Rhinocerebral zygomycosis following bone marrow transplantation in chronic myelogenous leukaemia. Report of a case and review of the literature

Nenoff, P.; Kellermann, S.; Schober, R.; Nenning, H; Kubel, M; Winkler, Jörg; Haustein, U.-F.

doi:10.1111/j.1439-0507.1998.tb00355.x

Cited by 18 publications

(9 citation statements)

References 33 publications

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“…Unfortunately, the prognosis for forms with intracranial involvement is poor, despite these therapeutic strategies. Many single cases reported in the literature [18–24] and those covered in reviews [10, 25] have had negative outcomes. In our case, the negative outcome was probably conditioned by severe diabetes and the late surgical approach.…”

Section: Discussionmentioning

confidence: 99%

Case report. Fatal rhinocerebral zygomycosis due to Rhizopus oryzae

Romano

Miracco²,

Massai

et al. 2002

Mycoses

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Section: Discussionmentioning

confidence: 99%

Case report. Fatal rhinocerebral zygomycosis due to Rhizopus oryzae

Romano

Miracco²,

Massai

et al. 2002

Mycoses

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“…However, this patient had the classical clinical picture of rhinocerebral involvement. 4 It is of interest to note that the patient was a dairy farmer and we speculate that he became colonised with A. corymbifera following occupational exposure to the fungus. Associations between Absidia infections and farms have been previously described.…”

Section: Discussionmentioning

confidence: 99%

“…A review of 30 reported cases in bone marrow transplant recipients found a mortality rate of 80%. 4 Rhinocerebral zygomycosis is the commonest clinical presentation and this usually begins in the nasal or paranasal sinuses. Fungi invade blood vessels causing a purulent arteritis and thrombosis with subsequent infarction and necrosis of tissue.…”

Section: Discussionmentioning

confidence: 99%

Fatal invasive cerebral Absidia corymbifera infection following bone marrow transplantation

Paterson

Marshall

Shaw

et al. 2000

Bone Marrow Transplant

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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...

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“…Symptoms and signs usually evolve over 4 weeks [19,28] Rarely, mucormycosis initiates in the ear [29][30][31][32][33] and can extend from there to the brain [29,30,32]. Patients had type 2 diabetes with no ketoacidosis [32,33], hematologic malignancies [29][30][31], or allogeneic bone marrow transplantation. A lethal infection by Absidia corymbifera starting at the auricle was reported in a 17-year-old girl with diabetic ketoacidosis [34].…”

Section: Clinical Presentationmentioning

confidence: 99%

“…It has been proposed that characteristic involvement of the basal ganglia on CT and MRI scans should suggest the diagnosis of mucormycosis in patients with isolated cerebral infection [30].…”

Section: Radiologymentioning

confidence: 99%

Mucormycosis of the Head and Neck

Dan

2011

Curr Infect Dis Rep

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Mucormycosis of the head and neck is the most common form of infection by the order Mucorales, accounting for up to half of all cases. About 70% of rhinocerebral cases develop in diabetic patients. Less often, rhinocerebral mucormycosis occurs in solid organ transplant recipients, those with prolonged neutropenia, and those on deferoxamine therapy. Recently, rhinocerebral disease has been increasingly recognized in patients undergoing hematopoietic stem cell transplantation. A growing number of cases have presented as breakthrough infection in patients receiving voriconazole. The mortality rate of rhinocerebral mucormycosis has remained at 30% to 70% despite aggressive therapy. Early initiation of treatment substantially improves outcome, underscoring the need to maintain a high index of suspicion and promptly biopsy potential lesions. Surgical excision of necrotic tissue is essential. Lipid formulations of amphotericin B have become the standard treatment for mucormycosis. Posaconazole is useful as salvage therapy, but cannot be recommended yet as primary therapy.

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Rhinocerebral zygomycosis following bone marrow transplantation in chronic myelogenous leukaemia. Report of a case and review of the literature

Cited by 18 publications

References 33 publications

Case report. Fatal rhinocerebral zygomycosis due to Rhizopus oryzae

Case report. Fatal rhinocerebral zygomycosis due to Rhizopus oryzae

Fatal invasive cerebral Absidia corymbifera infection following bone marrow transplantation

Mucormycosis of the Head and Neck

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