- Introduction: Kartagener's syndrome (KS) is characterized by the classic triad of chronic sinusitis, bronchiectasis and situs inversus. This is caused by dynein arm defects in axonemal microtubules in ciliary and flagellar structures. Male patients are invariably infertile because of the immotility of spermatozoa. We report a healthy birth that was achieved by intracytoplasmic sperm injection (ICSI) using ejaculated spermatozoa from a patient with KS. Case report: The wife of the patient was a 33-year-old woman with infertility of one-year duration.Her diagnostic infertility work-ups were normal. Her husband was 33 years old. He had normal secondary sex characteristics and both testicular volumes were normal. His serum FSH, LH, testosterone and prolactin levels were all within normal limits. He had dextrocardia and dysosmia as well as a history of chronic bronchitis and sinusitis, the classic triad disorders of KS, and was therefore diagnosed with KS. Several semen analyses showed sperm concentration of 77-87 × 10 6 /ml and ejaculated volume of 3.5-4.0 ml. However, only 2-28 motile spermatozoa were observed in the whole field with a Makler Counting Chamber. After eosin staining, 30% of the sperm were viable with unremarkable morphology. Transmission electron microscopy (TEM) of the sperm tail showed the absence of both inner and outer dynein arms. After counseling for this condition and suitability for ICSI, the couple proceeded with ICSI using ejaculated spermatozoa. In August 2005, after ovarian stimulation by a long protocol using GnRHa / hMG, twenty-one oocytes were obtained, and 18 metaphase II oocytes were retrieved. Fresh semen samples were obtained after masturbation. The sperm concentration was 57.2 × 10 6 /ml and the volume was 3.5 ml. Total motility was 0.3%, and no spermatozoa showed straight progressive motility. After swim-up preparation, we observed a few motile spermatozoa that appeared morphologically normal, and these motile spermatozoa were selected for ICSI. Thirteen oocytes were fertilized. All embryos were cryopreserved in the cleavage stage to prevent development of ovarian hyperstimulation syndrome. In October 2005, two--step embryo transfer was performed in the hormone replacement (HR) cycle. A twin pregnancy was achieved, one resulting in missed abortion but the other being uneventful. A healthy female infant was delivered in June 2006 at a gestation age of 37 weeks and weighing 2675 g. In April 2008, the couple was referred to our clinic for a second pregnancy. Four cycles of single blastocyst transfer using a cryopreserved-thawed blastocyst were repeatedly performed. Pregnancy tests were positive in three cycles, but all of the three pregnancies resulted in chemical abortion. In January 2009, after using an ovarian stimulation protocol similar to that used in the first cycle, five metaphase II oocytes were retrieved. Sperm concentration and total motility were 68.1 × 10 6 /ml and 0.1%, respectively, and a few progressive motile sperm were observed. The intracytoplasmic morphologically selected sperm injection (IMSI) procedure was performed with the progressive motile sperm. All oocytes were fertilized. One embryo was cryopreserved in the cleavage stage and the other four embryos were cultured for three more days, but no blastocyst was obtained. In March 2009, cryopreserved-thawed embryo transfer was performed in the HR cycle, but pregnancy was not achieved. Conclusion: This case report demonstrates that successful pregnancy after ICSI in couples with Kartagener's syndrome is possible with ejaculated spermatozoa. Our results suggest that even for KS patients, if motile sperm are observed, a fertilization rate comparable to that with ICSI of the other male factor can be expected. KS is a heterogeneous group of disorders with similar clinical presentations, and treatment should be individualized depending on sperm motility.