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Men with non-obstructive azoospermia (NOA) are candidates for microdissection testicular sperm extraction (microTESE). In men with NOA and varicocele, varicocelectomy has been associated with the appearance of sperm in the ejaculate. We report an infertile male with NOA, a solitary left testicle and a left clinical varicocele. The patient first underwent an extensive left microTESE. No spermatozoa were seen on initial microscopic evaluation but after an extensive search, spermatozoa were identified. ICSI did not result in pregnancy. Fifteen months after the microTESE, the patient underwent a left microsurgical varicocelectomy. A semen analysis post-varicocelectomy revealed the presence of sperm. Keywords: Varicocele, Non-obstructive azoospermia, MicroTESE