J Spinal Cord Med. 2009; 32(2): 191-7Singh PK, Singh VK, Azam A, Gupta SBACKGROUND/OBJECTIVE: Tarlov cysts or spinal perineurial cysts are uncommon lesions. These are mostly incidental findings on magnetic resonance imaging or myelograms. The objectives of this study were to describe Tarlov cysts of the sacral region as a potential cause for retrograde ejaculations and review available management options. METHODS: Case report and literature review. RESULTS: A 28-year-old man presented with back pain and retrograde ejaculations resulting in infertility. After microsurgical excision of large perineurial cysts, back pain resolved, but semen quality showed only marginal improvement. Later, the couple successfully conceived by intrauterine insemination. To the best of our knowledge, this is the first reported case of Tarlov cyst associated with retrograde ejaculation and infertility. CONCLUSIONS: Despite being mostly asymptomatic and an incidental finding, Tarlov cyst is an important clinical entity because of its tendency to increase in size with time. Tarlov cysts of the sacral and cauda equina region may be a rare underlying cause in otherwise unexplained retrograde ejaculations and infertility. Microsurgical excision may be a good option in a select group of patients.