OBJECTIVES: Target joints (TJs) are a common complication of severe haemophilia. While factor replacement therapy constitutes the majority of costs in haemophilia, the relationship between TJs and non-drug-related direct costs (NDDCs) has not been studied. METHODS: Data were drawn from the ‘Cost of Haemophilia across Europe – a Socioeconomic Survey’ (CHESS) study, a cost assessment in severe haemophilia A and B across five European countries (France, Germany, Italy, Spain, and the UK) in which 139 haemophilia specialists provided demographic and clinical information for 1,285 adult patients. NDDCs were calculated using publically available cost data, including 12-month ambulatory and secondary care activity: haematologist and other specialist consultant consultations, medical tests and examinations, bleed-related hospital admissions, and payments to professional care providers. A generalised linear model was developed to investigate the relationship between NDDCs and TJs (defined in the CHESS study as areas of chronic synovitis), adjusted for patient covariates including socio-demographic characteristics and comorbidities. Patients with a diagnosed inhibitor at the time of reporting (n=58) were excluded from the analysis. RESULTS: Five hundred and thirteen patients (42% of the sample) had no diagnosed TJs; a total of 1,376 TJs were recorded in the remaining 714 patients. Mean adjusted NDDCs for persons with no TJs were EUR 3,134 (standard error (SE) EUR 158); for persons with one or more TJs, mean adjusted NDDCs were EUR 3,913 (SE EUR 157; average marginal difference EUR 779; p<0.001). CONCLUSIONS: Our analysis suggests that the presence of one or more TJs has a significant impact on NDDCs for patients with severe haemophilia. Prevention and management of TJs should be an important consideration of managing haemophilia patients in the current cost constrained healthcare environment.