A female preterm infant, 18 days old with a birth weight of 890 g, was hospitalized for respiratory distress that had persisted for 18 days following birth at a gestational age of 25 weeks and 6 days. Empirical antibacterial therapy at an outside hospital was ineffective, and a blood culture was positive for Candida albicans. Despite six days of fluconazole treatment, her condition showed no improvement. Upon transfer to our hospital, she received initial therapy with caspofungin combined with meropenem for one week. However, follow-up blood culture remained positive, and cerebrospinal fluid (CSF) analysis revealed an elevated white blood cell count. Cerebrospinal fluid metagenomic next-generation sequencing (mNGS) confirmed Candida albicans infection. With the involvement of a clinical pharmacist in the treatment team, the antimicrobial regimen was adjusted to liposomal amphotericin B combined with fluconazole. After 5 days of this adjusted therapy, the CSF white blood cell count decreased significantly. Two weeks later, CSF parameters showed marked improvement and the blood culture turned negative, although a progressive decline in platelet count was observed during monitoring. The clinical pharmacist subsequently assisted in formulating a de-escalation treatment plan, with a total therapy duration of 7 weeks. Ultimately, the infant was cured and discharged. This case suggests that liposomal amphotericin B is an effective agent for treating Candida meningitis in extremely preterm infants, although its adverse effects require close monitoring during therapy. Clinical pharmacists, by collaboratively formulating treatment plans, play a crucial role in ensuring both anti-infective efficacy and medication safety for critically ill, extremely preterm infants.