Objective : Prevalence of Mycobacterium tuberculosis infection in children still high in the world including Indonesia. Ocular tuberculosis (TB) is a rare extrapulmonary manifestation of Mycobacterium tuberculosis but can causes blindness when not detected and treated promptly. 
Case : A marasmus 2-year-old girl was admitted due to subacute loss of vision, swelling, pain, and redness with pus in her left eye. She was diagnosed with endophthalmitis and underwent enucleation. Two weeks after, she suffered from loss of consciousness and focal seizure. She was not vaccinated for BCG. Physical examination showed GCS of E3M5V3, nuchal rigidity, increased physiological reflex, spasticity, and clonus. The histopathology displayed tubercles, epithelioid histiocytes, and Datia Langhans cells. The lumbar puncture exhibited 70% mononuclear cells, protein 240 mg/dL, glucose 5 mg/dL, and acid-fast bacilli +2.  Brain CT with contrast showed multiple nodules with perifocal oedema in right cerebellum and midbrain, causing hydrocephalus. 
During hospitalization, the girl was diagnosed with meningoencephalitis TB with secondary ocular TB infection. She was treated with ceftriaxone 350 mg b.i.d, fixed-dose combination (rifampicin, pyrazinamide, and INH) 1 tab q.d, dexamethasone 0.5 mg/kg/day, phenytoin injection 5 mg/kg/day, and VP shunt procedure. Throughout 16 days of care, there was no seizure and level of consciousness improved. She was then discharged to continue the regimen in outpatient clinic.
The ocular manifestation of TB can be caused by either hematogenous spread or direct peri-structural invasion from intracranial TB  as the main port d’entrée. It frequently misdiagnosed since it resembles many other unspecific ocular infections. 
Conclusion : In children with severe clinical manifestation in ocular with comorbidities marasmic malnutrition and lack of vaccination, should be considered as Mycobacterium tuberculosis ocular infection. Accurate diagnosis of extrapulmonary TB infection and anti-tuberculosis regimen turn serve as basis for prompt.