The present study has demonstrated for the first time the involvement of CFTR inspermatogenesis and elucidated the possible underlying signaling pathway, providingsupport to the long proposed but intensively disputed link between CFTR mutationsand defects in sperm production, such as non-obstructive azoospermia andoligospermia [13], [18], [19], [20], [37], [38]. Here, CFTR is linked to Azoospermia.