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Most dairy cows throughout the world are milked twice daily. In intensive dairying systems, however, it is not uncommon to increase milking frequency to between 3 and 6 times daily to increase milk production. Reducing milking frequency is much less common; however, once-daily milking of dairy cows, practiced either strategically during certain parts of the lactation or for the entire lactation, is not uncommon in key dairying countries where less emphasis is placed on milk production per cow. The practice fits well with more extensive dairy production systems, particularly those based on grazed pasture. A feature of once-daily milking is that it reduces milk yield by approximately 22%, depending on stage of lactation, breed, and parity, and it may adversely affect lactation length and persistency. However, it can offer several positive farm management options, especially related to labor requirements and farm working expenses. In addition, it may provide a tool to better manage the metabolism and energy balance of cows during early lactation or during periods of pasture deficit, and it may help to improve reproductive performance and animal health and welfare. Once-daily milking, representing one extreme of the mammary function spectrum, has attracted considerable research interest over the years. Consequently, substantial scientific information is available on its effects on mammary function, at both the physiological and molecular levels. This review focuses instead on the management of the cow milked once daily, covering the production response in relation to breed, stage of lactation, and parity, and its effect on energy status, reproduction, health and welfare, as well as on milk composition and processability.