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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
CASE REPORT Table of Contents   
Year : 2007  |  Volume : 11  |  Issue : 2  |  Page : 83-85
 

Rhinosporidiosis of the nose in the southern region of the Kingdom of Saudi Arabia


1 Ear, Nose and Throat Department, King Khalid University, Kingdom of Saudi Arabia
2 Histopathology Section, Assir Central Hospital, Kingdom of Saudi Arabia

Correspondence Address:
Mahmoud R Hussein
Department of Pathology, Faculty of Medicine, Assuit Univeristy, Egypt

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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.37390

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   Abstract 

Rhinosporidiosis is a chronic fungal infection caused by Rhinosporidium seeberi . It selectively strikes two target zones: nasal and conjunctival mucosae. Patients with nasal rhinosporidiosis usually present with nasal obstruction, discharge, epistaxis and mass lesions. This investigation presents the first indigenous case of tumoral rhinosporidiosis in the southern region (Assir province) of the Kingdom of Saudi Arabia. A 78-year-old male patient from a rural background of Assir region presented with epistaxis, nasal obstruction, discharge and a polympoid mass. The patient had not traveled abroad. Diagnosis was established on the morphological ground by the identification of l5- to 10-micron endospores and 50- to 1,000-micron sporangia. The morphologic features were examined using histochemical stains. The relevant monographs and case reports are discussed.


Keywords: Rhinosporidiosis, nose, Saudi Arabia


How to cite this article:
Al-Shehry A, Hussein MR. Rhinosporidiosis of the nose in the southern region of the Kingdom of Saudi Arabia. J Oral Maxillofac Pathol 2007;11:83-5

How to cite this URL:
Al-Shehry A, Hussein MR. Rhinosporidiosis of the nose in the southern region of the Kingdom of Saudi Arabia. J Oral Maxillofac Pathol [serial online] 2007 [cited 2020 Sep 29];11:83-5. Available from: http://www.jomfp.in/text.asp?2007/11/2/83/37390



   Introduction Top


Rhinosporidiosis is a chronic fungal infection caused by Rhinosporidium seeberi. It typically involves mucosal surfaces and adjacent skin, most frequently the nasal mucosa. Rhinosporidiosis affects not only human beings but also animals. [1] This disease is endemic in India, Ceylon and Sri Lanka, with only few cases being reported in Africa, South America and United States of America. The mode of transmission is not known, and dissemination of the organism is a rare event. R. seeberi is a large, endosporulating organism with characteristic morphological features that are readily recognizable in hematoxylin- and eosin-stained sections. Sporangia develop from individual spores into small uninucleate cysts that enlarge and develop a chitinous eosinophilic wall. Subsequent nuclear divisions give rise to about 16,000 spores in a sporangium (300 mm in diameter). Rupture of the cyst or release of the spores through a pore in the cyst wall leads to dissemination of the individual spores into the surrounding tissues.[2] Clinically, rhinosporidiosis usually presents as mass lesions (nasal polyps, tumor-like lesions or verrucous lesions). These lesions are soft, highly vascular, sessile or pedunculated. [3] Diagnosis of rhinosporidiosis rests on the finding of the typical structures of R. seeberi in the tissues. The organisms stain with the Periodic acid Schiff (PAS ) reaction at all stages. Methenamine Silver (GMS) and mucicarmine stains are not effective for organisms less than 100 mm in diameter.[1] Complications of the disease include extremely rare life-threatening dissemination, secondary bacterial infection and recurrence. [4] The disease is treated by surgical excision combined with electrocoagulation. Rhinosporidiosis is not responsive to medical treatment. [5]


   Case Report Top


This report describes a 78-year-old male patient with a nasal mass. The patient had been referred to the Department of Ear, Nose and Throat surgery of Assir Central Hospital and King Khalid University for evaluation of a nasal mass, obstruction, discharge and epistaxis of six-month duration. His general examination and history were unremarkable and did not contain previous complaint of ill health. Clinical examination revealed a right-sided irregular, reddish friable nasal mass. Clinically, rhinosporidiosis was not suspected. Endoscopy, as well as plain X-ray and computerized tomography, showed a polypoid mass originating from the right side of the nose, without any sign of bone destruction. The mass was removed under general anesthesia and submitted for pathological examination. Grossly, a 2.0 2.0 1.0 cm non-encapsulated polypoid mass with soft consistency and hemorrhagic cut surface was seen. On histology (hematoxylin-eosin stained sections), the respiratory epithelium was hyperplastic with papillomatosis and deep invaginations, some of which formed pseudocysts [Figure - 1],[Figure - 2]. Numerous globular cysts of varying shape, representing sporangia in different stages of development, give the epithelium a distinctive 'Swiss cheese' appearance. There is a surrounding and subepithelial dense, mixed inflammatory infiltrate composed of lymphocytes, histiocytes, occasional giant cells, plasma cells, neutrophils and eosinophils. Histochemical stains (Periodic Acid Schiff and Methenamine silver) confirmed the diagnosis. Cytologic criteria for diagnosis included the presence of 5- to 10-micron endospores and 50- to 1,000-micron sporangia. These findings made it easy to differentiate rhinosporidiosis from the more common nasal mycoses such as cryptococcosis [6] [Figure - 1],[Figure - 2].


