Orbital mucormycosis in immunocompetent children; review of risk factors, diagnosis, and treatment approach

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Orbital mucormycosis in immunocompetent children; review of risk factors, diagnosis, and treatment approach Ali Amanati1, Hamide Barzegar2*†, Gholamreza Pouladfar1, Anahita Sanaei Dashti1, Mohamad Bagher Abtahi2, Bijan Khademi3, Mohammad Javad Ashraf4, Parisa Badiee1, Seyedeh Sedigheh Hamzavi1 and Ali Kashkooe2*†

Abstract Background: Orbital mucormycosis is a rare but potentially severe and troublesome invasive fungal infection that could be occurred even in healthy individuals. The initial clinical presentation is similar to bacterial pre-septal or septal cellulitis, especially in early stages. Case presentation: Herein, we describe the successful management of a series of five cases presenting with orbital mucormycosis in previously healthy children. Conclusions: Orbital mucormycosis is extremely rare in healthy children and maybe life-threatening when diagnosis delayed given a similar clinical presentation with bacterial septal cellulitis. Intravenous antifungal therapy with amphotericin B and timely surgical drainage is live-saving. Keywords: Mucormycosis, Orbital, Immunocompetent, Children

Introduction Mucormycosis is the third most common invasive fungal infections following candidiasis and aspergillosis [1]. Mucormycosis is a life-threatening infection that could manifest as a local or systemic infection. Due to high mortality and morbidity, early diagnosis and treatment are crucial. One of the most common forms of mucormycosis is rhino-orbito-cerebral infection [2, 3]. Local pain, chemosis, multiple cranial nerve palsies, unilateral peri-orbital facial pain, blepharoptosis, proptosis, acute ocular motility changes, ophthalmoplegia, headache, and acute vision loss are the most common signs and symptoms [1]. Diagnosis usually based on the identification of organisms in tissue by histopathology, culture, KOH preparation and molecular tests [4]. Surgical debridement, in addition to systemic * Correspondence: [email protected]; [email protected] † Hamide Barzegar and Ali Kashkoie contributed equally to this work. 2 Shiraz University of Medical Sciences, Namazi Hospital, 7193711351, Zand Ave, Shiraz, Iran Full list of author information is available at the end of the article

antifungal therapy, is the standard treatment approach which should be started early after diagnosis [5, 6]. In this case series, we present five previously healthy children, primarily presented with orbital cellulitis. Successful treatment and complete cure occurred despite a late diagnosis in all cases.

Case presentation Case 1

The 7-month-old girl visited with edema and erythema in the left inferomedial canthus for 6 weeks before admission. Her mother told that they were villagers and lived in a rural place near Shiraz city. They were on vacation in an area around their home. In the spiral orbital computed tomography (CT) scan with contrast, periorbital abscess formation, and evidence of dacryocystitis found. She scheduled promptly for orbital biopsy and abscess drainage. The results of the biops