|Year : 2014 | Volume
| Issue : 3 | Page : 336-340
ADR: An atypical presentation of rare dematiaceous fungus
J Karthika1, V Ramesh2, Shivakamy1, Valli2
1 Department of Microbiology, Sri Sathya Sai Medical College and Research Institute, Thiruporur, Nellikuppam, Chengalpet, Kanchipuram, Tamil Nadu, India
2 Department of Ear, Nose, and Throat, Sri Sathya Sai Medical College and Research Institute, Thiruporur, Nellikuppam, Chengalpet, Kanchipuram, India
|Date of Submission||08-Nov-2013|
|Date of Acceptance||13-Nov-2013|
|Date of Web Publication||10-Jul-2014|
Department of Microbiology, Sri Sathya Sai Medical College and Research Institute, Thiruporur, Nellikuppam, Chengalpet, Kanchipuram, Tamil Nadu
Source of Support: None, Conflict of Interest: None
The association of fungus in allergic fungal rhino sinusitis has been around 200 times in the world literature. As per the available literature, the most common agent identified so far appears to be ASPERGILLUS, though the condition is increasingly associated with Dematiaceous fungi. Here we report for the first time the presence of unusual fungus in allergic rhino sinusitis, which has not been reported so far.
Keywords: Allergic fungal rhino sinusitis, dematiaceous fungi, fungal ball, rhino sinusitis
|How to cite this article:|
Karthika J, Ramesh V, Shivakamy, Valli. ADR: An atypical presentation of rare dematiaceous fungus. Indian J Med Microbiol 2014;32:336-40
|How to cite this URL:|
Karthika J, Ramesh V, Shivakamy, Valli. ADR: An atypical presentation of rare dematiaceous fungus. Indian J Med Microbiol [serial online] 2014 [cited 2020 Feb 27];32:336-40. Available from: http://www.ijmm.org/text.asp?2014/32/3/336/136595
| ~ Introduction|| |
Rhino sinusitis is the inflammation of nasal and para nasal sinus mucosa and is associated with mucosal alterations ranging from inflammatory thickening to gross nasal polyp formation.  This inflammation may be due to microbes (bacteria and fungi) or allergic and non-allergic causes.
Fungal rhino sinusitis (FRS) is classified into fungal ball, allergic fungal rhino sinusitis (AFRS), acute invasive fungal rhino sinusitis (AIFRS) or chronic invasive fungal rhino sinusitis (CIFRS) and granulomatous invasive fungal rhino sinusitis (GIFRS) depending on the invasion into the mucosa or surrounding structures.  While the other varieties are seen in immune comprised patients; fungal ball and AFRS are commonly seen in younger (<30 years) immuno competent patients, with unilateral or bilateral foci, sinus opacities with heterogeneous shadows and bony expansions in computed tomography (CT). But dural or periosteal invasions are not seen. Presence of a heterogeneous shadow inside the sinus cavity should raise the suspicion of a FRS.
AFRS is defined as an immuno competent patient developing allergy to the fungal spores in the nasal or sinus cavity. The fungi, which are the cause of hypersensitivity, reside in the Mucin and provide continued stimulation to the immune system, leading to the clinical manifestation. It is suggested that AFRS has a similar aetiology to allergic broncho pulmonary aspergillosis (ABPA).
AFRS constitutes about 7% of chronic rhino sinusitis cases requiring surgical management especially in the USA.  AFRS is more prevalent in warm and humid areas; but seen in dry weather also.  As per the available literature, the most common agent identified so far appears to be Aspergillus.  Dematiceous fungal species are a group of melanised fungi, which produce brown pigmentations like melanin and Bipolaris, Curvularia and Alternaria are some of the species. 
Pheohypomycosis is a group of clinical syndromes caused by melanised fungi and are classified into allergic disease, superficial and deep local infections and pulmonary and CNS infections and disseminated disease.
Clinical disease due to melanised fungi are an uncommon condition with an average of one case per million people per year in a metropolitan area. They are being increasingly recognised as important pathogens in individuals with compromised or normal immune status, and in fatal infections. 
Over 150 species and 70 genera of these fungi have been implicated in human and animal disease and Ochroconis is one among the human pathogens. It is known to cause severe necrotising encephalitis in man, cats and birds. But the association in allergic rhino sinusitis is reported for the first time in our patient!
| ~ Case Report|| |
A 30-year-old female [Figure 1] who was a house wife and agricultural worker was referred to the ENT Department in May 2013 with case of recurrent right sided headache, frequent sneezing and right nasal block for the past 1 year. She was treated earlier with medicines in several hospitals and was not relieved of her symptoms. She had also undergone a computed tomography (CT) of the brain for headache, which was reported as normal.
Nasal examination showed DNS, congested turbinates, mucopurulent nasal discharge and tenderness over the frontal and maxillary sinuses on right side. X-ray of the para nasal sinuses showed haziness of both maxillary sinuses. Nasal endoscopy showed a polyp and mucopurulent discharge in the middle meatus. A provisional diagnosis of allergic rhinitis with chronic rhino sinusitis was made.
