Background Little is known about the epidemiology of multidrug resistant tuberculous meningitis (TBM), defined as resistance to at least isoniazid and rifampin. We sought to describe cases of multidrug resistant TBM reported in the United States with respect to clinical characteristics and treatment outcomes.

Methods We conducted a retrospective cohort study using data collected by the National Tuberculosis Surveillance System at the Centers for Disease Control and Prevention in the United States. We included patients diagnosed with TBM between 1 January 1993 and 31 December 2005, with a positive culture from cerebrospinal fluid and drug susceptibility testing performed. We compared clinical and demographic characteristics of TBM patients with and without multidrug resistance, as well as the proportion of patients in each group that died while still receiving anti-tuberculosis therapy.

Results 26 of 1683 patients (1.5%) with cerebrospinal fluid culture-positive TBM were found to have multidrug resistance on initial susceptibility testing. Anti-tuberculosis therapy was stopped due to death in 19 of 26 patients (73%), after a median of 42 days of treatment (IQR 15–225). Of the 19 patients with known HIV status, 17 were HIV-positive (89%).

Discussion Most cases of multidrug resistant TBM in the United States occurred in HIV-infected patients, and the associated mortality was high. Due to the time period required to obtain drug susceptibility results based on conventional methods, rapid molecular diagnosis of drug resistance is of great potential benefit in this setting and needs to be evaluated further.