Email this article Mucormycosis of the maxillary sinus in an immunocompetent woman with oro-antral fistula – a case report
Abstract
Mucormycosis is a very rare fungal infection, characterized by vascular invasion, rapid progression and high mortality. Individuals suffering from systemic diseases influencing the immune system are predisposed to this infection which is caused by saprophytic fungi belonging to the class of Mucormycetes in the order of Mucorales. In this paper, we present an unusual case of mucormycosis of the maxillary sinus in an immunocompetent patient with oro-antral fistula. The infection may occur in either a fulminant or chronic form. In the patient described, the disease was chronic and characterized by a mild course with local symptoms. In the disseminated form of mucormycosis, the mortality rate is almost 100%. A definitively higher survival rate is observed in immunocompetent patients with a localized form of the infection. Diagnosis of the disease is very difficult because of its clinical and radiological similarity to the more common fungal infection, aspergillosis. Early diagnosis plays a key role in the course of treatment and allows to reduce the scope of surgical intervention. According to most authors, first line treatment should be the liposomal form of amphotericin B. Some also advise surgical treatment involving the removal of fungal masses. In the patient described, both surgical and pharmacological treatment was conducted resulting in the complete resolution of the fungal infection after 12 months.
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Walsh TJ, Gamaletsou MN, McGinnis MR, Hayden RT, Kontoyiannis DP. Early clinical and laboratory diagnosis of invasive pulmonary, extrapulmonary, and disseminated mucormycosis (zygomycosis). Clin Infect Dis 2012;54 Suppl 1:S55-60.
Abdollahi A, Shokohi T, Amirrajab N, Poormosa R, Kasiri AM, Motahari SJ, et al. Clinical features, diagnosis and outcomes of rhino-orbito-cerebral mucormycosis – a retrospective analysis. Curr Med Mycol 2016;2(4):15-23.
Sahota R, Gambhir R, Anand S, Dixit A. Rhinocerebral mucormycosis – report of a rare case. Ethiop J Health Sci 2017;27(1):85-90.
Dzierżanowska D, Gil L, Jakubas B, Krycz-Krzemień S, Styczyński J. Epidemiologia i diagnostyka mikrobiologiczna inwazyjnej choroby grzybiczej. Post Nauk Med. 2015;28(6):403-9.
Marin-Mendez H, Monroy-Aguirre D, Rodríguez-Perales M, Caretta-Barradas S. Síndrome de ápex orbitario causado por mucormicosis orbito cerebral crónica e indolente: reporte de dos casos. An Orl Mex 2005;50(1):64-8.
Spellberg B, Walsh TJ, Kontoyiannis DP, Edwards J Jr, Ibrahim AS. Recent advances in the management of mucormycosis: from bench to bedside. Clin Infect Dis 2009;48(12):1743-51.
Berczy K, Lorincz A, Fonyad L, Prinz G. Diagnostic problems of the mucormycosis of the maxillary sinus Case presentation. Fogorv Sz 2017;110(1):15-9.
Gholinejad Ghadi N, Seifi Z, Shokohi T, Aghili SR, Nikkhah M, Vahedi Larijani L, et al. Fulminant mucormycosis of maxillary sinuses after dental extraction in patients with uncontrolled diabetic: Two case reports. J Mycol Med 2018;28(2):399-402.
Prabhu RM, Patel R. Mucormycosis and entomophthoramycosis: a review of the clinical manifestations, diagnosis and treatment. Clin Microbiol Infect 2004;10 Suppl 1:31-47.
Lanternier F, Lortholary O. Zygomycosis and diabetes mellitus. Clin Microbiol Infect 2009;15 Suppl 5:21-5.
Vaezi A, Moazeni M, Rahimi MT, de Hoog S, Badali H. Mucormycosis in Iran: a systematic review. Mycoses 2016;59(7):402-15.
Narayanan S, Panarkandy G, Subramaniam G, Radhakrishnan C, Thulaseedharan NK, Manikath N, et al. The “black evil” affecting patients with diabetes: a case of rhino orbito cerebral mucormycosis causing Garcin syndrome. Infect Drug Resist 2017;10:103-8.
Gutiérrez-Delgado EM, Treviño-González JL, Montemayor-Alatorre A, Ceceñas-Falcón LA, Ruiz-Holguín E, Andrade-Vázquez CJ, et al. Chronic rhino-orbito-cerebral mucormycosis: A case report and review of the literature. Ann Med Surg 2016;6:87-91.
Bouza E, Munoz P, Guinea J. Mucormycosis: an emerging disease. Clin Microbiol Infect 2006;12(Suppl 7):7-23.
