Medical Science - Case Report Nasal rhinosporidiosis with an atypical presentation Manoharan Prakash, Johnny J. Carlton
Department of ENT, Sree Balaji Medical College and Hospital, Bharath University, Chromepet, Chennai, Tamil Nadu, India
ABSTRACT 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.
Address for correspondence: Dr. Manoharan Prakash, E-mail: [email protected] Received : 31-10-14 Review completed : 31-10-14 Accepted : 09-11-14
KEY WORDS: Bleeding polyps, nasal rhinosporidiosis, Rhinosporidium seebri
R
hinosporidiosis 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 Access this article online Quick Response Code:
Website: www.jpbsonline.org DOI: 10.4103/0975-7406.155810
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]
How to cite this article: Prakash M, Carlton JJ. Nasal rhinosporidiosis with an atypical presentation. J Pharm Bioall Sci 2015;7:S72-3.
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Journal of Pharmacy and Bioallied Sciences April 2015 Vol 7 Supplement 1
Prakash and Carlton: Nasal rhinosporidiosis with an atypical presentation
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[2-4] another addon 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
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[2-4] 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
Journal of Pharmacy and Bioallied Sciences April 2015 Vol 7 Supplement 1
References 1. 2. 3. 4. 5. 6. 7. 8.
Arseculeratne SN. Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian J Med Microbiol 2002;20:119-31. Gleeson M, George GB, Burton MJ, Clarke R, Hibbert J, Jones NS, et al. Scott-Brown’s Otorhinolaryngology and Head and Neck Surgery. 7th ed. Vol. 1. UK: Hodder Arnold Publications; 2008. p. 215. Cumming CW, Flint PW, Harker LA, Haughey BH, Richardson MA, Robbins KT, et al. Cummings Otolaryngology: Head and Neck Surgery. 5th ed. Vol. 1: Elsevier Mosby Publications; 2010. p. 940. Dhingra PL, Dhingra S. Diseases of Ear, Nose and Throat. 5th ed.: Elsevier Publications; 2010. p. 174. Arseculeratne SN, Sumathipala S, Eriyagama NB. Patterns of rhinosporidiosis in Sri Lanka: comparison with international data. Southeast Asian J Trop Med Public Health 2010;41:175-91. Satyanarayana C. Rhinosporidiosis. In: Ellis M, editor. Clinical Surgery. Ear, Nose and Throat. Ch. 13. London: Butterworths; 1966. p. 143. Kameswaran M, Kumar RS, Murali S, Raghunandhan S, Jacob J. KTP-532 laser in the management of rhinosporidiosis. Indian J Otolaryngol Head Neck Surg 2005;57:298-300. Shailendra S, Prepageran N. Oropharyngeal rhinosporidiosis in a migrant worker: A delayed presentation. The Medical journal of Malaysia. 2008;63:65-6.
Source of Support: Nil, Conflict of Interest: None declared.
S73
Copyright of Journal of Pharmacy & Bioallied Sciences is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Department of ENT, Sree Balaji Medical College and Hospital, Bharath University, Chromepet, Chennai, Tamil Nadu, India
ABSTRACT 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.
Address for correspondence: Dr. Manoharan Prakash, E-mail: [email protected] Received : 31-10-14 Review completed : 31-10-14 Accepted : 09-11-14
KEY WORDS: Bleeding polyps, nasal rhinosporidiosis, Rhinosporidium seebri
R
hinosporidiosis 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 Access this article online Quick Response Code:
Website: www.jpbsonline.org DOI: 10.4103/0975-7406.155810
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]
How to cite this article: Prakash M, Carlton JJ. Nasal rhinosporidiosis with an atypical presentation. J Pharm Bioall Sci 2015;7:S72-3.
S72
Journal of Pharmacy and Bioallied Sciences April 2015 Vol 7 Supplement 1
Prakash and Carlton: Nasal rhinosporidiosis with an atypical presentation
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[2-4] another addon 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
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[2-4] 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
Journal of Pharmacy and Bioallied Sciences April 2015 Vol 7 Supplement 1
References 1. 2. 3. 4. 5. 6. 7. 8.
Arseculeratne SN. Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian J Med Microbiol 2002;20:119-31. Gleeson M, George GB, Burton MJ, Clarke R, Hibbert J, Jones NS, et al. Scott-Brown’s Otorhinolaryngology and Head and Neck Surgery. 7th ed. Vol. 1. UK: Hodder Arnold Publications; 2008. p. 215. Cumming CW, Flint PW, Harker LA, Haughey BH, Richardson MA, Robbins KT, et al. Cummings Otolaryngology: Head and Neck Surgery. 5th ed. Vol. 1: Elsevier Mosby Publications; 2010. p. 940. Dhingra PL, Dhingra S. Diseases of Ear, Nose and Throat. 5th ed.: Elsevier Publications; 2010. p. 174. Arseculeratne SN, Sumathipala S, Eriyagama NB. Patterns of rhinosporidiosis in Sri Lanka: comparison with international data. Southeast Asian J Trop Med Public Health 2010;41:175-91. Satyanarayana C. Rhinosporidiosis. In: Ellis M, editor. Clinical Surgery. Ear, Nose and Throat. Ch. 13. London: Butterworths; 1966. p. 143. Kameswaran M, Kumar RS, Murali S, Raghunandhan S, Jacob J. KTP-532 laser in the management of rhinosporidiosis. Indian J Otolaryngol Head Neck Surg 2005;57:298-300. Shailendra S, Prepageran N. Oropharyngeal rhinosporidiosis in a migrant worker: A delayed presentation. The Medical journal of Malaysia. 2008;63:65-6.
Source of Support: Nil, Conflict of Interest: None declared.
S73
Copyright of Journal of Pharmacy & Bioallied Sciences is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
