We report a case of Penicillium marneffei infection in a previously healthy 3.5 years-old Chinese boy and review the literature. The details of the case are described with emphasis on his immune function and treatment outcome. This boy had transient immunodeficiency involving phagocytic and NK cells due to P. marneffei infection which resolved after treatment with gamma interferon and amphotericin B. A prolonged course of fluconazole of 1 year was successful in preventing relapse. Gamma interferon, amphotericin B and a prolonged course of fluconazole may be useful in the treatment of life-threatening infection by P. marneffei.