Rhinocerebral mucormycosis: A literature review and report of clinical case
DOI:
https://doi.org/10.47464/MetroCiencia/vol28/2/2020/38-47Keywords:
Mucormicosis, B-cell Acute Lymphoblastic Leukemia, immune system, angioinvasion, liposomal Amphotericin BAbstract
Mucormycosis is an uncommon opportunistic fungal infection caused by fungi of the mucoral order. Occurs at an annual rate of 1.7 cases per million people. It has a high mortality rate ranging from 30 to 90% according to the patient's systemic status. The most complex scenarios are observed in immunosuppressed patients, whereas, in competent patients, the fungal invasion is well controlled by the host's immune system. The infection begins after exposure, inhalation and invasion of spores into the oral and nasal cavity from where it can spread to other parts of the body, allowing different clinical presentations in susceptible patients. Currently, international records of mortality of mucormycosis in children with neoplasms range from 41.3 to 66.6. The objective of this study is to present a rare case of rhinocerebral mucormycosis in a 4-year-old male patient who also presented as a base disease a leukemia acute B-lymphoblastic disease. Our patient was treated with liposomal amphotericin B and surgical debridement of affected areas. In addition, a systematic review of the available literature was carried out with the aim of determining and describing the signs, symptoms, diagnosis, available treatment and prognosis of this disease.
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Cohen A, Shoukair F, Koren M, Shaulov A & Casap N. Successful Mandibular Mucormycosis Treatment in the Severe Neutropenic Patient. Journal of Oral and Maxillofacial Surgery; 2019. https://doi.org/10.1016/j.joms.2019.02.012
Singh VP, Bansal C, Kaintura M. Sinonasal Mucormycosis: A to Z. Indian J Otolaryngol Head Neck Surg. 2019;71(Suppl 3):1962-1971. https://doi.org/10.1007/s12070-018-1384-6
Jundt JS, Wong MEK, Tatara AM, Demian NM. Invasive Cutaneous Facial Mucormycosis in a Trauma Patient. J Oral Maxillofac Surg. 2018;76(9):1930.e1-1930.e5. https://doi.org/10.1016/j.joms.2018.01.022
Singh V, Sharma B, Sen R, Agrawal S, Bhagol A, & Bali R. Rhinocerebral Mucormycosis: A Diagnostic Challenge and Therapeutic Dilemma in Immunocompetent Host. Journal of Oral and Maxillofacial Surgery; 2012;70(6), 1369–1375. https://doi.org/10.1016/j.joms.2011.06.209
Pajpani M, & Webb R. Lingual necrosis caused by mucormycosis in a patient with aplastic anaemia: case report. British Journal of Oral and Maxillofacial Surgery; 2014; 52(10), e144–e146. https://doi.org/10.1016/j.bjoms.2014.09.012
Dogan C, Leblebisatan G, Haytac, C, Antmen B, & Surmegozler O. Oral mucormycosis in children with leukemia: report of 2 cases. Quintessence international; 2007; 38 6, 515-20. Disponible en: https://pubmed.ncbi.nlm.nih.gov/17625636/
Bonifaz A, Macias B, Paredes-Farrera F, Arias P, Ponce R, & Araiza J. Palatal zygomycosis: experience of 21 cases. Oral Diseases; 2008;14(6), 569–574. https://doi.org/10.1111/j.1601-0825.2007.01433.x
O’Neill M, Alessi S, George B, & Piro J. Disseminated Rhinocerebral Mucormycosis: A Case Report and Review of the Literature. Journal of Oral and Maxillofacial Surgery; 2006; 64(2), 326–333. https://doi.org/10.1016/j.joms.2005.10.017
Muggeo P, Calore E, Decembrino N, et al. Invasive mucormycosis in children with cancer: A retrospective study from the Infection Working Group of Italian Pediatric Hematology Oncology Association. Mycoses. 2019;62(2):165-170. https://doi.org/10.1111/myc.12862
Soni A. Paranasal Mucormycosis in an Immunocompetent Individual: Importance of Early Diagnosis. Int J Oral Health Med Res 2017;4(3):52-56.
Klimko N, Khostelidi S, Shadrivova O, Volkova A, Popova M, Uspenskaya O, Afanasyev B. Contrasts between mucormycosis and aspergillosis in oncohematological patients. Medical Mycology; 2019; 57(Supplement_2), S138–S144.
