This case report describes the first experience of laparoscopic transmesenterial resection of the lower segment of horseshoe kidney due to renal cell carcinoma. All stages were performed using the “window” in the mesenterium of the colon. The size of the “window” provided optimal ergonomics at all stages of the intervention. We consider that during isthmus dissection transmesenterial access provides more freedom compared to conventional techniques. Quick access to the isthmus and to the abdominal aorta through the minimal volume of tissues allows to perform the precision dissection, lymphadenectomy and isthmotomy. The latter was done without ligation of isthmus arteria and suturing devices, with only intracorporeal ligation with capron and transection using monopolar hook. Peculiarities of the collecting system of the horseshoe kidney allowed the preparation of the calyces and segmentary vessels in the sinus. Therefore, lower major calyces, intended to be removed with the tumor, were prepared before the resection of lower segment of left half. This maneuver did not require the warm ischemia, and Hem-o-lok clips let us secure the collecting system without intracorporeal suture of the pelvis. Warm ischemia time was 19 minutes, operation duration – 210 minutes. Intraand postoperative complications were not evident. The patient stayed 5 days in the hospital after the intervention. This case report demonstrates the possibility of non-standard access and non-standard application of the standard instruments, which permitted the laparoscopic segmentary resection of the kidney, given the peculiarities of anatomy.