Introduction: β-Thalassemia is genetic disorder which clinically presents as anaemia due to decreased production of beta-chains of haemoglobin molecule. Literature on oro-facial manifestations, oral health status is sparse and incon- clusive in the β-thalassemia major patients (TM). Methods: This cross-sectional study was done to assess oro-facial manifestations of β-Thalassemia. 31 TM patients in the age group of 6-18 years were clinically examined and oro-fa- cial manifestations were recorded. Oral health status including oral hygiene status, dental caries, gingival status of these TM children was compared with 42 healthy children. The mean values were compared using Student’s t-test. Treatment needs of both the groups were recorded using WHO oral health assessment form. Results: Lip incom- petence (38.75%) and maxillary protrusion (19.35%) were most common oro-facial manifestations of Thalassemia major. Mean OHI-S score for TM and healthy children were comparable 1.73 ± 1.10 and 1.36 ±1.17 respectively (p=.178) but mean GI score for TM was higher than healthy children, 1.42±.39 and .94±.51 respectively (p<.05). Mean DMFT scores for TM and healthy children were 1.5±1.8 and 1.2±2.1 respectively (p=.578). Treatment needs was relatively higher among TM children with 58% of these children requiring one surface filling. Conclusion: Lip incompetence followed by maxillary protrusion were most common oro-facial manifestations of Thalassemia major. There is no significant association between oral hygiene and dental caries with Thalassemia major. However, thalas- semia major is associated with gingivitis. Treatment requirement was relatively higher among TM children with one surface filling being the commonest need.