Treatment for latent tuberculous infection (LTBI) is a key strategy for the elimination of tuberculosis. Rare adverse reactions associated with LTBI treatment have been reported. We report the only case of acute kidney injury reported to Centers for Disease Control and Prevention surveillance for LTBI treatment-related adverse events. The patient experienced rapid intravascular hemolysis, resulting in heme pigment nephropathy; he was hospitalized and received three hemodialysis treatments, but recovered without sequelae. While LTBI treatment-related adverse events are rare, health care providers should maintain clinical vigilance and regularly counsel patients to facilitate prompt diagnoses and effective clinical management of affected patients.