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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 14  |  Issue : 1  |  Page : 156-159

An unusual presentation of rhinosporidiosis


Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India

Date of Submission22-Aug-2020
Date of Acceptance15-Oct-2020
Date of Web Publication09-Feb-2021

Correspondence Address:
Dr. Santosh Kumar Swain
Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan University, K8, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kleuhsj.kleuhsj_267_20

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  Abstract 


Rhinosporidiosis is a chronic granulomatous infection of the nose and nasopharynx. It is endemic in South Asia and commonly affects males in their second to fourth decades of life. Majority of the cases are found in the upper respiratory tract such as anterior nares, nasal cavity, inferior turbinate, nasal septum, floor, and nasopharynx. Although nose or nasopharynx is the most common location for rhinosporidiosis, it can also involve the skin, parotid duct, oropharynx, and larynx. Here, we report the case of a 42-year-old male who presented with extensive rhinosporidiosis in both nasal cavities and nasopharynx along with involvement of the roof of the oral cavity through erosion of the hard palate. The mainstay of treatment is wide excision followed by electrocautery of the base which ablates the recurrence. The rhinosporidiosis mass from different sites of the attachment was completely excised along with cauterization of the base. The diagnosis was confirmed by histopathological report.

Keywords: Nasal cavity, oral cavity, palate, rhinosporidiosis


How to cite this article:
Swain SK, Sahu A. An unusual presentation of rhinosporidiosis. Indian J Health Sci Biomed Res 2021;14:156-9

How to cite this URL:
Swain SK, Sahu A. An unusual presentation of rhinosporidiosis. Indian J Health Sci Biomed Res [serial online] 2021 [cited 2021 Mar 2];14:156-9. Available from: https://www.ijournalhs.org/text.asp?2021/14/1/156/308959




  Introduction Top


Rhinosporidiosis is a rare chronic granulomatous disease caused by Rhinosporidium seeberi under the class Mesomycetozoea.[1] The class Mesomycetozoea, earlier called the DRIP (Dermocystidium, Rosette agent, Ichthyophonus and Psorospermium clade) which is placed in between the fungi and animals and are usually parasites of the fish and other animals.[1] Rhinosporidiosis is endemic in some parts of India, Sri Lanka, tropical region of Africa, and South America. The nasal cavity is primarily affected by rhinosporidiosis. It commonly involves anterior nares, inferior turbinates, nasal septum, and floor of the nasal cavity. However, it can be found in nasopharynx, oropharynx, conjunctiva, rectum, and external genitalia.[2] Patients often present with large polypoidal lesions in the nasal cavity which are hyperplastic, highly friable, and pedunculated or sessile. Histopathological examination confirms the diagnosis. The treatment of choice is surgery, but it often poses a challenge because of its friable nature, site of origin, and recurrence.[3] Previously, rhinosporidiosis was endemic in India, but currently, sporadic cases are reported mostly as recurrent forms. Here, we report a recurrent case of rhinosporidiosis that extended into the oral cavity via the defect in the hard palate. Lesions of rhinosporidiosis spreading from the nasal cavity and extending to the oral cavity through the hard palate are an extremely rare presentation in medical literature.


  Case Report Top


A 42-year-old male patient attended the outpatient department of otorhinolaryngology with complaints of bilateral nasal block for 6 months. He had three episodes of nasal bleeding during the last 6 months. He also had a history of mouth breathing and snoring during sleep. He had occasional bleeding from the roof of the mouth, specifically during eating solid food. There were no such manifestations in his family members. He had a history of pond bathing in his village, which is found in the eastern part of India, since childhood. He had no history of excessive sneezing, rhinorrhea, or trauma to the nose. The patient was diagnosed as rhinosporidiosis 3 years back and once underwent excision of the mass before 3 years. On inspection of the nose, a red, friable, and polypoidal mass was protruding from both nostrils [Figure 1]. Oral cavity examination showed a soft, pinkish mass hanging from the hard palate [Figure 1]. Indirect laryngoscopic examination showed normal larynx and hypopharynx. There was no cervical lymphadenopathy. Based on the patient history and clinical findings, a provisional diagnosis of rhinosporidiosis was made. Routine blood tests and serological tests for HIV were within normal limits. Chest X-ray was normal. Computed tomography scan revealed opacity/mass in the right nostril. There was no evidence of bony erosion in the nasal wall. The nasal, nasopharyngeal, and palatal mass was completely excised, and cauterization of the base of the mass with electric diathermy was done under general anesthesia. Palpation of the palatal area after excision of the rhinosporidiosis showed a dehiscence in the bony partition between the oral cavity and nasal cavity. The bony dehiscence may be due to injury by a previous trauma or congenital bony defect or because of the invasiveness of the rhinosporidiosis. The mass was sent for histopathological examination. Histopathological examination revealed the presence of multiple sporangia which contained spores of R. seeberi [Figure 2]. The postoperative period was uneventful, and the patient was started on dapsone for prevention of recurrence. Postoperatively, the patient was treated with dapsone 100 mg, one tablet daily for 1 year, for preventing the recurrence. The patient was on regular follow-up for every 3 months, and there was no recurrence 1 year after the surgery. There was no evidence of synechia, adhesion, septal perforation, and any lesion in the oral cavity.
Figure 1: Patient showing reddish mass of rhinosporidiosis protruding from both nostrils and the palate

