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Table of Contents
CASE REPORT
Year : 2018  |  Volume : 15  |  Issue : 3  |  Page : 172-174

A rare case of isolated retro-orbital fungal granuloma


Department of ENT and Head and Neck Surgery, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth, Puducherry, India

Date of Web Publication10-Sep-2018

Correspondence Address:
Satvinder Singh Bakshi
Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth, Piliaiyarkuppam, Puducherry - 607 402
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/am.am_47_18

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  Abstract 


We report here the first case of retro-orbital fungal granuloma caused by Aspergillus versicolor. A 52-year-old male presented with protrusion of left eye associated with pain in the left eye and watering of the left eye for 6 months duration. Left eye examination showed axial proptosis, and fundus examination revealed blurring of the nasal disc margin. Diagnostic nasal endoscopy revealed an irregular pinkish proliferative mass medial to the middle turbinate. Plain computed tomography of the orbits showed a retrobulbar intraconal lesion in the left orbit. The patient was taken up for endoscopic orbital and optic nerve decompression with debulking or retro-orbital mass under general anesthesia. Culture on Sabouraud's dextrose agar grew Aspergillus species. The patient was started on oral itraconazole 100 mg twice daily, and over the course of 2 months, the left eye proptosis reduced significantly. Review at 2 years postoperatively did not reveal any recurrence of symptoms. Endoscopic clearance of the disease combined with oral antifungals is the appropriate modality of therapy for aspergillosis.

Keywords: Aspergillus versicolor, fungal, proptosis, retro-orbital granuloma


How to cite this article:
Kaipuzha RR, Pulimoottil DT, Bakshi SS, Gopalakrishnan S. A rare case of isolated retro-orbital fungal granuloma. Apollo Med 2018;15:172-4

How to cite this URL:
Kaipuzha RR, Pulimoottil DT, Bakshi SS, Gopalakrishnan S. A rare case of isolated retro-orbital fungal granuloma. Apollo Med [serial online] 2018 [cited 2019 Oct 22];15:172-4. Available from: http://www.apollomedicine.org/text.asp?2018/15/3/172/240942




  Introduction Top


Primary intraorbital fungal lesions are relatively rare and may masquerade as other entities. Aspergillosis belongs to the category of systemic mycoses. Though rare in immunocompetent individuals, there is an increased incidence of invasive aspergillosis in the immunocompromised patients, and its incidence has only increased over the past two decades.[1] Orbital fungal involvement is worrisome because of ready availability of pathways for further intracranial spread, such as superior orbital fissure, optic canal that directly opens into the middle cranial fossa.[2] Although fungal involvement of the orbit extending from the sinuses are quite frequent, a true orbital fungal granuloma is less so. Here, we report a case of isolated retro-orbital fungal granuloma involving a rare fungal species.


  Case Report Top


A 52-year-old male was referred to the Outpatient Department of Otorhinolaryngology of a tertiary care center with complaints of protrusion of the left eye associated with pain in the left eye and watering of the left eye for 6 months duration. The symptoms were slowly progressive; however, at the time of presentation, the patient complained of electric shock-like pain in the eye. He had no comorbidities and had a no history of trauma or surgery or any previous treatment for the condition. Examination of the left eye showed axial proptosis [Figure 1]a, with no pulsations and no increase with Valsalva maneuver. The conjunctiva, cornea, anterior chamber, iris, pupil, and lens were all normal. The orbital margins were free with no obvious palpable mass. Fundus examination revealed blurring of the nasal disc margin. Extraocular movements were unaffected and visual acuity was normal. Examination of the nose and paranasal sinuses was normal, and no obvious mass was detected on anterior rhinoscopy. Diagnostic nasal endoscopy revealed an irregular pinkish proliferative mass medial to the middle turbinate [Figure 2]a. Plain computed tomography of the orbits (axial view) was done and showed a retrobulbar intraconal lesion in the left orbit, probably arising from the inferior rectus muscle, which extended into the posterior aspect of the nasal cavity through the medial wall [Figure 2]b. Under local anesthesia, a biopsy was taken from the nasal mass and histopathology revealed features of chronic nonspecific inflammation. Considering a possible diagnosis of pseudotumor, the patient was started on oral methylprednisolone, but there was no improvement in signs even after 2 weeks of treatment. In view of nonimprovement of symptoms, the patient was taken up for endoscopic exploration under general anesthesia. Left uncinectomy, anterior and posterior ethmoidectomy, and sphenoidotomy were done. Following this, optic nerve decompression was done and the intraorbital mass was carefully debulked. Gram staining and Ziehl–Neelsen staining were negative for organisms and KOH mount did not reveal any fungal elements. Culture on Sabouraud's dextrose agar grew Aspergillus species. Slide culture revealed reduced Penicillium-like structures [Figure 2]c and [Figure 2]d with fully covered vesicles and the presence of Hülle [Figure 2]e cells, features consistent with Aspergillus versicolor, a rare species to be found intraorbitally. The postoperative period was uneventful and the patient was started on oral itraconazole 100 mg twice daily, and over the course of 2 months, the left eye proptosis reduced significantly [Figure 1]b. Review at 8 months postoperatively did not reveal any recurrence of symptoms.
Figure 1: The clinical presentation of the patient; (a) the initial presentation with left eye axial proptosis; (b) the postoperative appearance where the proptosis has resolved

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Figure 2: The various investigations done; (a) Diagnostic nasal endoscopy image showing pinkish proliferative mass medial to the middle turbinate (black arrow); (b) Plain computed tomography orbits (axial view) showing a retrobulbar intraconal lesion in the left orbit; (c) Slide culture showing greenish colonies of Aspergillus versicolor; (d) Light microscopy of the cultured fungal colonies showing reduced Penicillium-like structures (black arrow) with fully covered vesicles (white arrow); (e) Higher magnification showing the presence of Hülle cells

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  Discussion Top


Aspergillus species are ubiquitous, commonly found in the soil and decaying vegetation. The spores are typically inhaled or ingested without consequence in the normal host and exposure to this fungus is frequent, yet disease due to tissue invasion is uncommon in the immunocompetent host.[3] The portals of entry for Aspergillus include the respiratory tract, damaged skin or other operative wounds, the cornea, and the ear.[4] An intact immune system can prevent the disease in a healthy individual, and the mechanism of an isolated retro-orbital fungal infection in an immunocompetent individual is quite mysterious.

