Late-onset hydrocephalus in an 11-year-old girl with extensive drug-resistant tuberculosis—a rare occurrence

Abstract Introduction Central nervous system tuberculosis (CNS-TB) commonly presents with tuberculous meningitis and may be complicated by hydrocephalus, usually early in the disease course. Late-onset hydrocephalus occurring after prolonged anti-tubercular therapy (ATT) is rare. Case description We report an 11-year-old girl with extensively drug-resistant (XDR) CNS-TB with multiple tuberculomas who developed asymptomatic obstructive hydrocephalus after 18 months of ATT. She initially presented with miliary TB and subsequently developed TB meningitis with multiple intracranial tuberculomas. Despite prolonged second-line ATT, corticosteroids, and thalidomide for paradoxical upgrading reactions (PUR), follow-up MRI revealed moderate triventricular obstructive hydrocephalus due to persistent tuberculomas. She remained neurologically asymptomatic but required ventriculo-peritoneal shunt placement. Discussion This case highlights the possibility of late-onset hydrocephalus in drug-resistant CNS-TB, emphasizes the role of long-term neuroimaging surveillance, and underscores the need for vigilance even in clinically stable patients on prolonged ATT.