Case report/Cas clinique Non-sinonasal isolated facio-orbital mucormycosis A case report S.K. Swain a, *, M.C. Sahu a , A. Banerjee b a Department of Otorhinolaryngology, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India b Department of Anesthesia, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India 1. Introduction Mucormycosis is a rare and dreaded disease caused by a fungus of the order Mucorales. It has high morbidity and mortality [1]. Most commonly Rhizopus species are causative organisms for the mucormycosis. Diabetic ketoacidosis and neutropenia are common predisposing conditions. This is a life-threatening infection which has remarkable affinity for arteries [2]. This fungus often dissects internal elastic lamina from the media of the blood vessels, resulting extensive damage to the endothelium and lead to thrombosis. Mucormycosis is classified into different forms as per anatomic sites like rhino maxillary, central nervous system, cutaneous, pulmonary, disseminated and miscellaneous. The rhino-orbito-cerebral is the most common variety of mucormy- cosis [3]. The most common sites of mucormycosis infections are paranasal sinuses (39%), lungs (24%), skin (19%), brain (9%), gastrointestinal (7%) forms and other miscellaneous types are extremely rare [4]. The ideal treatment need correction of underlying risk factors, antifungal treatment with Amphotericin B and aggressive surgery. Here we are reporting a case non- sinonasal mucormycosis which only affecting the orbit and facial area. 2. Case report A 56-year-old lady attended the outpatient department of Otorhinolaryngology with a swelling (3 cm 3 cm) in the right facio-orbital area (Fig. 1) since 2 months. She was known case of diabetic mellitus and hypertensive patients. She was under treatment with oral hypoglycemic agents like metformin and glipizide but her blood sugar was poorly controlled (HbA1c 10.6%). Diagnostic nasal endoscopy showed normal nasal cavity and nasopharynx. Ophthalmological consultation revealed loss of the vision in the right eye. Computed tomography (CT) scan of the nose and paranasal sinuses revealed normal nasal cavity but a mass involving right orbit and adjacent facial area (Fig. 2). A small piece of biopsy sample was taken from the facio-orbital mass (Fig. 3) which showed the picture of mucormycosis with some foci of non- septate fungal hyphae with right angled hyphal branches (Fig. 4). She had undergone radical excision of the mass along with orbital exenteration followed by parenteral liposomal Amphotericin B (5 mg/kg). Patient follow-up showed no evidence of recurrence after six months of surgery. 3. Discussion Mucormycosis is caused by saprophytic fungi of many genera related to phycomycetes (zygomycetes) and order Mucorales [5]. The fungus has great affinity towards to arteries and adheres to the arterial wall. It grows along the internal elastic lamina of blood vessels causing thrombosis, ischemia and necrosis of the surrounding tissues. Mucorales are abundantly seen in soil, decaying vegetables, animal excreta and foodstuffs. They grow rapidly in humid environment and the sporangiospores are released and spread as airborne propagules. In India, the air borne spores are more during transition from summer to rainy season as it may be ideal for fungal growth [6]. Mucormycosis is a rare clinical entity and often affect immunocompromised patients. Journal de Mycologie Me ´ dicale 28 (2018) 538–541 A R T I C L E I N F O Article history: Received 3 January 2018 Received in revised form 30 April 2018 Accepted 3 May 2018 Keywords: Mucormycosis Facio-orbital Amphotericin B Surgical debridement A B S T R A C T Mucormycosis is a rare clinical entity, often affect immunocompromised patients. It is an emergency situation and has poor prognosis. Prompt diagnosis with tissue biopsy, local control of the disease by aggressive surgical debridement and appropriate systemic antifungal treatment improve the prognosis and survival of the patients. Treatment of mucormycosis needs antifungal agents such as Amphotericin B and wide surgical debridement. Early diagnosis and treatment is often needed for survival of the patients. We describe a rare case of mucormycosis affecting facio-orbital area without involving sinon- nasal cavity. C 2018 Elsevier Masson SAS. All rights reserved. * Corresponding author. Department of Otorhinolaryngology, IMS and SUM Hospital, Bhubaneswar, Odisha, India. E-mail address: santoshvoltaire@yahoo.co.in (S.K. Swain). Available online at ScienceDirect www.sciencedirect.com https://doi.org/10.1016/j.mycmed.2018.05.003 1156-5233/ C 2018 Elsevier Masson SAS. All rights reserved.