Disseminated Saksenaea infection in an immunocompromised host associated with a good clinical outcome: a case report and

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Disseminated Saksenaea infection in an immunocompromised host associated with a good clinical outcome: a case report and review of the literature N. Davidson1,2* , K. Campbell1, F. Foroughi3, V. Tayal1, S. Lynar1,4, L. C. Crawford5, S. E. Kidd5, R. Baird3, J. Davies1,4† and E. M. Meumann1,4†

Abstract Background: Saksenaea species (spp.) are uncommon causes of mucormycosis but are emerging pathogens mostly associated with trauma and soil contamination often in immunocompetent hosts. Due to lack of sporulation in the laboratory, diagnosis and susceptibility testing is difficult so optimal treatment regimens are unknown. Case presentation: A 67 year-old man from the Northern Territory in Australia, with a history of eosinophilic granulomatosis with polyangiitis, developed disseminated Saksenaea infection after initially presenting with symptoms consistent with bacterial pyelonephritis. Despite a delay in diagnosis; with aggressive surgical management and dual therapy with amphotericin B and posaconazole, he survived. Conclusions: We describe an unusual case of disseminated infection with a favourable outcome to date. Keywords: Mucormycosis, Disseminated fungal infection, Amphotericin B, Posaconazole

Introduction Saksenaea spp. are of the order Mucorales and are infrequent causes of mucormycosis worldwide [1]. Saksenaea spp. were first described in 1953 (as Saksenaea vasiformis) from soil in India and since then have been increasingly reported in human disease causing a diverse spectrum of clinical illness mostly in tropical and subtropical regions [2]. With the advent of molecular typing, multiple species have been described within the genus including S. vasiformis, S. erythrospora, S. oblongispora, S. loutrophoriformis, S. trapezispora and S. dorisiae [3–5]. In contrast to other causes of mucormycosis, Saksenaea spp. infections often cause soft tissue or bone * Correspondence: [email protected] † J. Davies and E. M. Meumann contributed equally to this work. 1 Division of Medicine, Royal Darwin Hospital, Darwin, Australia 2 Sullivan and Nicolaides Pathology, Brisbane, Australia Full list of author information is available at the end of the article

and joint infections in immunocompetent hosts following traumatic inoculation. Rhinosinusitis and disseminated disease are much less common but have been associated with poor outcomes with survival reported in only two previous cases of disseminated infection [1]. We describe a case of disseminated infection by a Saksenaea species with a good clinical outcome and highlight rheumatological conditions as under-recognised risk factors for mucormycosis.

Case report A 67 year-old male living in tropical northern Australia presented with left sided abdominal pain, fever and pyuria. Computed tomography (CT) demonstrated abnormal enhancement of the lower pole of his left kidney, thickened pelvic urothelium and perinephric fat stranding. A provisional diagnosis of pyelonephritis was made,

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