Print this page Email this page
Users Online: 3018
Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 


 
 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 9  |  Issue : 1  |  Page : 16-20

Computed tomographic findings of sinonasal granulomatous mucormycosis in an immunocompetent patient in northwest Nigeria


1 Department of Radiology, Kaduna State University/BarauDikko Teaching Hospital, Kaduna, North-West Nigeria
2 National Ear Care Centre, Kaduna, North-West Nigeria

Date of Submission24-Jan-2020
Date of Acceptance12-Feb-2020
Date of Web Publication16-Apr-2020

Correspondence Address:
Dr. Tokan S Baduku
Department of Radiology, Kaduna State University/BarauDikko Teaching Hospital, Kaduna
North-West Nigeria
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ais.ais_4_20

Rights and Permissions
  Abstract 


Sinonasal phycomycosis or zygomycosis is an opportunistic infection caused by mold fungi belonging to the class Zygomycetes. These fungi are widely distributed in nature and mainly infect immunosuppressed patients. Herein, we present the rare case of sinonasal phycomycosis in an immunocompetent 37-year old man presented to an ENT surgeon with a history of rhinorrhea, nasal blockage, and progressive swelling of the cheek for 5 years, prior to presentation to hospital. A sinonasal soft tissue mass was observed with biopsy results yielding phycomycosis. Surgery was planned twice but was suspended by the patient himself. He finally absconded following a third appointment for surgery due to financial constraints. The purpose of this paper is to draw the attention of clinicians to strongly consider the inclusion of fungal infection as a differential diagnosis of orofacial or cheek swelling.

Keywords: Cheek swelling, chronic sinusitis, granulomatous mass, sinonasal phycomycosis


How to cite this article:
Baduku TS, Isah FB. Computed tomographic findings of sinonasal granulomatous mucormycosis in an immunocompetent patient in northwest Nigeria. Arch Int Surg 2019;9:16-20

How to cite this URL:
Baduku TS, Isah FB. Computed tomographic findings of sinonasal granulomatous mucormycosis in an immunocompetent patient in northwest Nigeria. Arch Int Surg [serial online] 2019 [cited 2020 Aug 4];9:16-20. Available from: http://www.archintsurg.org/text.asp?2019/9/1/16/282580




  Introduction Top


Tumors of the nose and paranasal sinuses are rare, accounting for less than 1% of all tumors.[1] Similarly, chronic fungal infection of the nose and paranasal sinuses are relatively rare with very few cases being reported from Nigeria.[1],[2] Rarer still is phycomycosis which was found in 0.7% of autopsies in patients with sinonasal fungal infections and makes up about 0.02% of hospital admissions in the United States of America.[3]

Fungal infection of the paranasal sinuses commonly affects the maxillary antra[4] with high mortality rates.[5] The common species involved are either aspergillus or other fungi including phycomycosis also called mucormycosis or zygomycosis.[6] Mucormycosis is naturally widely distributed in the environment.[7] However, it is mostly found in the soil and on decaying vegetation where it has the ability to grow rapidly and release large numbers of spores that become airborne and gain entrance to the human body through inhalation or ingestion.[8] Given its ubiquitous nature, individuals are exposed on a regular basis.[2] Though fungal infection is mostly seen among immunocompromised individuals,[5] it can occur infrequently in apparently healthy immunocompetent individuals, and this may pose a diagnostic challenge and therapeutic dilemma for the clinicians who are unfamiliar with its clinical presentation.[9]

We present a case of progressive nasal blockage and facial swelling in an immunocompetent adult which was ultimately confirmed by histology.


  Case Report Top


A 37-year-old trader who first presented with a 5-year history of progressive bilateral nasal obstruction and discharge. There was a history of nasal itching, sneezing, hyponasal speech, and anosmia prior to these symptoms. He also had a nonprogressive protrusion of the right eye which was not associated with loss of vision, diplopia, or epiphora. Although physical examination showed axial proptosis of the right eyeball, normal visual acuity was observed. There was no ophthalmoplegia. An assessment of the nasal cavities showed bilateral blockage by mobile grape-like glistering masses, soft palatal depression, and a granular posterior pharyngeal wall. There was splaying of the left nasal bridge. Anterior rhinoscopy showed a pale polypoidal mass in the left nasal cavity. The other surrounding regions showed no abnormality. He had a rigid nasal endoscopy which showed a polypoid mass with greenish cheesy secretions filling both nasal cavities. An advanced telescope could not view the nasopharynx hence a biopsy was taken for histology. The histologic results of both nasal and pharyngeal specimens yielded granulomatous mucormycosis while other laboratory investigations including blood sugar and immunologic tests were normal.

