Professor Samia Temtamy is a great Egyptian icon of human genetics and one of the first three most eminent international human genetics scientists who obtained a PhD degree in the aforementioned field. Prof. Temtamy had successfully laid the foundation for the science of human genetics and genomics at the National Research Centre shortly after the discovery of the correct number of human chromosomes and all the way to the human genome era. The story began more than 50 years ago with the establishment of a small unit at the National Research Centre in Cairo, Egypt. The unit started as a clinic that accepted patients with various inherited abnormalities. Later, that small unit became the Human Genetics Department in 1977. Prof. Temtamy had made a great effort to continue improving the services provided to patients with genetic disorders and the research facilities by converting the Human Genetics Department into the Division of Human Genetics and Genome Research that included several specialized departments in 2003. The Center of Excellence for Human Genetics was then established in 2014, which is considered the largest institute of human genetics in Egypt. This review summarizes the long journey taken and the great efforts put in by Prof. Temtamy and her team at the National Research Centre in the field of human genetics in Egypt, to keep pace with the continuous rapid advancement of human genetics and genomics and contribute to the international scientific community.