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 Table of Contents  
CASE REPORT: MYCOLOGY
Year : 2013  |  Volume : 15  |  Issue : 2  |  Page : 75-78

Invasive sinonasal Scopulariopsis mimicking mucormycosis


Department of Microbiology, Government Medical College, Calicut, Kerala, India

Date of Web Publication7-Jan-2014

Correspondence Address:
Mini P Narayanan
Department of Microbiology, Government Medical College, Calicut, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1282.124594

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  Abstract 

We report a case of invasive sinonasal Scopulariopsis that mimicked mucormycosis in a 63-year-old diabetic patient. The isolated fungus from functional endoscopic sinus surgery (FESS) specimen was identified as Scopulariopsis brevicaulis. The patient was treated with Amphotericin B followed by Itraconazole. Aetiological identification of the fungus is very essential for preventing recurrence.

Keywords: Amphotericin B, Itraconazole, mucormycosis, Scopulariopsis brevicaulis, sinonasal Scopulariopsis


How to cite this article:
Narayanan MP, Kuttiyil A, Orvankundil S, Surendran RD. Invasive sinonasal Scopulariopsis mimicking mucormycosis. J Acad Clin Microbiol 2013;15:75-8

How to cite this URL:
Narayanan MP, Kuttiyil A, Orvankundil S, Surendran RD. Invasive sinonasal Scopulariopsis mimicking mucormycosis. J Acad Clin Microbiol [serial online] 2013 [cited 2017 May 26];15:75-8. Available from: http://www.jacmjournal.org/text.asp?2013/15/2/75/124594


  Introduction Top


Over the past decade, an increasing number of opportunistic fungal infections have been reported in immunocompromised patients. Fungi of low virulence or environmental fungi may be a potential pathogen in various types of immunocompromised patients. The common opportunistic fungi isolated from immunocompromised patients include Candida, Cryptococcus, Aspergillus, Chrysosporium, and Penicillium species. [1] With the exception of Candida species which is endogenous, the great majority of opportunistic fungi are moulds which are environmental contaminants of soil origin.

Recently, an increasing number of cases, especially immunocompromised patients, have been reported with invasive sinonasal fungal infections. Besides the common fungal agents, there are a few reports of unusual fungal agents that can also cause sinonasal infections. Here, we describe a case of invasive sinonasal infection with Scopulariopsis in a 63-year-old diabetic patient with ptosis and defective vision. The patient underwent functional endoscopic sinus surgery (FESS) and was treated with Amphotericin B and Itraconazole.


  Case Report Top


A 63-year-old lady admitted in Government Medical College, Kozhikode, presented with right-sided headache and defective vision of 1 week duration. She also had drooping of eyelids for 3 days and two episodes of vomiting. She was a known diabetic and hypertensive for the past 1 year and was on regular treatment. There was no history of fever, weakness or ear discharge.

On examination, the patient was conscious and oriented. Pulse rate was 76 per minute and blood pressure was 160/100.

Examination of the central nervous system showed the patient had ptosis and diplopia of the right eye with third, fourth, fifth and sixth cranial nerve palsy. Other systems were within normal limits.

Ocular examination revealed the following:

Right eye: Pupillary reaction was sluggish, there was only slight lateral and medial movement of the eye ball with complete ptosis, cornea was clear, sensation was decreased and counting fingers was possible close to the face. Left eye: Normal, except for vision which was only counting fingers at 5 m.

Laboratory studies of blood gave the following results:

Haemoglobin (Hb) 14.38 g/dl, total count 10,800 cells/cc, with a differential of neutrophils (N) 50% and lymphocytes (L) 32%

Platelet count 3.07 lakhs

Erythrocyte sedimentation rate (ESR) 50 mm during first hour,

Fasting blood glucose level 149 mg/dl, postprandial blood sugar (PPBS) 334 mg/dl

Mantoux test was negative

Serum was negative for antibodies to Human Immunodeficiency Virus (HIV) by standard methods.