Computed tomography of the paranasal sinuses showed an extensive mild contrast-enhancing hyperdense lesion in the postnasal space and the right maxillary antrum, with the extension of the soft tissue mass to the nasal cavity. There was also bowing and thinning of the bony walls [Figure 1]a,[Figure 1]b,[Figure 1]c. There was erosion of the right medial wall and bowing of the posterior wall of the ipsilateral maxillary antrum. There was also an extension of the mass into the hypopharynx [Figure 2]. The maxillary and nasal bones were eroded showing as asymmetry in the 3-D image [Figure 3]. However, the skull base was not eroded.


Click here to view
Figure 2: Longitudinal slice showing soft tissue filling of the right sinonasal spaces and thinning of the palate

Click here to view
Figure 3: A 3-D image of the facial bone frontally, showing erosion and asymmetry of the nasal and maxillary bones

Click here to view


He was booked for endoscopic assisted polypectomy. In the course of the preoperative evaluation, he was found to have persistently elevated blood pressure which warranted physician and psychologic assessment prior to surgery but the patient decided not to undergo the surgery. He reappeared 2 years later with the same complaints of intermittent nasal blockage, bilateral rhinorrhea, and left epiphora. He was not convinced about the surgery and therefore disappeared for the second time. He represented for the third time with left-sided nasal blockage. A repeat CT scan of the paranasal sinuses showed the same images as in the first CT scan [Figure 1], [Figure 2], [Figure 3]. However, while amphotericin B was planned to be started and the patient was being prepared to have surgery done, he absconded for the third time and had not reported for 1 year till now.


  Discussion Top


Mycotic infection of the nose and paranasal sinuses is less than 0.2% in immunocompromised people,[8],[10] and rarer still in immunocompetent persons.[3] This can be caused by various fungi, namely, Aspergillus, Phycomycetes, Candida, and Rhinosporidium species.[9] These organisms are mostly found in the tropical forests of Africa and Asia with few cases being reported from the arid zones of the developed continents.[1],[2] The commonest fungal infection of the sinonasal cavities is caused by Aspergillus species.[1],[2],[11] However, phycomycosis, which is a rarer fungal infective agent, is caused by a species called the Phycomycetes.[3],[8] This species is common in the soil but is not usually pathogenic except in the immunocompromised.[9] Contact with this category of patients is characterized by high morbidity and mortality because the disease is more invasive.[3],[12]

Although fungal infection of the nose and sinuses may occur in healthy people, it is usually protracted in diabetics,[5],[13] immunocompromised, and debilitated patients[9] who are on extensive anti-suppressive drugs and prolonged antibiotic use, resulting in suppression of the normal bacterial flora of the nose with consequent infection and debilitating diseases.[14] Though mucormycosisis is rare when compared to Aspergillus and Candida, it is said to be more invasive,[7] and about 70% of the cases occur in patients with diabetes mellitus and renal patients who are on immunosuppressive drugs.[9],[13] The patient under consideration had no debilitating disease and had no history of drug abuse. The sugar and immunologic laboratory investigations revealed normal findings.

Invasive phycomycosis is rare, with an incidence ranging between 0.2 and 1.9% in immunocompromised persons, and rarer still in the immunocompetent individuals with an incidence of 0.00017 to 0.00042%.[10] Multiple sinuses are typically involved but it might be limited to only one sinus, later spreading to the opposite side, depending on the duration of the disease and the invasive rate.[9] The granulomatous invasive type of sinus phycomycosis is usually seen in patients from Sudan, India, Pakistan, and Saudi Arabia who are less developed.[7] Phycomycosis was first reported in Nigeria in two patients in Ibadan by Martinson in 1960.[15] These patients had normal immune status with a relatively slow time course of the disease. Generally, patients present with an enlarging mass in the cheek, orbit, nose, or sinuses.[12]

