<p>Background: Repeated <em>in vitro</em> Fertilization (IVF) failure together with Ovarian Hyperstimulation Syndrome (OHSS) is one of the distressing situations leading couples to search for alternative treatment options. For such patients with Polycystic Ovarian Syndrome (PCOS) who have experienced Ovarian Hyperstimulation Syndrome, mild ovarian stimulation with <em>in vitro</em> oocyte maturation could be a promising alternative. Testicular Sperm Aspiration (TESA) of spermatozoa from a known obstructive azoospermic patient is a limiting factor for IVM (<em>in vitro</em> maturation) but the couple reported here accepted mild IVF -IVM with TESA.<br /> Case Presentation: In the treatment of a 32-year old woman, 9 immature oocytes were retrieved, 5 <em>in vitro</em> matured oocytes (maturation rate 55%) and after fertilization by Intracytoplasmic Sperm Injection (ICSI), 3 oocytes (fertilization rate 60%) which had two pronuclei and two good quality embryos on day 2, were transferred. A live pregnancy was observed by ultrasound scan and healthy infant was delivered.<br /> Conclusion: Although the number of births from ICSI of immature oocytes injected with surgically derived sperm is quite low, the selection of this mode of therapy as an alternative to conventional IVF will overcome the limitations and provides a new option in IVF practice.</p>