INTRODUCTION: About 30-40% of the population report sexual dysfunction. Although it is well known that the brain controls sexual behavior, little is known about the neural basis of sexual dysfunction. AIM: To assess convergence of altered brain activity associated with sexual dysfunction across available functional imaging studies. METHODS: We used activation likelihood estimation meta-analysis to quantify interstudy concordance across 14 functional imaging studies reporting 179 foci from 40 individual analyses involving 191 subjects with sexual dysfunction and 123 controls. MAIN OUTCOME MEASURE: Activation likelihood estimation scores were used to assess convergence of findings. RESULTS: Consistently decreased brain activity associated with sexual dysfunction was identified in the dorsal anterior cingulate cortex, ventral striatum, dorsal midbrain, anterior midcingulate cortex, and lateral orbitofrontal cortex. CLINICAL IMPLICATION: These findings can serve as a basis for further studies on the pathophysiology of this highly common disorder with the view to development of more-specific treatment strategies. STRENGTH & LIMITATIONS: Findings are based on an observer-independent meta-analysis that provides robust evidence for and anatomic localization of altered brain activity related to sexual dysfunction. Our analysis cannot distinguish between the putative sources of sexual dysfunction, but it provides a more ubiquitous and general pattern of related altered neural activity. CONCLUSION: The identified regions have previously been shown to be critically involved in mediating sexual arousal and to be part of the sympathetic division of the autonomic nervous system. This suggests that the disturbance of brain activity associated with sexual dysfunction primarily affects sexual arousal already at early stages that are controlled by the sympathetic nervous system. Poeppl TB, Langguth B, Laird AR, et al. Meta-analytic Evidence for Neural Dysactivity Underlying Sexual Dysfunction. J Sex Med 2019;XX:XXX-XXX.