Lindnera (Pichia) fabianii (Candida fabianii teleomorph) is a yeast species that is an uncommon cause of invasive human infections. This report describes what we believe to be the first human case of a catheter-related L. fabianii bloodstream infection in a neutropenic patient. The Clinical and Laboratory Standards Institute guidelines do not offer antifungal breakpoints in this neutropenic case and empirical chemotherapy was considered. Sharing our experience, we will discuss the choice of an effective antifungal agent in this uncommon clinical situation.