Since 1997, there has been increasing research focusing on muscle dysmorphia, a condition underpinned by people's beliefs that they have insufficient muscularity, in both the Western and non-Western medical and scientific communities. Much of this empirical interest has surveyed nonclinical samples, and there is limited understanding of people with the condition beyond knowledge about their characteristics. Much of the existing knowledge about people with the condition is unsurprising and inherent in the definition of the disorder, such as dissatisfaction with muscularity and adherence to muscle-building activities. Only recently have investigators started to explore questions beyond these limited tautological findings that may give rise to substantial knowledge advances, such as the examination of masculine and feminine norms. There is limited understanding of additional topics such as etiology, prevalence, nosology, prognosis, and treatment. Further, the evidence is largely based on a small number of unstandardized case reports and descriptive studies (involving small samples), which are largely confined to Western (North American, British, and Australian) males. Although much research has been undertaken since the term "muscle dysmorphia" entered the psychiatric lexicon in 1997, there remains tremendous scope for knowledge advancement. A primary task in the short term is for investigators to examine the extent to which the condition exists among well-defined populations to help determine the justification for research funding relative to other public health issues. A greater variety of research questions and designs may contribute to a broader and more robust knowledge base than currently exists. Future work will help clinicians assist a group of people whose quality of life and health are placed at risk by their muscular preoccupation.