   Discussion Top


Rhinosporidiosis is a chronic fungal infection that is rare in the Middle East. To date, one case has been reported from Egypt. [1] Here, we report the first case observed in the southern region of the Kingdom of the Saudi Arabia. A 78-year-old male presented with a nasal mass obstructing the right nasal fossa, having developed over more than six months, occasionally accompanied by epistaxis. Diagnosis was made through an anatomo-pathological examination of the biopsy. The treatment essentially consisted of surgical excision. [7] Other problems considered in the differential diagnosis of this case included nasal mucocele, malignancy and Coccidioides immitis. [1] The morphologic characteristics of Rhinosporidium seeberi resemble those of Coccidioides immitis. Both organisms have mature stages that consist of large, thick-walled, spherical structures containing smaller daughter cells (endospores). Therefore, the sporangia of Rhinosporidium seeberi should be distinguished from the spherules produced by Coccidioides immitis. [8],[9] Coccidioides immitis spherules stained in H and E, silver or PAS-stained tissue are usually 20-80 m in diameter, with thick, double refractile wall. The spherules are filled with small (2-4 m in diameter) endospores. The intact spherules are surrounded by a granulomatous inflammatory infiltrate (lymphocytes, plasma, epithelioid and giant cells), whereas the ruptured spherules are surrounded by neutrophils. These features distinguish Coccidioides immitis from Rhinosporidium seeberi . [1]

Rhinosporidiosis was initially described by Seeber in 1900 in an individual from Argentina. It is most commonly observed in persons from, or residing in, the Indian subcontinent or Sri Lanka. The etiologic agent Rhinosporidium seeberi has never been successfully propagated in vitro. Initially, Rhinosporidium seeberi organism was thought to be a parasite. Then it was recognized as a 'water mold'-related fungus, for the past 25 years. Recently, molecular techniques have again demonstrated that this organism is probably an aquatic protistan parasite. Rhinosporidiosis infection is usually limited to the covering surface epithelium (nose, conjunctive, skin, urethra, etc.) and results from a local traumatic inoculation with the organism. Disease progresses with the local replication of R. seeberi and associated hyperplastic growth of host tissue and an inflammatory response. Infection of the upper respiratory tract (nose and nasopharynx) and conjunctivae is observed in 70% and 15%, respectively of persons with rhinosporidiosis. [1]

 
   References Top

1.Hussein MR, Rashad UM. Rhinosporidiosis in Egypt: A case report and review of literature. Mycopathologia 2005;159:205-7.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Thakur SK, Sah SP, Badhu BP. Oculosporidiosis in eastern Nepal: A report of five cases. Southeast Asian J Trop Med Public Health 2002;33:362-4.  Back to cited text no. 2  [PUBMED]  
3.Ahluwalia KB. Causative agent of rhinosporidiosis. J Clin Microbiol 2001;39:413-5.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Arseculeratne SN, Hussein FN, Atapattu DN, Pathmanathan R. Failure to infect congenitally immunodeficient SCID and NUDE mice with Rhinosporidium seeberi . Med Mycol 2000;38:393-5.  Back to cited text no. 4  [PUBMED]  
5.Charles D. Deep mycoses rarely described. Med Trop (Mars) 1986;46:263-7.  Back to cited text no. 5  [PUBMED]  
6.Allison N, Willard MD, Bentinck-Smith J, Davis K. Nasal rhinosporidiosis in two dogs. J Am Vet Med Assoc 1986;188:869-71.  Back to cited text no. 6  [PUBMED]  
7.de Silva NR, Huegel H, Atapattu DN, Arseculeratne SN, Kumarasiri R, Gunawardena S, et al . Cell-mediated immune responses (CMIR) in human rhinosporidiosis. Mycopathologia 2001;152:59-68.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Fredricks DN, Jolley JA, Lepp PW, Kosek JC, Relman DA. Rhinosporidium seeberi : A human pathogen from a novel group of aquatic protistan parasites. Emerg Infect Dis 2000;6:273-82.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Kennedy FA, Buggage RR, Ajello L. Rhinosporidiosis: A description of an unprecedented outbreak in captive swans (Cygnus spp.) and a proposal for revision of the ontogenic nomenclature of Rhinosporidium seeberi . J Med Vet Mycol 1995;33:157-65.  Back to cited text no. 9    


    Figures

  [Figure - 1], [Figure - 2]



 

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    Abstract
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    Case Report
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