CC BY-NC-ND 4.0 · Int Arch Otorhinolaryngol 2019; 23(01): 092-100
DOI: 10.1055/s-0038-1667005
Original Research
Thieme Revinter Publicações Ltda Rio de Janeiro, Brazil

An Emergent Entity: Indolent Mucormycosis of the Paranasal Sinuses. A Multicenter Study

Erika Celis-Aguilar
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Alan Burgos-Páez
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Nadia Villanueva-Ramos
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
José Solórzano-Barrón
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Alma De La Mora-Fernández
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Juan Manjarrez-Velázquez
2   Department of Otolaryngology, Culiacan General Hospital, Culiacán, Sinaloa, México
,
Sergio Verdiales-Lugo
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Lucero Escobar-Aispuro
1   Department of Otolaryngology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS) de la Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Perla Becerril
3   Department of Otolaryngology, General Hospital Regional No. 1 “Ignacio García Téllez” del IMSS, Mérida, Yucatán, México
,
Ana Valdez-Flores
4   Department of Pathology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS), Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Francisco Javier Merino-Ramírez
4   Department of Pathology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS), Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
,
Carmen Beatriz Caballero-Rodríguez
4   Department of Pathology, Centro de Investigación y Docencia en Ciencias de la Salud (CIDOCS), Universidad Autónoma de Sinaloa, Culiacán, Sinaloa, México
› Author Affiliations
Further Information

Publication History

29 October 2017

23 May 2018

Publication Date:
24 October 2018 (online)

Abstract

Introduction Indolent or chronic mucormycosis is a rare entity that affects both immunosuppressed and immunocompetent individuals. Additionally, its clinical evolution is nonspecific and there is no standardized treatment for this condition.

Objective To describe the clinical characteristics and management of patients with indolent mucormycosis.

Methods In the project of study with chart review in the Interinstitutional secondary care centers, patients with evidence of indolent mucormycosis, defined as pathological confirmation of nasal/paranasal sinus mucormycosis for more than 1 month, were included. All patients underwent complete laboratory workup, imaging studies, surgical treatment and adequate follow-up. No evidence of disease status was defined when patient had subsequent biopsies with no evidence of mucormycosis.

Results We included seven patients, three female and four male subjects. The mean age was 53.14 years. Four patients were immunosuppressed and three immunocompetent. Among the immunosuppressed patients three had diabetes and one had dermatomyositis. The symptoms were nonspecific: facial pain/headache, mucoid discharge and cacosmia were the ones most frequently reported. Maxillary sinus involvement was present in all patients. Two immunosuppressed subjects received amphotericin. Posaconazole was the only treatment in one immunosuppressed patient. All immunocompetent patients had single paranasal sinus disease and received only surgical treatment. All patients are alive and free of disease.

Conclusion Indolent mucormycosis is a new and emerging clinical entity in immunosuppressed and immunocompetent patients. Single paranasal sinus disease is a frequent presentation and should not be overlooked as a differential diagnosis in these patients. Immunocompetent patients should only be treated surgically.