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Figure 2: Microphotograph showing histopathological picture of rhinosporidiosis with presence of multiple sporangia and sporocysts (H and E, ×100)

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  Discussion Top


Rhinosporidiosis is a chronic granulomatous mucocutaneous infection caused by R. seeberi.[4] The first known case of rhinosporidiosis was identified in 1892 by Malbran from Buenos Aires.[5] Guillermo Seeber published about rhinosporidiosis first and described its details in 1900.[5] Asworth documented the life cycle and etiology of rhinosporidiosis, and Karunaratne described a detailed review of rhinosporidiosis in humans in 1964.[5] It is widely thought to be caused by a fungus but it is disputed by many. A recent work on rhinosporidiosis suggests that it is caused by Cyanobacteria. It is endemic in South Asia, with the largest number of patients reported in India and Sri Lanka.[4] Previously, this disease was more prevalent in the Indian subcontinent, but in the present era, the incidence of rhinosporidiosis had significantly reduced except some recurrent cases. The present case is also a recurrent case of rhinosporidiosis which was operated before 3 years. The taxonomy and life cycle of rhinosporidiosis are incompletely understood and controversial.?[1] Hence, till date, this disease is usually associated with diagnostic and treatment challenges to clinicians. Rhinosporidiosis commonly affects mucous membranes of the nasal cavity and nasopharynx. It also occasionally affects lips, palate, uvula, maxillary sinus, lacrimal sac, conjunctiva, epiglottis, larynx, trachea, bronchus, scalp, skin, vulva, vagina, and penis.[6] This disease is usually transmitted through bathing in a stagnant pond where animals also bathe. In the present case, the patient also had the habit of pond bathing. Although rare, other modes of transmission include autoinoculation and hematogenous spread.[7] Many authors have proven the mode of transmission of R. seeberi which are based on the findings of such protistan parasites in ponds and rivers[8] The patients with rhinosporidiosis are often used to take bathe and the same history was found in our case, which could be the possible cause for the acquisition of infection.

Rhinosporidiosis is commonly found in males, with a male-to-female ratio of 4:1 and common in the age group between 10 and 40 years.[9] Nasal cavity and nasopharynx are the most common locations of this infection followed by the eye. In our case, the location is atypical as the infection of the nasal cavity spread to the oral cavity via the hard palate. Patients with rhinosporidiosis often present with a reddish nasal mass which bleeds profusely if injured or sometimes spontaneously. The mass is typically soft, fleshy, vascular, and polypoidal in appearance. Sometimes, rhinosporidiosis at the posterior part of the nasal cavity and nasopharynx may mimic juvenile nasopharyngeal angiofibroma and create a clinical dilemma. Disseminated rhinosporidiosis is common in immunocompromised patients with a high chance of mortality.[7] The diagnosis is often done on the basis of clinical presentation and nasal endoscopy and confirmed on the basis of histopathological examination which shows round-to-oval sporangia and spores in the affected mass or tissue.[2] R. seeberi is intractable for isolation and culture and shows morphological appearance of the fungi and protozoa.[10] However, R. seeberi is widely believed as a fungus. It can be observed through fungal stains such as Gomori methenamine silver, periodic acid–Schiff, and hematoxylin and eosin (H and E) staining.[8] The differential diagnosis includes infected nasal polyp, inverted papilloma, fungal polyp, and angiofibroma.

