A very low birth weight female infant developed a systemic infection by Candida albicans. Her clinical conditions were very poor; involvement of CNS was demonstrated my multifocal hyperechogenic images, spreading to the periventricular and subcortical areas of brain, confirmed by CT as circumscribed hyperdense punctiform granulations suggesting initial cerebritis. The introduction of liposomal Amphotericin-B in the therapy resulted in a marked improvement of clinical conditions together with the disapparance of pathological brain images. Longitudinal examinations carried out every three months during the first year, and every six months during the second, have shown adequate psychomotor development. The results of late cerebral nuclear magnetic resonance (NMR) imaging were within the norm. The patient is now seven-years-old and shows a well-balanced neuropsychological development. Systemic Candida infections of VLBW infants with CNS involvement have usually a very poor prognosis in terms of death or residual brain damage. The use of liposomal Amphotericin-B allowed us to achieve the complete cure of our patient, suggesting an high efficacy together with at a lower toxicity in respect of traditional treatments.