Objective: The consequences of patient non-compliance have been well documented in the literature and have been associated with poor health conditions and increased health care costs. Several studies have shown that the overall health care costs were lower for compliant patients than for non-compliant patients despite an increase in the pharmaceutical costs. Although, plenty research has been done describing the relationship between compliance and total health care costs, none has been conducted on specialty medications that are used to treat Multiple Sclerosis (MS). The primary purpose of this study was to examine the relationship between patients' compliance to MS specialty medications and the total health care (pharmacy and medical) costs. Methods: The study was conducted using a retrospective study design utilizing all MS-related pharmacy and medical claims maintained by a large PBM. Patients who were diagnosed with MS and who have had at least one prescription for specialty medications between July-December 2005 were identified and selected for the analysis. Patient's compliance to specialty medications was assessed in terms of MPR. Patients were categorized into three different groups as - compliant (MPR > 0.8), partially compliant (MPR 0.5-0.8), and non-compliant (MPR <0.5). All drug costs; all medical non drug costs as well as total health care costs were computed and compared across the three groups using independent t-tests. Results: A total of 104 patients met the inclusion criteria and were included for the analysis. Compliant group patients had significantly high all drugs cost and significantly lower medical non-drug cost as compared to the non-compliant and partially compliant groups. The total health care costs were found to be comparable and non-significant among the three groups. Conclusion: Patients who were compliant to their specialty medications reported higher pharmacy cost and lower medical cost, thereby implying that there exists an inverse relationship between compliance and medical costs.