Introduction: Tuberculosis (TB) is a significant health problem in developing countries like India. The rising trend of multidrug-resistant (MDR) TB and extrapulmonary TB (EPTB) imposes an additional burden on TB management. Though TB is endemic in India, calvarial TB is very rare, contributes only to 0.2–1.3% of skeletal TB and very limited data is available on calvarial MDR-TB. In this report, we present a rare case of multidrug-resistant tuberculous osteomyelitis affecting the frontal bone and involving the orbit in a young boy. Case description: we present a case of a 14-year-old boy presented with a 3-month history of a gradually progressive, painless swelling in the scalp, located just above the forehead on the left side. additionally, he reported a 1-month history of swelling in the left eyelid. He also had history of fever, evening rise of temperature, loss of appetite and loss of weight of 4 kg over 3 months. On examination, there was a fluctuant swelling of 6 x 7 cm in left side of the forehead, without any signs of inflammation or sinuses. The left eyelid was swollen with yellowish material inside, leading to severe mechanical ptosis. The right eye was normal. Systemic examination was normal. On investigation, CECT revealed focal area of osteolysis and erosion in the left half of the frontal bone with osteolytic extension into the roof and lateral wall of the left orbit along with adjacent interconnecting epidural and subgaleal collection. GeneXpert ULTRA from the aspirated pus detected Mycobacterium tuberculosis and Rifampicin resistance. Later TB culture confirmed the presence of Mycobacterium tuberculosis and the line probe assay detected Rifampicin resistance and high-level Isoniazid resistance. Hence, a definitive diagnosis of multidrug resistant tuberculous osteomyelitis affecting the left frontal bone with orbital involvement was made. The patient showed significant clinical improvement with bedaquiline-based oral longer MDR-TB regimen without any surgical intervention. Discussion: Calvarial tuberculosis is a very rare entitity, but can be managed effectively with proper antituberculous drug regimen. Hence tuberculous osteomyelitis should be considered as a possible diagnosis when encountering lytic lesions of the skull. Maintaining a high index of suspicion, facilitating early diagnosis, and ensuring appropriate treatment is essential for achieving favorable outcomes in patients with calvarial tuberculous osteomyelitis.