Almyroudis NG, Sutton DA, Linden P, Rinaldi MG, Fung J, Kusne S. Zygomycosis in solid organ transplant recipients in a tertiary transplant center and review of the literature. Am J Transplant 2006;6(10):2365-74.
Dimaka K, Mallis A, Naxakis SS, Marangos M, Papadas TA, Stathas T, et al. Chronic rhinocerebral mucormycosis: a rare case report and review of the literature. Mycoses 2014;57(11):699-702.
Kwon-Chung KJ. Taxonomy of fungi causing mucormycosis and entomophthoramycosis (zygomycosis) and nomenclature of the disease: molecular mycologic perspectives. Clin Infect Dis 2012;54 Suppl 1(Suppl 1):S8-S15.
Celis-Aguilar E, Burgos-Paez A, Villanueva-Ramos N, Solorzano-Barron J, De La Mora-Fernandez A, Marnjarrez-Velazquez J, et al. An emergent entity: Indolent mucormycosis of the paranasal sinuses. A multicenter study. Int Arch Otorhinolaryngol 2019;23(1):92-100.
Ibrahim AS, Kontoyiannis DP. Update on mucormycosis pathogenesis. Curr Opin Infect Dis 2013;26(6):508-15.
Wolkow N, Jakobiec FA, Stagner AM, Cunnane ME, Piantadosi AL, Basgoz N, et al. Chronic orbital and calvarial fungal infection with Apophysomyces variabilis in an immunocompetent patient. Surv Ophthalmol 2017;62(1):70-82.
Mignogna MD, Fortuna G, Leuci S, Adamo D, Ruoppo E, Siano M, et al. Mucormycosis in immunocompetent patients: a case-series of patients with maxillary sinus involvement and a critical review of the literature. Int J Infect Dis 2011;15(8):e533-40.
Sun HY, Singh N. Mucormycosis: its contemporary face and management strategies. Lancet Infect Dis 2011;11(4):301-11.
Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human disease. Clin Microbiol Rev 2000;13(2):236-301.
Spellberg B, Edwards J Jr, Ibrahim A. Novel perspectives on mucormycosis: pathophysiology, presentation, and management. Clin Microbiol Rev 2005;18(3):556-69.
Masci JR, Wormser GP, ediotr. Mandell, Douglas, and Bennett’s Principles and Practice of Infectious Disease. 6th ed. Philadelphia: Churchill Livingstone; 2005. p. 125-28.
Biradar S, Patil SN, Kadeli D. Mucormycosis in a diabetic ketoacidosis patient: a case report. J Clin Diagn Res 2016;10(5):OD09-10.
Turner JH, Soudry E, Nayak JV, Hwang PH. Survival outcomes in acute invasive fungal sinusitis: a systematic review and quantitative synthesis of published evidence. Laryngoscope 2013;123(5):1112-8.
Vignale R, Mackinnon JE, Casella de Vilaboa E, Burgoa F. Chronic, destructive, mucocutaneous phycomycosis in man. Sabouraudia 1964;3(2):143-7.
Kim ST, Kim WS, Lee HH, Kim JY. Successful treatment of invasive rhinopulmonary mucormycosis with an indolent presentation by combined medical and surgical therapy. J Craniofac Surg 2013;24(2):e182-4.
Virk RS, Arora P. Chronic sinonasal aspergillosis with associated mucormycosis. Ear Nose Throat J 2007;86(1):22.
Jung H, Park SK. Indolent mucormycosis of the paranasal sinus in immunocompetent patients: are antifungal drugs needed? J Laryngol Otol 2013;127(9):872-5.
Jad B, Pottathil S, Raina S, Singh V. Paranasal Sinus Mucormycosis in an Immunocompetent Host: A Case Report. Int J Health Sci Res 2015;5(9):617-20.
Hemashettar BM, Patil RN, O’Donnell K, Chaturvedi V, Ren P, Padhye AA. Chronic rhinofacial mucormycosis caused by Mucor irregularis (Rhizomucor variabilis) in India. J Clin Microbiol 2011;49(6):2372-5.
Rumboldt Z, Castillo M. Indolent intracranial mucormycosis: case report. AJNR Am J Neuroradiol 2002;23(6):932-4.
Bertin H. Mucormicosis rinosinusal. Rev Otorrinolaringol Cir Cabeza Cuello 2003;63:122-6.
Odessey E, Cohn A, Beaman K, Schechter L. Invasive mucormycosis of the maxillary sinus: extensive destruction with an indolent presentation. Surg Infect (Larchmt) 2008;9(1):91-8.
Ketenci I, Unlü Y, Sentürk M, Tuncer E. Indolent mucormycosis of the sphenoid sinus. Otolaryngol Head Neck Surg 2005;132(2):341-2.