CT scan of head showed no evidence of intracranial pathology. An inflammatory lesion with air fluid levels was noted in the left maxillary sinus. An almost similar dense lesion was noticed in the sphenoid sinus also. Inflammatory thickening was seen in the ethmoid and frontal sinuses on both sides and in the right maxillary sinus [Figure 1]. Magnetic resonance imaging (MRI) brain showed significant inflammatory mucosal disease in the right sphenoid sinus resulting in mild lateral bulge into right cavernous sinus corresponding to the course of right third, fourth, fifth and sixth cranial nerves.
Figure 1: CT scan - Inflammatory lesion with a fluid level in the left maxillary sinus

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Patient was posted for direct nasal endoscopy for biopsy from the right sphenoid sinus. After the FESS, the specimens were sent for histopathological examination and for fungal culture. Histopathological report showed fungal sinusitis suggestive of mucormycosis [Figure 2] and [Figure 3].
Figure 2: Light brown powdery growth on SDA

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Figure 3 (a-b): Fungal hyphae in histopathological examination

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Direct microscopy using 10% KOH showed broad fungal filaments suggestive of mucormycosis.

Culture on plain and antibiotic (Chloramphenicol and Actidione)-incorporated Sabouraud's Dextrose Agar (SDA) was done. On the 5 th day, growth was obtained on SDA without antibiotics. The colony was white, later became powdery with light brown, and reverse of the colony was tan [Figure 2].

Lactophenol cotton blue (LCB) staining and microscopy of the fungal growth showed broad septate hyphae with plenty of conidia which were scattered and a few were in chains.

Slide culture was put up to study the undisturbed morphology of the fungal isolate for identification [Figure 4]. On the 7 th day, the slide culture revealed septate hyphae, often branched conidiophores bearing annellides that were cylindrical or tenpin shaped. The annellides form in brush-like groups or singly. The conidia are hyaline or pale brown, arranged in chains, round, thick walled, rough and spiny when mature (5-8 μm). The isolate was identified as Scopulariopsis brevicaulis.
Figure 4: Septate hyphae with round conidia in chains

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The patient was treated with Amphotericin B 500 mg in 500 ml 5% Dextrose over 4 h once daily for 15 days and IV Dexamethasone injection thrice daily. Her eye movements became full with no ptosis, but vision remained the same. Later, Itraconazole 500 mg/day in three divided doses was added to the therapeutic regimen in view of reports of Amphotericin B-resistant isolates. The patient responded well with initial Amphotericin B followed by Itraconazole. There has been no recurrence even after 1 year of initial treatment.


  Discussion Top


Scopulariopsis is a filamentous fungus that inhabits soil, plant materials, feathers and insects. It is distributed worldwide. "Scopula" is a Greek word meaning broom, which describes the striking shape of Scopulariopsis conidiophores [2] [Figure 4]. Scopulariopsis species are classified in the order Microascales and genus Microascus. Microascus species and their anamorphs Scopulariopsis species have been recovered from a wide geographic range. S. brevicaulis is a saprophytic organism. [3],[4] It is considered by many mycologists as a common laboratory contaminant, but the isolation of this species from clinical specimens implies a pathogenic role. [1]

The genus is unique in that it contains both moniliaceous (hyaline) and dematiaceous species, with several being clinically significant. [3] The most common species is S. brevicaulis and is a hyaline mould. Other species of hyaline fungi are S. candida, S. kiningii, S. acremonium and S. flava. The phaeoid group includes S. cinerea, S. trigonosporus, S. brumptii, S. chartarum, S. fusca and S. asperula. [3],[5]

S. brevicaulis is a keratophilic fungus, the most frequently reported opportunistic fungus isolated in humans, and is associated mainly with onychomycosis accounting for 1-10%. [6] Rarely, it may be responsible for cutaneous lesions also. Disseminated and invasive tissue disease is associated with a very poor outcome. Some cases were reported in immunocompromised patients. The most common site of isolation of Scopulariopsis is the respiratory tract. [7] Sporadic cases of ocular infection have been recorded previously. In one case of endophthalmitis following retinal detachment surgery, the eye became phthisical and required enucleation, despite therapy. [4] One case of corneal ulcer with Scopulariopsis has been reported from India, but without much clinical details. [8]

The infection in this case was probably acquired from soil contamination. Although many of the fungal infections were described in immunocompromised patients, to our knowledge, this is the first reported case of sinonasal Scopulariopsis in a diabetic patient who was otherwise healthy. In most of the reported cases of sinonasal Scopulariopsis, recurrence of infection is reported when treated with Amphotericin B alone. In contrast to this, this is the second reported case with no recurrence. This may be due to prompt treatment with addition of the drug Itraconazole to Amphotericin B.

