Summary:A 56-year-old dairy farmer received a fully HLA matched unrelated donor marrow transplant for high risk CML in chronic phase. His early post-transplant course was complicated by a series of massive intracerebral bleeds and by sepsis related to a malignant otitis externa. The microbial pathogen isolated from ear swabs was found to be Absidia corymbifera, but CT scan at the time showed no intracerebral extension. Despite neutrophil engraftment and aggressive antifungal management he succumbed. Autopsy revealed invasion of Absidia into the brain from the ear. We speculate that colonisation by Absidia resulted from occupational exposure. Bone Marrow Transplantation (2000) 26, 701-703. Keywords: zygomycosis; Absidia corymbifera; bone marrow transplantation; leukaemia Zygomycosis is an uncommon, although frequently lethal, opportunistic fungal infection caused by members of the order Mucorales most commonly Rhizopus, Absidia and Rhizomucor. 1 These fungi are ubiquitous saprophytes found in soil, manure, plants and decayed food, and can be pathogenic in immunocompromised patients. 1 Zygomycosis is most commonly seen in patients with diabetic ketoacidosis but the incidence in those with haematological malignancies is increasing, probably as a result of newer, more aggressive chemotherapy and bone marrow transplantation regimens. 1,2 Rhinocerebral involvement is the commonest presentation overall although pulmonary infection is more common in haematology patients. 1,3 We describe an unusual case of zygomycosis in a dairy farmer with CML who had undergone an HLA matched unrelated donor bone marrow transplant (BMT).
Case reportA 56-year-old dairy farmer had high risk CML, having trisomy 8 in addition to the Philadelphia chromosome. His disease had failed to respond to alpha-interferon and he therefore underwent high-dose chemotherapy, followed by a T cell-depleted, fully matched unrelated donor BMT. He was an extremely fit and well man who gave no significant past medical history. He tolerated the conditioning regimen consisting of Campath-lH, fludarabine, cyclophosphamide and single fraction TBI very well with no significant complications.On day +4 post transplant, he complained of pain in his right ear, which had developed over the previous 12 h. Direct questioning revealed intermittent earache over several years, which he had previously not disclosed. Direct view of his right eardrum revealed no obvious erythema of the tympanic membrane which was intact. Although afebrile, swabs were taken from the ear and throat and he was commenced on piperacillin/tazobactam 4.5 g three times a day. On day +5 the pain extended to the right jaw and he developed a lower motor neurone right VIIth cranial nerve palsy. The tympanic membrane now looked swollen and erythematous with purulent material in the external auditory canal, which was thought to represent malignant otitis externa by the ENT surgeons, who recommended additional topical gentamicin drops. Urgent CT scan showed some opacification of the right mastoid with soft ti...