Objective <p>The one-step percutaneous transhepatic cholangioscopic lithotripsy (PTCSL) technology for the treatment of hepatolithiasis is still considered challenging. The aim of the present study was to evaluate the influence of three-dimensional (3D) visualization on one-step PTCSL , Perioperative and long-term clinical data were profoundly analyzed during the treatment of intrahepatic bile duct stones.&#xa0;</p> Methods <p>We reviewed data on 231 hepatolithiasis patients receiving surgical treatment in our department from January 2015 to January 2019, including 139 patients in group A treated with one-step PTCSL assisted by 3D visualization and 92 patients in group B treated with one-step PTCSL alone.</p> Results <p>The short-term outcomes including hospital stay, intraoperative blood loss, immediate stone removal rate, and the long-term outcomes including postoperative complications and recurrence-free survival (RFS) were compared. The results showed that one-step PTCSL assisted by 3D visualization slightly reduced Intraoperative blood Loss (21.14 ± 17.91 ml vs. 21.47 ± 25.91 ml; P = 0.000) and hospital stay (12.21 ± 6.14 days vs. 18.77 ± 8.73 days; P = 0.000). Moreover, there are no significant differences in hospitalization expenses, stone final clearance rate and postoperative complications between the two groups.</p> Conclusion <p>The 3D visualization can make one-step PTCSL a safe, effective, and promising treatment for patients with complicated primary hepatolithiasis. The perioperative and long-term outcomes are satisfactory for complicated primary hepatolithiasis.</p>