Background: The anterior and lateral position of the anterolateral papillary muscle (ALPM) has found to be reached with better catheter stability and less mechanical bumping via the transseptal (TS) compared to the retrograde aortic (RA) approach. Aim: To compare the TS and RA approaches on mapping and ablation of ventricular arrhythmias (VAs) arising from ALPMs. Methods: Thirty-two patients with ALPM-VAs undergoing mapping and ablation via the TS approach were included and compared with 31 patients via the RA approach within the same period. Acute success was compared, as well as other outcomes including misinterpreted mapping results due to bumping, radiofrequency (RF) attempts, procedural time and success rate at 12 months’ follow-up. Results: Acute success was achieved in more cases in the TS group (96.4% vs 72.0%, P=0.020). During activation mapping, bump-provoked premature ventricular complexes (PVCs) misinterpreted as clinical PVCs were more common in the RA group (30.0% vs 58.3%, P=0.036), leading to more RF attempts (3.5±2.7 vs 7.2±6.8, P=0.006). Suppression of VAs were finally achieved in the unsuccessful cases by changing to the alternative approach, but the procedural time was significantly less in the TS group (90.0±33.0 vs 113.7±41.1min, P=0.027) with less need to change the approach, although follow-up success rates were similar (75.0% vs 71.0%, P=0.718). Conclusion: A TS rather than RA approach as the initial approach appears to facilitate mapping and ablation of ALPM-VAs, specifically by decreasing the possibility of misleading mapping results caused by bump-provoked PVC, and increase the acute success rate thereby.