Nasal rhinosporidiosis with an atypical presentation.

Rhinosporidiosis is a unique disease, which is seen to be endemic in certain places in India such as Tamil Nadu and Sri Lanka. The disease is caused by Rhinosporidium seebri and it is transmitted by bathing in ponds contaminated by cattle feces containing spores of the organism. The disease usually presents as multiple granulomatous bleeding polyps. The case described here is a unique presentation where it occurs only in a single site and that too in an uncommon location where the suspicion of rhinosporidiosis is a last possibility.

Rhinosporidiosis has been reported from about 70 countries[1] with diverse geographical features. The highest incidence has been from India and Sri Lanka.[1] Most cases of human rhinosporidiosis in western temperate and Middle Eastern countries occurred in expatriate Indians who probably acquired the disease in their native lands. It has also been increasing in the United States due to refugees from Asia. It commonly occurs in the nose and the nasopharynx but can be widespread with systemic manifestation involving palate, vagina, vulva, larynx, conjunctiva, etc. The manifestation is usually as a leafy polypoidal bleeding mass which is usually attached to the nasal septum or lateral wall appearing blue to purple in color studded with white dots that are usually the sporangia. Its usual presentation in the nose is epistaxis that is due to its high vascularity.

Case Report

Our case is a 37-year-old male who hails from Sivagangai District, Tamil Nadu. The patient developed epistaxis from the left nasal cavity since 2 days. The patient had minimal bleeding of 20 ml/day within the past 2 days which was not associated with pain, postural change, trauma or nose picking. Nasal block was predominantly on the left side for the past 6 months. He had a history of headache, that was dull aching in type and confined to the frontal region. The patient gives a classical history of residing in an endemic region with cattle rearing as his occupation and also gives a history of routine bathing in the ponds along the animals. On examination, a small single fleshy bluish pink pedunculated polypoidal mass was seen to be attached to the anterior aspect of inferior turbinate. Its surface was smooth, and it was not probed due to anticipation of bleeding. All routine investigations were done, and contrast computed tomography paranasal sinuses was done which showed a single nonenhancing mass seen to be attached to the inferior turbinate with no signs of bony erosion and any other abnormalities. The patient was later planned for elective surgery, and an endoscopic excision was done and the base was cauterized to prevent recurrence. The specimen was sent for histopathological examination where it showed that the polyp was lined by stratified squamous epithelium and it was infiltrated by numerous lymphocytes and plasma cells. Is had numerous spore in various stages of maturation typical of rhinosporidiosis. Later follow-up of the case was done at 6 months and after 1-year where no signs of recurrence seen.

Discussion

Rhinosporidiosis is seen to be endemic is seen in about 70 countries but its incidence is very high in India and Sri Lanka,[1] In India certain places Tamil Nadu like, Madurai, Sivagangai, Ramanathapuram, Chenglepet (Singaperumalkoil) districts are highly endemic for the disease. The organism causing the disease is described in textbooks as the fungi Rhinosporidium seebri[234] but latest research conclude that the actual organism belongs to clusters with novel group of fish parasites referred to as the dermocystidium, rossete agensts, icthyophorus and psorospermium clade.[1] The presentation of the disease classical as the person belongs to an endemic area, bathes in ponds and is also a cattle raiser all of which have documental evidence of being associated with rhinosporidiosis.[5] The atypical part of the case was the site and number of presenting lesions. It is described that the polyp usually presents at multiple sites and is attached to the septum and lateral wall.[4] The presentation in our case is solitary, and the site is also very unique as it is attached to the inferior turbinate. There has also been some literature where the inferior turbinate involvement has been seen.[6] This also promotes us to the fact that we might have diagnosed the patient in the very early course of the disease and if not he would have progressed to the advanced stages and presented with the more textbook features. More importantly the recurrence is also high with florid rhinosporidiosis. The treatment which we have offered is simple endoscopic excision and cauterization of the base which is approved by standard textbooks[234] another add-on therapy, which could have been added is dapsone to prevent recurrence.[4] The histopathological findings also are very standard when compared with some findings like pseudoepithelomatous squamous cell metaplasia [Figure 1]. At present, the latest methods of treatment include the use of KTP-532 potassium titanyl phosphate laser for treatment and has been showing promising results.[7] The classical findings like the fungal spores were also visualized which were similar to the images in Figure 1.[8]

Figure 1

Rhinosporidial spores