The clinical features include nasal obstruction, rhinorrhea, epistasis, proptosis, and facial swelling. Various clinical forms of fungal infection of the nose have been described as allergic, noninvasive, and invasive or fulminant.[16] More than one form of the disease can be described in many individuals. Our patient had the granulomatous type of mucormycosis which was histologically proven. Mucosal swelling and sinus obstruction are implicated as a predisposing factor in fungal infection of the nose.[17] Our patient initially had symptoms of allergic rhinosinusitis prior to the cheek swelling.

Imaging has classically been used to evaluate fungal sinusitis in support of the clinical suspicion of invasive disease.[11] Computed tomography (CT) and magnetic resonance imaging (MRI) are highly accurate and noninvasive modalities for the assessment and extent of this lesion.[4],[18] However, the latter is more useful in the assessment of suspected extension beyond the sinonasal cavities such as the orbits and brain.[11] Radiographically, aspergillosis shows radiological concretions whereas opacification is seen in the case of mucormycosis[19] as also being seen in the present case. Bone destruction, as well as the involvement of contiguous structures such as the orbit and brain, have been prime determinants of aggressive disease.[18] These findings are useful in any surgical planning. Our patient's lesion did not extend beyond the paranasal sinuses, except for the destruction of the bony borders.

Fungal tumors can either be benign or malignant and may vary in location, sizes, and types.[20] Care for these tumors depend on individual patients, and the management may involve a multidisciplinary approach.[21] Possible treatments include surgery, radiation therapy, and chemotherapy depending on the particular tumor and the needs of the patient.[1],[2] However, early diagnosis is very important for the effective management of the patient's condition.[21]

In most cases, the prognosis of mucormycosis is poor and has varied mortality rates depending on its form and severity.[3] Early recognition of the disease and treatment of the underlying cause of mucormycosis such as diabetes are keys for improving outcomes.[7] The antifungal treatment of choice for mucormycosis is amphotericin B.[21] Very high doses are required because of the relative resistance of the fungus to the drug.[22] Our patient was to start treatment with amphotericin B prior to the surgery, to be followed with postoperative doses but for the abscondment.[23]


  Conclusion Top


The clinical diagnosis of fungal infection of the nose poses a serious challenge since the features are not clear-cut and the course of the disease is insidious. A high index of suspicion is required to make an early diagnosis of the condition and to come out with a definitive diagnosis of the species involved. Patients on treatment for chronic allergic rhinosinusitis may benefit from mycotic studies, especially when on steroids. The invasive form of fungal disease of the nose and sinuses, as seen in this case may mimic a malignant condition. Ignorance and poverty, religious beliefs, and cosmetic after-effect of the surgery are still very major factors in patient management in the tropics.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Oyewole EA, Agboola AOJ, Banjo AAF, Deji-Agboola AM. Fungal granuloma of the nose in an African male: A case report. J Rural Trop Public Health 2006;5:46-8.  Back to cited text no. 1
    
2.
Bakari A, Shuaibu IY, Usman MA, Mayun A, Ahmed SA. Rhinoentomophthoromycosis in Northern Nigeria: Report of two cases. Sub-Saharan Afr J Med 2016;3:118-21.  Back to cited text no. 2
  [Full text]  
3.
Roden MM, Zaoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL, et al. Epidemiology and outcome of zygomycosis: A review of 929 reported cases. Clin Infect Dis 2005;41:634-53.  Back to cited text no. 3
    
4.
Dhong H, Lanza DC. Fungal rhinosinusitis. In: Kennedy DW, Bolger WE, Zinreich SJ, editors. Diseases of the Sinuses: Diagnosis and Management. Hamilton, ON: BC Decker; 2001. p. 187-93.  Back to cited text no. 4
    
5.
Gillespie MB, O'Malley BW. An algorithmic approach to the diagnosis and management of invasive fungal rhinosinusitis in the immunocompromised patient. Otolaryngol Clin North Am 2000;33:323-34.  Back to cited text no. 5
    
