: Extreme dipping (i.e. a marked blood pressure fall during night-time period) is an alteration of circadian blood pressure (BP) rhythm frequently observed in the setting of systemic hypertension as well as in the general population. Some reports have suggested that cardiovascular prognosis in extreme dippers (ED) is similar as in dippers, whereas other studies have documented either a better or worse prognosis in ED. Available information on clinical and prognostic implications of ED is scanty and data provided by studies are controversial. Furthermore, a comprehensive report summarizing the key features of this BP pattern is lacking. The present review focuses on a number of issues concerning ED pattern such as the prevalence and clinical correlates, mechanisms underlying this BP phenotype association with hypertension mediated organ damage (HMOD) and prognostic value in predicting cardiovascular events and all-cause mortality. The reported prevalence of this BP rhythm alteration ranges from 5% to 30%, depending on diagnostic criteria, clinical and demographic characteristics of subjects. Most studies targeting the association of this condition with HMOD failed to find consistent findings in support of an adverse impact of ED on vascular, renal of cardiac structure and function. Available data on ED as compared to low risk reference group (i.e. dippers) do not allow to conclude that high BP variability resulting from a marked BP fall at night adversely affects cardiovascular prognosis at the community level and in the general hypertensive population. Thus, further studies aimed to assess the prognostic significance of ED as well as the impact of therapeutic interventions aimed to normalize this circadian BP pattern, are highly needed.