Congenital tuberculosis (CTB) is the transmission of Mycobacterium tuberculosis infection from mother to infant during the intrauterine period or delivery. An 82-day-old infant presented to Nilratan Sircar Medical College and Hospital, Kolkata, with a history of persistent fever from Day 15 of age. Over the course of more than 2 months, there were pneumonia, hepatosplenomegaly and endophthalmitis which were unresponsive to a range of antibiotics, and there had been several admissions to local hospitals. On this admission, his chest radiograph and contrast-enhanced computed tomography (CECT) of the thorax demonstrated bilateral nodules and enlarged mediastinal lymph nodes. Ultrasound and CECT of the abdomen demonstrated hepatosplenomegaly with multiple hypodense enhancing lesions. GeneXpert of a gastric lavage on Day 2 of this admission detected M. tuberculosis without rifampicin resistance. The infant was diagnosed with tuberculosis (TB) complicated by caseating hepatic granulomas, which fulfilled Cantwell's diagnostic criteria for CTB. Magnetic resonance imaging of the orbit demonstrated focal heterogeneous lesions involving the anterior portion of the left ocular bulb and vitreous, suggesting panophthalmitis, which was unresponsive to intravitreal antibiotics. Following commencement of standard anti-TB therapy from Day 90 of life, there was clinical and radiological recovery of endophthalmitis. The mother had a cachectic appearance owing to weight loss, and she had attended only one antenatal appointment. She had a positive acid-fast bacilli sputum stain but was unwilling to allow a genital tract smear. In the spectrum of CTB, TB panophthalmitis is an extremely rare presentation, and, as far as we are aware, it has not been reported in a child.Abbreviations: AFB: acid-fast bacilli; ATT: anti-tuberculous therapy; CTB: congenital tuberculosis; CECT: contrast-enhanced computed tomography; CSF: cerebrospinal fluid; ESR: erythrocyte sedimentation rate; HIV: human immunodeficiency virus; TB: tuberculosis; MRI: magnetic resonance imaging; USG: ultrasonogram.