We describe the first report of intrabronchial Pseudallesheriasis in a 74-year-old Japanese woman with a one-year history of refractory cough and an intrabronchial lesion. After endoscopic removal of a part of the lesion, her symptoms were markedly improved. Oral itraconazole therapy was begun, but right lower lobe pneumonia developed due to S. apiospermum. Continuous infusion of amphotericin B in a total dose of 1096 mg was performed and the pneumonia resolved completely. Our experience suggests that intravenous amphotericin B may be the treatment of choice for Pseudallesheriasis, even if the lesion is limited in bronchus.