Cornu M, Bruno B, Loridant S, Navarin P, François N, Lanternier F, Sendid B. Successful outcome of disseminated mucormycosis in a 3-year-old child suffering from acute leukaemia: the role of isavuconazole? A case report. BMC Pharmacology and Toxicology; 2018; 19(1). https://dx.doi.org/10.1186/s40360-018-0273-7
Bellanger P, Berceanu A, Rocchi S, Valot B, Fontan J, Chauchet A, Millon L. Development of a quantitative PCR detecting Cunninghamella bertholletiae to help in diagnosing this rare and aggressive mucormycosis. Bone Marrow Transplantation; 2018. https://doi.org/10.1038/s41409-018-0194-5
Vaughan C, Bartolo A, Vallabh N, & Leong C. A meta-analysis of survival factors in rhino-orbital-cerebral mucormycosis-has anything changed in the past 20 years? Clinical Otolaryngology; 2018. https://doi.org/10.1111/coa.13175
Debureaux PE, Paccoud O, Guitard J, et al. Rhino-orbital Mucormycosis presenting as facial cellulitis in a patient with high-risk acute myeloid leukemia in relapse. Curr Res Transl Med. 2019;67(2):76-78. https://doi.org/10.1016/j.retram.2019.01.004
Sipsas NV, Gamaletsou MN, Anastasopoulou A, Kontoyiannis DP. Therapy of Mucormycosis. J Fungi (Basel). 2018 Jul 31;4(3):90. https://doi.org/10.3390/jof4030090
Jeong W, Keighley C, Wolfe R, et al. The epidemiology and clinical manifestations of mucormycosis: a systematic review and meta-analysis of case reports. Clin Microbiol Infect. 2019;25(1):26-34. https://doi.org/10.1016/j.cmi.2018.07.011
Roden M, Zaoutis E, Buchanan L, Knudsen A, Sarkisova A, Schaufele L, Walsh J. Epidemiology and Outcome of Zygomycosis: A Review of 929 Reported Cases. Clinical Infectious Diseases; 2005; 41(5), 634–653. https://doi.org/10.1086/432579
Grossi O, Pineau S, Sadot-Lebouvier S, Hay B, Delaunay J, Miailhe F, Le Pape P. Disseminated mucormycosis due to Lichtheimia corymbifera during ibrutinib treatment for relapsed chronic lymphocytic leukaemia: a case report. Clinical Microbiology and Infection; 2018. https://doi.org/10.1016/j.cmi.2018.10.004
Lien Y, Chou H, Lin C, Bai Y, Chiu F, Yeh P. Epidemiology and risk factors for invasive fungal infections during induction chemotherapy for newly diagnosed acute myeloid leukemia: A retrospective cohort study; 2018; 13(6): e0197851. https://doi.org/10.1371/journal.pone.0197851
Cheema SA, Amin F. Five cases of rhinocerebral mucormycosis. Br J Oral Maxillofac Surg. 2007;45(2):161-162. https://doi.org/10.1016/j.bjoms.2005.06.021
Petrikkos G, & Tsioutis C. Recent Advances in the Pathogenesis of Mucormycoses. Clinical Therapeutics; 2018; 40(6), 894–902. https://doi.org/10.1016/j.clinthera.2018.03.009
Ergun O, Tahir E, Kuscu O, Ozgen B, & Yilmaz T. Acute Invasive Fungal Rhinosinusitis: Presentation of 19 Cases, Review of the Literature, and a New Classification System. Journal of Oral and Maxillofacial Surgery; 2017; 75(4), 767.e1–767.e9. https://doi.org/10.1016/j.joms.2016.11.004
Klein-Awarjanow K, Nowicki K, Bieniaszewska M, & Szurowska E. Common radiological findings in fungal infections in hematological patients – Review. Polish Annals of Medicine; 2017; 24(2), 287–294. https://doi.org/10.1016/j.poamed.2016.12.001
Marx R & Stern D. Oral and Maxillofacial Pathology: a rationale for diagnosis and treatment. Second edition; 2012; 110 - 112.
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Copyright (c) 2020 Marco Andrés Cedillo Bailón, Andrea Pesántez, Edison René Díaz Mora, Fernando Sandoval Portilla
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