Background and aim Despite the high incidence of tuberculosis infection (TBI) in patients with hematological diseases, there were few researches on TBI in MDS patients. We retrospectively analyzed the characteristics of MDS patients with TBI in our center to improve the clinical diagnosis and treatment of these patients.Methods Primary MDS patients diagnosed between 2014 and 2019 at the First Affiliated Hospital of Zhejiang University were retrospectively reviewed, TBI was diagnosed by positive culture(s) or acid fast bacilli on modified Ziehl-Neelsen staining on clinical samples as well as compatible symptoms and signs.Results Thirty-four of 1347 MDS patients were diagnosed with TBI, nine patients had blast<5%, twelve patients were MDS with excess blast-1(MDS-EB1), and the rest 13 patients were MDS with excess blast-2 (MDS-EB2). Ten patients had lower risk (low and intermediate) and 24 patients had higher risk (high and very high) according to revised international scoring system (IPSS-R). Moreover, fifteen patients were classified into synchronous group (defined as TBI occurring within 3 months of MDS diagnosis), and the rest 19 patients were non-synchronous group (defined as TBI developed after MDS more than 3 months). Patients in synchronous group have higher rate of persistent fever (11/15 vs. 7/19, p=0.03), higher risk classification ( 14/15 vs. 8/19, P=0.002 ), and higher tendency to transform to acute myeloid leukemia (AML) (8/15 vs. 4/19, P=0.04 ) than those in non-synchronous group. The median time to AML was significantly shorter in synchronous group (7.68 months vs. 23.83 months, P<0.01). Most patients received regular anti-tuberculosis treatment (ATT)(33/34) and support care(22/34) for MDS, patients received support care were more prone to proceed to AML than those received active therapy such as decitabine (12/22 vs. 2/12, P=0.03). The median OS of all patients in synchronous group was just 14.17 months, while in non-synchronous group, it was 34.07 moths ( P = 0.01). However, treatments for MDS had no effect on OS(P=0.67).Conclusion TBI is not uncommon among patients with MDS, patients with synchronous TBI and MDS progress faster and have shorter OS, which may be associated with genetic susceptibility and lack of active therapy.