The Good's syndrome is a rare immunodeficiency disease characterized by thymoma and hypogammaglobulinemia. The most common cause of infection in patients with Good's syndrome is the recurrent sinopulmonary infection. One may assume that there could be an unknown immunocompromising condition such as Good's syndrome in cases of chronic rhinosinusitis refractory to treatment. We experienced a patient with Good's syndrome, presenting with chronic rhinosinusitis refractory to treatment. We report this case with a brief review of the literature.