Background: Advanced paternal age associated comorbidities and reduced fertility possess significant risks of genetic disorders to the offspring. With a broad implementation of a non-invasive prenatal testing (NIPT) and invasive assessment leads to more cases of genetic disorders, including sex discordance are revealed. Among biological causes of sex discordance are disorders of sexual development. The main aim of present case study is prenatal diagnosis of sex differentiation disorders with focus on the role of ultrasound scans for assessment of prenatal diagnosis. Case Presentation: In this report 37-year-old pregnant woman with 22-23 weeks gestation refer to center expert opinion regarding perineal abnormalities. On sonography at our centre we notice positive findings are as: micropenis with bifid scrotum or it could be ambiguous genitalies, which falls in group of disorders of sexual differentiation. As Karyotyping was primarily assess by local doctors by amniocentesis which was XY. Conclusion: Genotype-phenotype correlation might improve prenatal diagnosis of Fetuses who are having DSD (disorder of sex differentiation). Combining ultrasonic examination with genetic analysis will definitely improve accurate prenatal diagnosis.