Investigations showed RBS: 81 mg/dl, Hb: 12 g/dl, TLC 9900 cells; DLC-Eosinophil count: 20% and ESR 35 mm/h. CT of the PNS showed right pan sinusitis with a heterogeneous opacity in right maxillary sinus [Figure 2]a].
|Figure 2: Pre op CT PNS coronal view showing the heterogeneous opacity in the RT. Maxillary sinus (a) Pre op CT PNS axial view showing the heterogeneous opacity in the rt. Maxillary sinus|
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AFRS was suspected and patient was treated with inj. ceftrioxone and inj. prednisolone 5 mg bid for 2 days followed by oral prednisolone 5 mg tid and topical nasal steroids for a week and on 06 June 2013, FESS using Karl Zeiss 0* rigid nasal endoscope was done under general anaesthesia. After a right uncinectomy, the polyps from the right middle meatus were removed and sent for HPE. When the polyps around the right maxillary sinus out from the right maxillary sinus [Figure 3] and they were sent for fungal culture and KOH mounting. The maxillary sinus was thoroughly irrigated with normal saline.
The patient was treated with inj. ceftriaxone, tab. prednisolone 5 mg tid, tab. cetirizine, tab. ranitidine, alkaline nasal wash and other supportive measures. Endoscopic nasal cleaning was done on the second and fourth post-operative days and the blood clots and uneventful and the patient was discharged after 1 week.
|Figure 3: The brown colored material removed from the Rt. Maxillary sinus|
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KOH mounting reported as positive for fungus with septate hyphae. The HPE showed a granulomatous polyp [Figure 4]. After 1 week of inoculation into SDA media; brownish black colonies with a velvety texture were seen [Figure 5]. Microscopy with LPCB showed a typical two celled clavate conidia,  which confirmed the diagnosis of the rare Dematiceous fungi Ochroconis gallopavum [Figure 6]. The Mason-Fontana stain showed characteristic black coloured hyphae,  which helped in to confirm the presence of Dematiceous fungus [Figure 7].
The patient was symptom free when reviewed after 3 weeks and a repeat CT PNS and nasal endoscopy were done. The postoperative CT showed total clearance of the right maxillary sinus [Figure 8]. The nasal cavity was clear with good airway and no fungal colonies were seen. In view of the AFRS, a retrospective history taking was done and it revealed that she lives in a thatched house and keeps cattle (cows) in her house for the past 1 year. She is currently taking steroid nasal spray, oral antihistamine, tab. monteleucast and is being reviewed monthly.
| ~ Discussion|| |
Ochroconis gallopavum has been isolated from some hot springs in Japan.  The source of Ochroconis gallopavum is usually from the contaminated soil. Several publications show that the Ochroconis gallopavum have been associated with infections of CNS and lungs in Immuno compromised or in transplant patients. In our search of the available literatures, we could not come across any case of sinus infection with Ochroconis gallopavum in an immune competent patient.
Rhino sinusitis is a common disorder affecting approximately 20% of the population at some time of their lives.  FRS is a subset of rhino sinusitis where the etiological role of fungi is proven by isolating the fungi from biopsy samples or culture. FRS is being increasingly recognised in immuno competent patients of all age groups. The resulting morbidity and high mortality of acute invasive FRS has huge socioeconomic effects including both direct and indirect costs to the society. Hence the early diagnosis of FRS is very important and it is based on high index of clinical suspicion, supported by direct microscopy, culture and HPE of the tissue. The direct microscopy along with culture helps in clinching the aetiological agent  and HPE helps to distinguish between invasive and non-invasive stages. 
FRS, which was once considered as a rare disorder, is now being recognised and reported with increasing frequency worldwide. Plaignaud in1791 described a 'FUNGUS TUMOUR' in the maxillary sinus of a 33-year-old soldier.  Mackenzie in 1894 described the first case of apparent non-invasive fungal sinusitis; later in 1897, Aspergillus species was described in an infection of the sphenoid sinus with extension in to the cerebrum through an erosion of the bony wall in 1965 HORA recognised two categories of fungal sinusitis: Invasive and non-invasive.
Several hypotheses have been proposed for the pathophysiology of AFRS. The inhaled fungal spores could act like an antigen, provoking the nasal mucosa and precipitating an inflammatory response and a thicker mucin production with plenty of eosinophils (Eosinophilic Mucin). New hypothesis with microbial super antigens have also been suggested.
Our patient is a homemaker doing domestic and agriculture works apart from cattle rearing. We believe that while handling wet hay stakes or while working in the agricultural soil, the causative fungal spores might have been inhaled, leading to the development of AFRS.
AFRS is classified into stages 0-3 according to the endoscopic findings and diagnostic criteria [Table 1].  Based on the clinical, endoscopic, HPE and culture f findings, our patient is categorised, improving the sinus ventilation and topical nasal steroids for 1 year. Anti-fungals are not recommended.
| ~ Conclusion|| |
We report for the first time, a rare case of AFRS with rare melanised fungi-Ochroconis gallopavum involving the maxillary sinus. Incidence of FRS is on the rise and a strong clinical suspicion with good laboratory workout is needed for the correct diagnosis of FRS. Early diagnosis and appropriate treatment is imperative to improve the quality of life and avoid complications.
'In The Field Of Observation, Chance Favours Only the Prepared Mind'-Louis Pasteur.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]