechanism of BLM in treating extracranial AVM.Methods:Samples of human extracranial AVM (n=3) with no pharmacological treatment were harvested.AVM endothelial cells were isolated and cultured in primary cell culture.The transcriptome was examined using RNA-sequencing,and differentially expressed C-type lectin domain family 14 member A (CLEC14A) was validated at the transcriptomic and protein levels.Immunocytochemical staining of CLEC14A was performed in samples of human extracranial AVM,with and without BLM treatment.Results:Through second-generation sequencing,we found that the expression of 5689 genes were differentially increased or decreased following 24-h BLM stimulation.We found that CLEC14A may play an important role in the progression of AVM and can be inhibited by BLM treatment.Conclusion:BLM inhibited CLEC14A expression to attenuate the progression of AVM.