Genital tuberculosis is a major cause of infertility in countries where tuberculosis is endemic. With recent advances in assisted reproductive technology, women with tuberculous endometritis may become pregnant and ultimately have children with congenital tuberculosis. In vitro fertilisation represents a useful treatment for infertility. Congenital tuberculosis is a rare and severe, rapidly progressive, disease. Making an early diagnosis of congenital tuberculosis is difficult, because it can mimic many neonatal illnesses. To the best of our knowledge, we report the first cases of premature twins with congenital tuberculosis following in vitro fertilisation. After the diagnosis of genital tuberculosis in the mother, the twins were screened and diagnosed for tuberculosis. The twins dramatically improved after anti-tuberculosis therapy. We also wish to draw attention to the importance of searching for genital tuberculosis as the possible cause of congenital infection, even if the mother is asymptomatic.