Histopathologically, this fungal infection resembles mucormycosis. Invasive sinonasal infections with fungi of the order mucorales are common in immunocompromised patients. Scopulariopsis species, like Fusarium and Curvularia species, are not reliably susceptible to Amphotericin B in vitro. [5] Pierre Sellier et al. reported recurrence of S. brevicaulis infection in a liver transplant recipient after 6 years of transplant and the patient was treated initially with Amphotericin B and later with Terbinafine 250 mg daily. [9] Another case of systemic S. brevicaulis infection in a patient with myeloid leukemia was reported from Malaya in the year 2003. [1] The persistent fever did not respond to Amphotericin B and antibiotics alone. Later, Itraconazole 400 mg/day was added and the infection subsided without recurrence. This study also emphasises the importance of identification of aetiological agents in treating fungal infections. The risk of recurrence should be kept in mind while treating S. brevicaulis infection in immunocompromised patients.

 
  References Top

1.Ng KP, Soo-Hoo TS, Na SL, Gan GG, Sangkar JV, Teh AK. Scopulariopsis Brevicaulis Infection in a Patient with Acute Myloid Leukemia. Med J Malaysia 2003;58:608-12.  Back to cited text no. 1
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2.Philips P, Wood WS, Philips G, Rinaldi MG. Invasive hyalohyphomycosis caused by Scopulariopsis Brevicaulis in a patient undergoing allogeneic bone marrow transplant. Diagn Microbiol Infect Dis 1989;12:429-32.  Back to cited text no. 2
    
3.Bainier. Scopulariopsis. World Register of Marine Species. Belgium: World Register of Marine Species (WoRMS); 1907. (Available form: http://www.marinespecies.org/aphia.php?p=taxdetails&id=100241 . Last accepted date on 2011 Jan 14  Back to cited text no. 3
    
4.Ragge NK, Dean Hart JC, Easty DL, Tyers AG. A case of fungal keratitis caused by Scopulariopsis brevicaulis: Treatment with antifungal agents and penetrating keratoplasty. Br J Ophthalmol 1990;74:561-2.  Back to cited text no. 4
    
5.Kriesel JD, Adderson EE, Gooch WM 3 rd , Pavia AT. Invasive sinonasal disease due to Scopulariopsis candida: Case report and review of scopulariopsosis. Clin Infect Dis 1994;19:317-9.  Back to cited text no. 5
    
6.Summerbell RC, Kane J, Krajden S. Onychomycosis, tinea pedis and tinea manuum caused by non-dermatophytic filamentous fungi. Mycoses 1989;32:609-19.  Back to cited text no. 6
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7.Marcelo Sandoval-Denis, Deanna AS, Annette WF. Scopulariopsis, a Poorly Known Opportunistic Fungus; Spectrum of Species in Clinical Samples and In vitro Responses to Antifungal Drugs. J Clin Microbiol 2013;51:3937-43.  Back to cited text no. 7
    
8.Nitzulescu V, Niculescu M. Mycotic panophthalmitis caused by Scopulariopsis brevicaulis. Arch Roum Pathol Exp Microbiol 1976;35:273-6.  Back to cited text no. 8
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9.Sellier P, Monsuez JJ, Lacroix C, Feray C, Evans J, Minozzi C, et al. Recurrent subcutaneous infection due to Scopulariopsis Brevicaulis in a liver transplant recipient. Clin Infect Dis 2000;30:820-3.  Back to cited text no. 9
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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