Background: According to State Health Society, there are about 30 lakhs sickle cell carrier and 1.5 lakhs sufferers in Maharashtra, prevalence being more in tribal population. As Sickle cell disease is associated with increased morbidity and mortality, it is important for patients to have awareness regarding its cause, treatment and genetic implication. To assess Knowledge, Attitude and Practices regarding sickle cell disease in adult sufferers and carriers. Materials and Methods: The present study is an cross sectional study, carried out in 232 adult sickle cell disease sufferers and carriers in rural area of Chandrapur district from July 2014 to December 2015. Results: Out of 232 study subjects, 17.24% were sufferers and 82.76% were carriers. Only 62.50% knew the correct cause and only 42.67% had correct knowledge of types of disease. 76.30% had correct knowledge about symptoms of disease. 40.09% patients were availing treatment of some kind. It was found that only 6.77% had done premarital screening of their spouses and 100% had screened their children for sickle cell disease. Conclusion: Knowledge regarding sickle cell disease, its cause, types, treatment and prevention is satisfactory in sufferers but not in carriers. Overall attitude towards treatment and prevention is more or less positive. Treatment seeking behavior is good in sufferers but not in carriers.