 
  • References

  • 1 Ibrahim AS, Kontoyiannis DP. Update on mucormycosis pathogenesis. Curr Opin Infect Dis 2013; 26 (06) 508-515
  • 2 Finn DG, Farmer Jr JC. Chronic mucormycosis. Laryngoscope 1982; 92 (7 Pt 1): 761-766
  • 3 Teixeira CA, Medeiros PB, Leushner P, Almeida F. Rhinocerebral mucormycosis: literature review apropos of a rare entity. BJM Case Rep 2013 Doi: 10.1136/bcr-2013-008552 https://www.ncbi.nlm.nih.gov/pubmed/23389725
  • 4 Marin-Mendez H. Monroy- Aguirre D, Rodríguez- Perales M. Caretta-Barradas Sergio. Síndrome de ápex orbitario causado por mucormicosis orbito cerebral crónica e indolente: reporte de dos casos. An Orl Mex 2005; 50: 64-68
  • 5 Tyson JC, Gittelman PD, Jacobs JB, Holliday R, Press R. Recurrent mucormycosis of the paranasal sinuses in an immunologically competent host. Otolaryngol Head Neck Surg 1992; 107 (01) 115-119
  • 6 Waizel-Haiat S, Cohn-Zurita F, Vargas-Aguayo AM, Ramírez-Aceves R, Vivar-Acevedo E. Mucormicosis rinocerebral invasora crónica. Cir Cir 2003; 71 (02) 145-149
  • 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 (02) e182-e184
  • 8 Dimaka K, Mallis A, Naxakis SS. , et al. Chronic rhinocerebral mucormycosis: a rare case report and review of the literature. Mycoses 2014; 57 (11) 699-702
  • 9 Gutiérrez-Delgado EM, Treviño-González JL, Montemayor-Alatorre A. , et al. Chronic rhino-orbito-cerebral mucormycosis: A case report and review of the literature. Ann Med Surg (Lond) 2016; 6: 87-91
  • 10 Virk RS, Arora P. Chronic sinonasal aspergillosis with associated mucormycosis. Ear Nose Throat J 2007; 86 (01) 22
  • 11 Jung H, Park SK. Indolent mucormycosis of the paranasal sinus in immunocompetent patients: are antifungal drugs needed?. J Laryngol Otol 2013; 127 (09) 872-875
  • 12 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: 617-620
  • 13 Wolkow N, Jakobiec FA, Stagner AM. , et al. Chronic orbital and calvarial fungal infection with Apophysomyces variabilis in an immunocompetent patient. Surv Ophthalmol 2017; 62 (01) 70-82
  • 14 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 (06) 2372-2375
  • 15 Mignogna MD, Fortuna G, Leuci S. , 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 (08) e533-e540
  • 16 Ruoppi P, Dietz A, Nikanne E, Seppa J, Markkanen H, Nuutinen J. Paranasal sinus mucormycosis: a report of two cases. Acta Otolaryngol 2001; 121 (08) 948-952
  • 17 Sun HY, Singh N. Mucormycosis: its contemporary face and management strategies. Lancet Infect Dis 2011; 11 (04) 301-311
  • 18 Rumboldt Z, Castillo M. Indolent intracranial mucormycosis: case report. AJNR Am J Neuroradiol 2002; 23 (06) 932-934
  • 19 Vignale R, Mackinnon JE, Casella de Vilaboa E, Burgoa F. Chronic, destructive, mucocutaneous phycomycosis in man. Sabouraudia 1964; 3 (02) 143-147
  • 20 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 (05) 1112-1118
  • 21 Harril WC, Stewart MG, Lee AG, Cernoch P. Chronic rhinocerebral mucormycosis. Laryngoscope 1996; 106 (10) 1292-1297
  • 22 Dooley DP, Hollsten DA, Grimes SR, Moss Jr J. Indolent orbital apex syndrome caused by occult mucormycosis. J Clin Neuroophthalmol 1992; 12 (04) 245-249
  • 23 Rao SP, Kumar KR, Rokade VR, Khanna V, Pal C. Orbital Apex Syndrome due to mucormycosis caused by Rhizopus microsporum. Indian J Otolaryngol Head Neck Surg 2006; 58 (01) 84-87
  • 24 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 (01) 91-98
  • 25 Ferstenfeld JE, Cohen SH, Rose HD, Rytel MW. Chronic rhinocerebral phycomycosis in association with diabetes. Postgrad Med J 1977; 53 (620) 337-342
  • 26 Bertin H. Mucormicosis rinosinusal. Rev Otorrinolaringol Cir Cabeza Cuello 2003; 63: 122-126
  • 27 Ketenci I, Unlü Y, Sentürk M, Tuncer E. Indolent mucormycosis of the sphenoid sinus. Otolaryngol Head Neck Surg 2005; 132 (02) 341-342
  • 28 Del Valle Zapico A, Rubio Suárez A, Mellado Encinas P, Morales Angulo C, Cabrera Pozuelo E. Mucormycosis of the sphenoid sinus in an otherwise healthy patient. Case report and literature review. J Laryngol Otol 1996; 110 (05) 471-473