The coronal plane lower limb alignment plays an important role in the occurrence and progression in knee osteoarthritis. There have been reports of the valgus knee in patients with unilateral developmental hip dislocation (UDHD) with the relatively small sample size. Besides, few studies have analyzed the lower limb alignment of the contralateral side. The purpose of our study was to identify the coronal plane alignment of both the ipsilateral and the contralateral lower limb in patients with UDHD and find out the difference between patients with Hartofilakidis type II and III. The radiographic data of all UDHD patients who met the inclusion criteria from March 2011 to February 2017 were retrospectively reviewed, including the hip-knee-ankle angle (HKA), mechanical lateral distal femoral angle (mLDFA), anatomical lateral distal femoral angle (aLDFA), mechanical proximal tibial angle (MPTA) and the lateral distal tibial angle (LDTA). Besides, the femoral torsion angle was measured on the images of CT scan. The average HKA was 3.42°(range: −4.3–12.8°) on the affected side, and −2.11°(range: −11.4–5.4°) on the contralateral side (P?0.0001). The valgus lower limb alignment on ipsilateral side was most frequently seen in both Hartofilakidis type II (20cases, 51.3%) and type III groups (25cases, 67.6%), whereas for the contralateral side, the neutral alignment in type II group (27 cases, 69.2%) and varus alignment in type III group (19 cases, 51.4%) were most commonly observed. Both the mLDFA (P?0.001) and aLDFA (P?0.001) of ipsilateral side were significantly smaller than those of contralateral side. The average femoral torsion angle was 37.9°(range: 10.4–64.4°) on the affected side, and 27.1°(range: 9.7–45.5°) on the contralateral side (P?0.001). In conclusion, UDHD patients may present with lower limb malalignment on both sides. The valgus lower limb alignment is the most common deformity on ipsilateral side, which is caused by increased femoral torsion angle as well as the decreased aLDFA. The patients with Hartofilakidis type III UDHD may be more prone to present varus alignment deformity than those with Hartofilakidis type II on the contralateral side. The lower limb malalignment and deformity of ipsilateral distal femur should be considered during any surgery involving hip, knee or femur