AIM: This study was conducted to assess the prevalence of dental caries, malocclusion and facial changes in thalassemia major patients of western Rajasthan population. Methods: In this cross-sectional study, 50 patients with thalassemia major, and 50 healthy matched control individuals, were included. Dental caries indices, DMFT and DMFS were measured and compared with age matched healthy controls. Data were analyzed by t-student test. Results: Poor oral hygiene was generally observed. Dental caries were significantly higher in thalassemic patients (p<0.0001) in comparison with the healthy control group. Various malocclusion stages were seen. Facial changes were also seen (mongoloid features due to prominence of cheek bones, protrusion of the maxillary anterior teeth, and the depression of the bridge of the nose). Conclusion: Patients with β- thalassemia major showed highly significant differences in orodental and orofacial manifestations as compared to control group.