Aortic cusp prolapse is an acquired complication and usually precedes the development of aortic regurgitation (AR) in unoperated outflow ventricular septal defect (VSD). However, its impact on postoperative AR-progression is unknown. 161 patients with outflow-VSD and AR who underwent surgery between 2006 and 2012 were studied retrospectively. 31 patients without prolapse (group-I), 87 with only right coronary cusp (RCC) (group-II), 43 with noncoronary cusp (NCC) prolapse (group-III: 23 only NCC (IIIa), 20 both NCC-RCC (IIIb)) were followed postoperatively for a mean 6.05+/-2.4 years (range 3-12 years). Moderate or severe-AR was present in 4.2%, 36.8%, 52.2% and 80% preoperatively; in 3.2%, 10.3%, 39.1% and 30% patients at follow-up in group-I, II, IIIa, and IIIb, respectively. Although freedom from significant-AR (moderate or severe AR) or aortic valve replacement (AVR) at 10 years was lesser in subaortic-VSD than subpulmonic-VSD (64.3+/-7.5% vs 87.9+/-3.6 %; p =0.02), the difference was not significant when compared within prolapse groups (80+/-8% vs 88.7+/-4.0%, p=0.28 in group-II; 40.7+/-11.8 vs 70+/-14.5%, p=0.48 in group-III). The significant-AR or AVR free survival in patients with trivial or mild preoperative-AR was not significantly different between prolapse groups (98.2+/-1.8% vs 75+/-21.7% in group-II and III respectively; p=0.85). However, in those with moderate or severe preoperative-AR it was significantly lesser in group-III than II (30.1+/-9.8% vs 65.6+/-8.4%, respectively; p=0.04). Group-III, compare to group-II, had 3.28 and 5.24-time risk of development of significant-AR or requirement of AVR, respectively. Prolapse of NCC alone or in addition to RCC prolapse has unfavourable impact on the postoperative outcomes, especially in subaortic-VSD after development of more than mild AR preoperatively.