Bhansali A, Bhadada S, Sharma A, Suresh V, Gupta A, Singh P, et al. Presentation and outcome of rhino-orbital-cerebral mucormycosis in patients with diabetes. Postgrad Med J 2004;80(949):670-4.
Marx RE, Stern D. Oral and maxillofacial pathology: a rationale for diagnosis and treatment. 1st ed. Chicago: Quintessence Publishing; 2006. p. 104-6.
Major-Gralec J, Sempińska-Szewczyk J, Filczak K. Ophtalmic symptoms in rihno-orbito-cerebral mucormycosis. Case report. Okulistyka – Kwart Med 2009;4:53.
Galletti B, Gazia F, Galletti C, Perani F, Ciodaro F, Freni F, et al. Rhinocerebral mucormycosis with dissemination to pontine area in a diabetic patient: Treatment and management. Clin Case Rep 2019;7(7):1382-7.
Nithyanandam S, Jacob MS, Battu RR, Thomas RK, Correa MA, D’Souza O. Rhino-orbito-cerebral mucormycosis. A retrospective analysis of clinical features and treatment outcomes. Indian J Ophthalmol 2003;51(3):231-6.
Najafi N, Kermani F, Gholinejad Ghadi N, Aghili SR, Seifi Z, Roilides E, et al. Fatal rhinocerebral mucormycosis in a patient with ulcerative colitis receiving azathioprine and corticosteroid. Curr Med Mycol 2019;5(1):37-41.
Wang Y, Zhu M, Bao Y, Li L, Zhu L, Li F, et al. Cutaneous mucormycosis caused by Rhizopus microsporus in an immunocompetent patient. A case report and review of literature. Medicine (Baltimore) 2018;97(25):e11141.
Mills SEA, Yeldandi AV, Odell DD. Surgical treatment of multifocal pulmonary mucormycosis. Ann Thorac Surg 2018;106(2):e93-5.
Alghamdi S, Idress B, Alharbi A, Aljurais N. Case report: disseminated pulmonary mucormycosis involving spleen in diabetic patient with aggressive surgical approach. Int J Surg Case Rep 2019;54:42-6.
Song BI. F-18 fluorodeoxyglucose positron emission tomography/computed tomography image of gastric mucormycosis mimicking advanced gastric cancer. A case report. World J Clin Cases 2019;7(10):1155-60.
Termos S, Othman F, Alali M, Al Bader BMS, Alkhadher T, Hassanaiah WF, et al. Total gastric necrosis due to mucormycosis: a rare case of gastric perforation. Am J Case Rep 2018;19:527-33.
Mohammadi A, Mehdizadeh A, Ghasemi-Rad M, Habibpour H, Esmaeli A. Pulmonary mucormycosis in patients with diabetic ketoacidosis: a case report and review of literature. Tuberk Toraks 2012;60(1):66-9.
Munir N, Jones NS. Rhinocerebral mucormycosis with orbital and intracranial extension: a case report and review of optimum management. J Laryngol Otol 2007;121(2):192-5.
Thahim K, Jawaid MA, Marfani MS. Presentation and management of allergic fungal sinusitis. J Coll Physicians Surg Pak 2007;17(1):23-7.
Grant P, Skilbeck CJ. Rhinocerebral mucormycosis: a devastating rhinological condition. Practical Diabetes 2014;31(1):37-9.
Baldin C, Soliman SSM, Jeon HH, Alkhazraji S, Gebremariam T, Gu Y, et al. PCR-based approach targeting mucorales – specific gene family for diagnosis of mucormycosis. J Clin Microbiol 2018;56(10):e00746-18.
Millon L, Scherer E, Rocchi S, Bellanger AP. Molecular strategies to diagnose mucormycosis. J Fungi (Basel) 2019;5(1):24.
Tarani L, Costantino F, Notheis G, Wintergerst U, Venditti M, Di Biasi C, et al. Long-term posaconazole treatment and follow-up of rhino-orbital-cerebral mucormycosis in a diabetic girl. Pediatr Diabetes 2009;10(4):289-93.
Sipsas NV, Gamaletsou MN, Anastasopoulou A, Kontoyiannis DP. Therapy of mucormycosis. J Fungi (Basel) 2018;4(3):90.
Slonimsky G, Slonimsky E, Yakirevitch A, Sagiv D, Duvdevani S, Talmi YP, et al. The significance of computed tomography in invasive paranasal mucormycosis. Rhinology 2018;56(1):54-8.
John BV, Chamilos G, Kontoyiannis DP. Hyperbaric oxygen as an adjunctive treatment for zygomycosis. Clin Microbiol Infect 2005;11(7):515-7.
DOI: https://doi.org/10.21164/pomjlifesci.768
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