Contents lists available at ScienceDirect Medical Mycology Case Reports journal homepage: www.elsevier.com/locate/mmcr Unusual case of otomycosis caused by Saksenaea vasiformis H. Trabelsi a , S. Neji a , I. Hadrich a,* , M. Sellami b , N. Khemakhem a , H. Sellami a , F. Makni a , B. Hammami b , A. Ayadi a a Laboratory of Parasitology-Mycology, Habib Bourguiba Hospital, Sfax, Tunisia b ENT Department, Habib Bourguiba Hospital, Sfax, Tunisia ARTICLE INFO Keywords: Mucormycosis Malignant otitis externa Saksenea vasiformis ABSTRACT Saksenaea vasiformis is a species of the order Mucorales rarely reported as a cause of human mucormycosis. We report an unusual case of S. vasiformis otitis occurring in a diabetic woman after penetration of an insect in the right ear. Direct microscopic examination of the clinical sample showed hyaline and non septate hyphae be- longing to the order Mucorales. Fungal identification was performed by sequencing the ITS region of the rDNA. To our knowledge, this is the first report of S. vasiformis infection in Tunisia. 1. Introduction Saksenaea vasiformis is an emerging fungus which was described in 1953 by Saksena as a new zygomycete [1]. It was first isolated from a forest soil in India [1]. It has been found in soil samples, driftwood, grains and in other geographic areas [2,3]. Saksenaea is able to cause severe human infections in both immunocompromised and im- munocompetent hosts. It is most often associated with cutaneous or subcutaneous lesions after trauma. Malignant otitis with this fungus is uncommon. In this report, we describe a case of malignant otitis externa complicated with parotid abscess and caused by Saksenaea vasiformis in a diabetic woman. 2. Case report A 54-year-old diabetic woman was referred to the otolaryngology department on the first of July 2015 (Sfax-Tunisia) with complaint of ear infection and facial asymmetry. She was suffering from otalgia and purulent otorrhea of the right ear since 7 days. There was no history of fever or other systemic symptoms. A probabilistic antibiotherapy was prescribed, without any improvement. An aggravation of the facial asymmetry with parotid abscess, right ear cellulitis and fever were noted. The patient indicated the penetration of an insect in the right ear. Left ear examination and physical examination was normal. The otoscopy examination showed the presence of pus in the right external auditory canal which was very inflammatory and the presence of granulations tissue and central perforation of right tympanic mem- brane. Neurological examination showed peripheral facial paralysis with a painful and inflammatory parotid tumefaction. The CT scan (at day + 1) found sub-mastoidal and para-pharyngeal abscess and cerebral venous thrombosis complicating a right malignant otitis externa (Fig. 1). Auricular swab, aspiration and samples of retro auricular collection, were practiced at day +17. The bacterial cultures were negative while the direct examination showed hyaline, broad and aseptate mycelium suggestive of Mucorales fungi. Auricular culture on Sabouraud medium without actidione allowed isolating cottony and white colonies. The microscopic examination revealed only broad aseptate hyphae without sporulation (Fig. 2). Histopathologic examination of the resected tissues showed zygomycete hyphae. Molecular identification based on PCR amplification and sequencing of rDNA internal transcribed spacer (ITS) regions was performed to identify the Mucorales fungi. The ITS1–5.8S-ITS2 regions of rDNA were amplified with the fungal universal primer pairs ITS1/ITS4 [4]. PCR product was sequenced and the etiological agent was than identified as Saksenaea vasiformis. The best match was obtained (99% similarity) with sequence of S. vasiformis EU644757. Our sequence (TN254AU15) was submitted to GenBank under the accession number: KU314816. The above mentioned sequences were aligned using the ClustalX V2.1 program [5] as implemented in BioEdit [6], sequence alignment Editor Version. 7.0.9.0 Software (Fig. 3). Multiple alignments of our sequence with those of two S. vasiformis isolates (GenBank accession numbers EU644757 and AY211275) are shown in Fig. 3. Additionally, https://doi.org/10.1016/j.mmcr.2020.02.001 Received 5 October 2019; Received in revised form 18 January 2020; Accepted 5 February 2020 * Corresponding author. Laboratory of Parasitology- Mycology, CHU Habib Bourguiba, Sfax, Tunisia. E-mail addresses: thouaida@yahoo.fr (H. Trabelsi), nejisourour@yahoo.fr (S. Neji), ineshadrich@yahoo.fr (I. Hadrich), sellami_moncef@yahoo.fr (M. Sellami), Khemakhem.Nahed@yahoo.fr (N. Khemakhem), hayetsellami1@gmail.com (H. Sellami), famakni@gmail.com (F. Makni), bouthainahk@yahoo.fr (B. Hammami), ali.ayadi@rns.tn (A. Ayadi). Medical Mycology Case Reports 27 (2020) 68–71 Available online 07 February 2020 2211-7539/ © 2020 Published by Elsevier B.V. on behalf of International Society for Human and Animal Mycology. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/). T