6.
Hedayati MT, Pasqualotto AC, Warn PA, Bowyer P, Denning DW. Aspergillusflavus: Human pathogen, allergen and mycotoxin producer. Microbiology 2007;153:1677-92.  Back to cited text no. 6
    
7.
Cornely OA, Arikan-Akdagli S, Dannaoui E, Groll AH, Lagrou K, Chakrabarti A, et al. ESCMID and ECMM joint clinical guidelines for the diagnosis and management of mucormycosis 2013. Clin Microbiol Infect 2014;20(Suppl 3):5-26.  Back to cited text no. 7
    
8.
Antachopoulos C, Walsh TJ, Roilides E. Fungal infections in primary immunodeficiencies. Eur J Pead 2007;166:1099-117.  Back to cited text no. 8
    
9.
Soni A. Paranasal mucormycosis in an immunocompetent individual: Importance of early diagnosis. Arch Intern Med 1978;138:1274-6.  Back to cited text no. 9
    
10.
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;15:533-40.  Back to cited text no. 10
    
11.
Thotan SP, Kumar V, Gupta A, Mallya A, Rao S. Subcutaneous phycomycosis—fungal infection mimicking a soft tissue tumor: A case report and review of literature. J Trop Pediatr 2010;56:65-6.  Back to cited text no. 11
    
12.
Sahoo NK, Bhandari AK, Kumar A. Mucormycosis of the frontal sinus: A rare case report and review. Ann Maxillofac Surg 2017;7:120-3.  Back to cited text no. 12
[PUBMED]  [Full text]  
13.
Asman S, Behnia MM. Rhinocerebral mucormycosis: A rare fungal infection linked to diabetes. J Am Acad Physician Assist 2011;24:E1-4.  Back to cited text no. 13
    
14.
Lasisi OA, Nwarogu OG, Akang EU. Sinus mycosis-A case of persistent chronic sinusitis (Case report). Nigerian J Surg 1997;4:77-80.  Back to cited text no. 14
    
15.
Martinson FD. Clinical epiodemological and therapeutic aspects of entomorphthoromycosis. Ann Soc Belge Med Trop 1972;52:329-42.  Back to cited text no. 15
    
16.
Soni A. Paranasal mucormycosis in an immunocompetent individual: Importance of early diagnosis. Int J Oral Health Med Res 2017;4:52-6.  Back to cited text no. 16
    
17.
Mackay IS, Bull TR. Rhinology. In: Kerr AG, editor. Scott-Brown's Otolaryngology. Vol 4. Oxford: Butterworth Heinemann; 1997. p. 39-49.  Back to cited text no. 17
    
18.
Silverman CS, Mancuso AA. Periantral soft-tissue infiltration and its relevance to the early detection of invasive fungal sinusitis: CT and MR findings. Am J Neuroradiol 1998;19:321-5.  Back to cited text no. 18
    
19.
Rao VM, El-Noveam KI. Sinonasal imaging: Anatomy and pathology. Radiol Clin North Am 1998;36:921-39.  Back to cited text no. 19
    
20.
Bassiouny A, Maher A, Bucc TJ, Moawad MK, Hendawy DS. Non-invasive antromycosis (Diagnosis and treament). J Laryn Otology 1982;96:215-28.  Back to cited text no. 20
    
21.
Chaturvedi J, Sachdeva R, Singh P, Savio J, Divya P, Balasubramanya AM. Mucormycosis of the paranasal sinus with gas forming maxillary osteomyelitis—A case report. Internet J Microbiol 2012;10:1-6.  Back to cited text no. 21
    
22.
Guevara N, Roy D, Dutruc-Rosset C, Santini J, Hofman P, Castillo L. Mucormycosis-early diagnosis and treatment. Rev Laryngol Otol Rhinol (Bord) 2004;125:127-31.  Back to cited text no. 22
    
23.
Gupta AK, Mann SBS, Khosla VK, Sastry KV, Hundal JS. Non-randomized comparison of surgical modalities for paranasal sinus mycoses with intracranial extension. Mycoses 1999;42:225-30.  Back to cited text no. 23
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
 
  Search
 
Similar in PUBMED
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed1385    
    Printed64    
    Emailed0    
    PDF Downloaded101    
    Comments [Add]    

Recommend this journal