Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 26 (2014) 545–548 Contents lists available at ScienceDirect Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology j o ur nal ho me pa ge: www.elsevier.com/locate/jomsmp Case Report A rare case of maxillary osteomyelitis caused by Curvularia species Arpita Rai a,∗ , Lijoy Abraham b , Venkatesh G. Naikmasur b , R.D. Kulkarni c , S. Padmini d a Department of Oral Medicine, Faculty of Dentistry, Jamia Millia Islamia, New Delhi 110 025, India b Department of Oral Medicine and Radiology, S.D.M. College of Dental Sciences & Hospital, Dharwad, Karnataka 580 009, India c Department of Microbiology, S.D.M. College of Medical Sciences & Hospital, Dharwad, Karnataka 580 009, India d Department of Oral Pathology, S.D.M. College of Dental Sciences & Hospital, Dharwad, Karnataka 580 009, India a r t i c l e i n f o Article history: Received 2 July 2013 Received in revised form 9 January 2014 Accepted 9 April 2014 Available online 26 June 2014 Keywords: Fungal osteomyelitis Curvularia Chronic sinusitis Maxillary sinusitis Immunocompromised patients a b s t r a c t Orofacial fungal infections display different etiologies, pathogenesis, and clinical presentations. The inci- dence of rare mycoses of oral cavity is very low. We report a case of chronic invasive sinusitis caused by Curvularia infection which presented as osteomyelitis of maxilla. Intraoral manifestation of Curvu- laria infection is seldom reported in the literature. Most cases of fungal infection in immunocompetent patients present in a noninvasive fashion as an allergic fungal sinusitis. Sinusitis due to unusual fungal pathogens, like Curvularia, is thought to occur primarily in immunocompromised individuals and poorly controlled diabetics. Invasive fungal sinus disease has a very high morbidity and mortality. For optimal outcome, rapid diagnosis and reversal of the underlying condition is critical to success. © 2014 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.  1. Introduction Dematiaceous or darkly pigmented fungi comprise large, het- erogeneous group of organisms that have been associated with a variety of clinical syndromes. In recent years, these fungi have been increasingly recognized as important pathogens, and the spectrum of diseases with which they are associated has also broadened. Curvularia infections in humans are relatively uncommon despite the ubiquitous presence of this soil-dwelling dematiaceous fungus in the environment. Most cases of sinusitis caused by Curvularia are primarily allergic in nature. Involvement of this fungus in invasive sinusitis is quite rare and intraoral involvement is seldom. Diagno- sis depends on a high degree of clinical suspicion and appropriate pathological and mycological examination of clinical specimens. Itraconazole and voriconazole demonstrate the most consistent in vitro activity against this group of fungi [1]. We report a case of chronic invasive sinusitis caused by Curvularia infection which presented as osteomyelitis of maxilla.  Asian AOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathol- ogy; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. ∗ Corresponding author. Tel.: +91 8802536376. E-mail address: arpitadoc@gmail.com (A. Rai). 2. Case report A 77-year-old male reported to the Department of Oral Medicine and Radiology with a complaint of halitosis for a duration of 15 days. The patient reported a history of recurring rhinosinusitis over the preceding several years which had worsened 9 months earlier when he experienced orofacial pain mainly localized to right max- illa associated with a rapidly progressing swelling of the right side of the face. He also reported nasal congestion, posterior nasal dis- charge and progressive fatigue. The patient had consulted an ENT surgeon and multiple courses of antibiotics had been prescribed. Relief in pain was noticed but the swelling persisted, and there- fore, the patient underwent endoscopic sinus surgery following 6 months of medications. Following the sinus surgery, patient was asymptomatic for 2 months, when finally he had approached us with complaints of halitosis. There was congenital and familial history of hearing loss and aphasia. The patient also reported a past medical history of diabetes mellitus since 20 years. The patient was earlier taking glimiperide 3 mg twice daily before food and metformin 500 mg thrice daily after food, but due to poor glycemic control the patient had been upgraded to insulin by his primary care physician 6 months previously. The patient had been prescribed human mixtard insulin (30:70) administered through subcutaneous route. Presently the patient was taking 18 IU of insulin half an hour before breakfast and 14 IU of insulin half an hour before dinner http://dx.doi.org/10.1016/j.ajoms.2014.04.004 2212-5558/© 2014 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved. 