<p>Post-infectious hydrocephalus (PIH) in neonates is associated with substantial neurodevelopmental morbidity, and cerebrospinal fluid diversion alone does not address the underlying inflammatory and parenchymal injury. Mesenchymal stem cells derived from human exfoliated deciduous teeth (SHED) exhibit neurotrophic and immunomodulatory properties with potential translational relevance in inflammatory brain conditions. We report a preterm neonate with severe Acinetobacter-associated PIH requiring ventriculoperitoneal shunt placement who concurrently received intraventricular administration of allogeneic SHED under a compassionate-use framework. The procedure was technically feasible and well tolerated, without immediate procedure-related adverse events. Follow-up neuroimaging demonstrated reduced ventricular size and progressive cortical mantle thickening, accompanied by clinical gains in alertness, feeding, and motor function. Although causal inference cannot be established from a single observation and concurrent cerebrospinal fluid diversion represents a major confounder, this first-in-human experience suggests short-term procedural safety and supports further controlled clinical investigation of intraventricular SHED therapy in neonatal post-infectious hydrocephalus.</p>