Aspergillosis is the second most frequently seen fungal infection of the face and mouth in patients receiving chemotherapy. It is second to Candida in its frequency. Aspergillosis of the head and neck region primarily affects the nasal cavity and paranasal sinuses. Aspergillus as a pathogen cannot actively penetrate undamaged and intact mucus membrane or skin as it lacks keratolytic enzymes.[5]

Rarely has invasive Aspergillus infection been described in immunocompetent patients. Orbital involvement in invasive aspergillosis is known to occur by the contiguous spread of the disease from paranasal sinuses either by expansion or bone erosion due to pressure effect of the polyps or by fungal tissue invasion. Early diagnosis of intraorbital fungal infection remains a challenge. The presentation of orbital aspergillosis can mimic diseases such as mucormycosis, neoplasms, vascular etiology, and neuro-ophthalmic diseases.[2]

A. versicolor is a cosmopolitan fungus often found in damp indoor environments. It commonly causes onychomycosis, otomycosis, cutaneous disease, osteomyelitis, and pulmonary infections. There are rare reports of endophthalmitis and epicorneal aspergilloma caused by this organism.[6] However, there are no reports of retro-orbital fungal granuloma caused by A. versicolor.

Colonies of A. versicolor are variously colored (versi-colored) and range from very pale green to greenish-beige, pinkish-green, or dark green. The fungal hyphae are septate and hyaline and the vesicles are small, with metulae and phialides covering most of the vesicle. Reduced conidial structures resembling those seen in Penicillium species are common and globose Hülle cells may also be present.[7]

Intravenously administered conventional amphotericin B is often used for the treatment of infections by A. versicolor. However, conventional amphotericin B has reduced bioavailability; therefore, intravenous liposomal amphotericin B and intravitreal amphotericin B are preferred.[8] Recently, A. versicolor has been shown to be resistant to amphotericin B and there have also been reports of treatment failure.[9] Alternatively, oral itraconazole has good activity against A. versicolor, but its effectiveness is limited by its reduced bioavailability. Voriconazole, a second-generation triazole, has very good bioavailability, is active against A. versicolor and other Aspergillus species, and is currently the drug of choice for the treatment of invasive aspergillosis.[1]


  Conclusion Top


Successful treatment of aspergillosis requires prompt diagnosis and rapid institution of therapy because delay or nonaggressive therapy can result in the spread of infection with lethal consequences. Radiological imaging is an extremely important diagnostic modality in these types of lesions in both making a diagnosis and in directing treatment. Endoscopic clearance of the disease combined with oral antifungals appears to be the appropriate modality of therapy.

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.
Sivak-Callcott JA, Livesley N, Nugent RA, Rasmussen SL, Saeed P, Rootman J, et al. Localised invasive sino-orbital aspergillosis: Characteristic features. Br J Ophthalmol 2004;88:681-7.  Back to cited text no. 1
    
2.
Neeli AS, Kotrashetti SM, Vallavan P. Sino-orbital aspergillosis: A case report and brief review of literature. World J Dent 2012;3:363-6.  Back to cited text no. 2
    
3.
Fuqua TH Jr., Sittitavornwong S, Knoll M, Said-Al-Naief N. Primary invasive oral aspergillosis: An updated literature review. J Oral Maxillofac Surg 2010;68:2557-63.  Back to cited text no. 3
    
4.
Denning DW. Invasive aspergillosis. Clin Infect Dis 1998;26:781-805.  Back to cited text no. 4
    
5.
Lin SJ, Schranz J, Teutsch SM. Aspergillosis case-fatality rate: Systematic review of the literature. Clin Infect Dis 2001;32:358-66.  Back to cited text no. 5
    
6.
Peterson SW. Phylogenetic relationships is Aspergillus based on rDNA sequence analysis. In: Samson RA, Pitt JI, editors. Integration of Modern Taxonomic Methods for Penicillium and Aspergillus Classification. Amsterdam, the Netherlands: Harwood Scientific Publishers; 2000. p. 323-55.  Back to cited text no. 6
    
7.
Richardson MD. Aspergillus and Penicillium species. In: Ajello L, Hay R, editors. Topley and Wilson's Microbiology and Microbial Infections. 9th ed. Vol. 4. London, England: Edward Arnold; 1998. p. 281-312.  Back to cited text no. 7
    
8.
Liu, Z. T. Hou, Q. Shen, W. Liao, and H. Xu1 995. Osteomyelitis of sacral spine caused by Aspergillus versicolor with neurologic deficits. Chin Med J (Engl). 108:472-5.  Back to cited text no. 8
    
9.
Naik MN, Vemuganti GK, Honavar SG. Primary orbital aspergilloma of the exenterated orbit in an immunocompromized patient. Indian J Med Microbiol 2006;24:233-4.  Back to cited text no. 9
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