Successful treatment of rhinocerebral zygomycosis and pulmonary aspergillosis in a patient with thymic acute lymphoblastic leukemia

R. Kuehnbach, A. Muth, M. Dellian, S. Boeck, X. Schiel, B. Grabein, E. Hiller

Abstract: 

Posaconazole (POS) is an extended-spectrum triazole antifungal agent under clinical investigation that has activity against many pathogenic yeasts and moulds, including Zygomycetes and Aspergillus species. This report describes the successful treatment of a disseminated fungal infection involving the lung and brain in a leukemic patient with surgical resection, liposomal amphotericin B (L-AMB) and POS.

A 46-year-old man with T-cell acute lymphoblastic leukemia presented with left maxillary sinusitis and exophthalmos at the end of leukocyte recovery after the first cycle of chemotherapy in July 2003. Imaging showed inflammation of the left maxillary sinus, orbit and masseter compartment; Rhizopus species was cultured from a biopsy. High-dose L-AMB (15 mg/kg/d) was started immediately followed by radical surgery of all paranasal sinuses and the left periorbital region, which resulted in a partial response. During a period of pancytopenia after the second cycle of chemotherapy, progression of zygomycosis in the left pterygopalatine fossa and new intraorbital and CNS manifestations occurred. A radical excision including enucleation of the left eye and resection of the left temporal dura were performed. Cultures from all locations were positive for Rhizopusspecies. During September 2003, a CT scan of the lung showed an aspergilloma in the left lower lobe and the patient underwent lobectomy. The histology was positive for mycelium, the culture showed no growth. L-AMB (total dose = 43g) was continued for another 6 weeks; however, renal toxicity and polyneuropathy resulted in a decrease of L-AMB dosing and finally enrollment in an open-label treatment use program with POS. L-AMB was discontinued and POS oral suspension 800 mg/d was initiated in October 2003. The patient became clinically stable in the following period and underwent reinduction chemotherapy. Several episodes of severe pancytopenia occurred during subsequent chemotherapy cycles, but there was no relapse of zygomycosis and no new manifestations of fungal infection. As of August 2004, the patient remains clinically stable on POS without evidence of active fungal infection. This outcome suggests that POS is well tolerated and protected our patient from relapse of fungal infections, particularly zygomycosis.

2004

Full conference title: 

Réunion Interdisciplinaire de Chimiothérapie Anti-Infectieuse
    • RICAI 24th (2004)