This case reports a delivery of a child with limb and renal malformations following intracytoplasmic sperm injection. A 29-year-old woman and a 39-year-old man with primary infertility underwent IVF-ICSI-ET therapy cycle. The couple delivered an infant with acrorenal syndrome. When assisted reproductive techniques are used, especially ICSI, the risk of genetics abnormalities should be discussed in detail with the couple and sufficient informed consent should be obtained before starting IVF-ET procedures.