OBJECTIVES: Although acromegaly is acknowledged as requiring resource-intensive treatment, its ultimate economic burden is unclear. As an extension of work presented at ISPOR 2013 International Conference (New Orleans, US), the objective of this research is to measure the annual economic burden of acromegaly in France using a case-review methodology with a prevalence-based sample of patients diagnosed with acromegaly. METHODS: A case-review method was used with a sample of 22 endocrinologists reviewing 88 patient cases (4 cases per physician) diagnosed with acromegaly.  The patient case histories included: resource utilization including office visits and hospitalization, diagnostic procedures and labs, medications prescribed, medical procedures preformed, and an estimate of lost productivity.  A micro-costing analysis was conducted to obtain costs in the prior 12 months for each patient case reviewed using published literature, medical fee schedules, and pharmaceutical cost databases to assign costs to treatments and medical procedures identified in the survey data.   Annual costs were examined across a broad range of patients of different ages, gender and time from diagnosis.  Two biomarkers were used to categorize acromegaly patients as Controlled vs. Uncontrolled: Insulin Growth Factor-1 (IGF-1) and Growth Hormone (GH).  Several patient characteristics were used as control factors when comparing annual economic costs: age, sex, and time from diagnosis.  Statistical tests and confidence intervals were calculated to determine the significance of patient characteristic effects on economic burden. RESULTS:  Three patient subgroups were used to classify uncontrolled acromegaly patients: IGF-1, GH and both IGF-1 and GH.  The annual per-patient economic burden of disease costs ranges from € 29,000 to € 79,400 across these groups.  These cost ranges are benchmarked to other studies to provide context and validity.  CONCLUSIONS:  The total economic burden of acromegaly in France is significant. Understanding the factors impacting burden of illness will inform future improvements in treatment practice.