Facioscapulohumeral muscular dystrophy (FSHD) is an autosomal dominant inherited condition that causes lifelong progressive muscle weakness. Despite the significant disease burden, little has been published on the costs associated with a diagnosis of FSHD to patients and their families. While cost of illness studies of facioscapulohumeral muscular dystrophy (FSHD) have been performed in Europe, no comprehensive studies have been published in the United States. To our knowledge, efforts that are underway in the United States often only look at insurance claims data. However, these studies underestimate the full economic impact of FSHD because they do not account for out-of-pocket expenses, such as home modifications and special transportation needs, and other indirect costs, such as absenteeism and lost opportunities. To better understand the full economic impact of FSHD over the course of a lifetime in the United States, we conducted a survey of the community to understand the cost burden carried by people with FSHD and their families. We report here results from the survey, representing 354 FSHD individuals residing in 312 households across the United States. Preliminary analysis of survey data reveals average estimated annual out-of-pocket per-patient costs to be >$9,000 for direct medical costs and >$19,000 for direct non-medical costs, totaling >$28,000 per year. We also discuss indirect costs, such as family planning services, lost income, and absenteeism. An accompanying study examined the medical claims costs for individuals with FSHD in the United States.