The prevalence of septate/subseptate and arcuate uterine anomaly in infertile patients varies in the literature due to different modes of diagnoses and methodological bias. Our group previously reported that mid fundal length on transvaginal 3D US (TV 3D US) with or without saline infusion sonohysterography (SIH) tends to be underestimated in patients with subtle incomplete septum and arcuate uterine anomaly (Abuzeid O. et al. Fertil Steril 2015). The aim of this study is to determine the prevalence of significant subseptate and arcuate uterine anomaly on hysteroscopy and TV 3D US in infertile patients with reproductive failure. All patients who presented to our unit with a history of infertility (2008 – 2016) were studied. Optimal testing for the diagnosis of arcuate and septate/subseptate uterus (ASRM Class Vb and VI) was used. The criterion for optimal testing was TV 3D US with or without SIH, and hysteroscopy with or without laparoscopy. Five hundred fifty six patients who fulfilled inclusion criteria were included in this study. Inclusion criteria included the diagnosis of subseptate or arcuate uterus on hysteroscopy and TV 3D US with or without SIH with measurement of mid fundal protrusion length. For this study we used strict criteria for the diagnoses of the above-mentioned anomalies on hysteroscopy. Such criteria would be a mid fundal protrusion length of ≥15 mm. Such measurement was done at time of diagnostic hysteroscopy using straight resectoscope loop. The TV 3D US definition of such anomalies was a mid fundal protrusion length ≥10 mm. Mean age (years) was 32.6 ± 5.2, mean duration of infertility (years) was 2.9 ± 2.5, mean BMI (kg/m2) was 27.3 ± 6.8, mean FSH level (mIU/ml) was 7.1 ± 2.9 and primary infertility was present in 68.7%. Of the 556 patients included in this analysis, 83 patients had significant subseptate uterus and 161 patients had significant arcuate uterine anomaly on hysteroscopy. Fourteen patients had subseptate uterus and 12 patients had arcuate uterine anomaly on TV 3D US. The prevalence of significant subseptate uterus (14.9%) and arcuate uterine anomaly (29.0%) on hysteroscopy was significantly higher than the prevalence subseptate uterus (2.5%) and arcuate uterine anomaly (2.2%) on TV 3D US (p = 0.000). Our data suggest that TV 3D US tends to underestimate the prevalence of significant subseptate uterus and arcuate uterine anomaly compared to hysteroscopy. Prevalence of such anomalies in infertility patients is much higher than what is reported in the literature. Diagnostic hysteroscopy is the gold standard for diagnosis of such anomalies.