Treatment of rhinosporidiosis is considered a gray area in otolaryngology practice. This is because of the high chance of recurrence after surgery. The mainstay of treatment of rhinosporidiosis is complete excision of the mass by electric diathermy or laser. Cauterization of the base of the mass is meant to prevent recurrence. In previous surgeries, in spite of the use of diathermy at the attachment side of the lesion, recurrences were noted and a new site such as oral cavity was involved. Medical treatment with dapsone is recommended for prevention of recurrence of rhinosporidiosis although with doubtful results. Dapsone at a dose of 100 mg per day for 6 months to 1 year is useful to reduce the recurrence.[11] Dapsone appears to arrest the maturation of the sporangia and also promote fibrosis in the stroma.[12] Laser can be used to provide adequate cauterization and good hemostasis so that the chances of recurrence and complications can be reduced. Nowadays, lasers are used for excision of rhinosporidiosis at some centers. However, lasers are still not freely available in endemic regions for rhinosporidiosis, such as India and other developing countries. Use of CO2 and KTP laser for treatment of rhinosporidiosis is an alternative for achieving a bloodless field without any advantage over recurrence. Laser can also be used as an adjunct during surgery of rhinosporidiosis, but care should be taken for eradication of the disease completely and the attachments should be cauterized or laserized after removal of the disease bulk.[13] Although rhinosporidiosis is not fatal, it often recurs which may need multiple surgeries, which, in turn, may lead to increased morbidity.


  Conclusion Top


Although nasal and nasopharyngeal rhinosporidiosis are commonly reported in the medical literature, the incidence of rhinosporidiosis eroding through the palate and extending to the oral cavity is extremely rare. Complete excision of rhinosporidiosis from the nasal cavity along its extension into the oral cavity is often challenging for the surgeon. Proper identification of the site of attachment and identifying its extension with cauterization of the base and postoperative dapsone is very much helpful for its cure and prevention of recurrence. Early identification or diagnosis with prompt surgical management will reduce significant morbidity associated with rhinosporidiosis. Clinicians must be aware regarding the invasive property of rhinosporidiosis during managing patients with lesion in the oral cavity although it is extremely rare.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Vilela R, Mendoza L. The taxonomy and phylogenetics of the human and animal pathogen Rhinosporidium seeberi: A critical review. Rev Iberoam Micol 2012;29:185-99.  Back to cited text no. 1
    
2.
Almeida FA, Feitoza Lde M, Pinho JD, Mello GC, Lages JS, Silva FF, et al. Rhinosporidiosis: The largest case series in Brazil. Rev Soc Bras Med Trop 2016;49:473-6.  Back to cited text no. 2
    
3.
Sudarshan V, Goel NK, Gahine R, Krishnani C. Rhinosporidiosis in Raipur, Chhattisgarh: A report of 462 cases. Indian J Pathol Microbiol 2007;50:718-21.  Back to cited text no. 3
[PUBMED]    
4.
Lupi O, Tyring SK, McGinnis MR. Tropical dermatology, fungal tropical disease. J Am Acad Dermatol 2005;53:931.  Back to cited text no. 4
    
5.
Loh KS, Chong SM, Pang YT, Soh K. Rhinosporidiosis: Differential diagnosis of a large nasal mass. Otolaryngol Head Neck Surg 2001;124:121-2.  Back to cited text no. 5
    
6.
Banerjee SB, Sarkar A, Mukherjee S, Bhownik A. Laryngeal rhinosporidiosis. J Indian Med Assoc 1996;94:148-50.  Back to cited text no. 6
    
7.
Madana J, Yolmo D, Gopalakrishnan S, Saxena SK. Rhinosporidiosis of the upper airways and trachea. J Laryngol Otol 2010;124:1139-41.  Back to cited text no. 7
    
8.
Morelli L, Polce M, Piscioli F, Del Nonno F, Covello R, Brenna A, et al. Human nasal rhinosporidiosis: An Italian case report. Diagn Pathol 2006;1:25.  Back to cited text no. 8
    
9.
Bakshi SS. Rhinosporidiosis. J Allergy Clin Immunol Pract 2017;5:1739.  Back to cited text no. 9
    
10.
Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidium seeberi's 18S small-subunit ribosomal DNA groups this pathogen among members of the protoctistan Mesomycetozoa clade. J Clin Microbiol 1999;37:2750-4.  Back to cited text no. 10
    
11.
Kaushal S, Mathur SR, Mallick SR, Ramam M. Disseminated cutaneous, laryngeal, nasopharyngeal, and recurrent obstructive nasal rhinosporidiosis in an immunocompetent adult: A case report and review of literature. Int J Dermatol 2011;50:340-2.  Back to cited text no. 11
    
12.
Janardhanan J, Patole S, Varghese L, Rupa V, Tirkey AJ, Varghese GM. Elusive treatment for human rhinosporidiosis. Int J Infect Dis 2016;48:3-4.  Back to cited text no. 12
    
13.
Tiwari. Rhinosporidiosis: A riddled disease of man and animals. Adv Anim Vet Sci 2015;3:55.  Back to cited text no. 13
    


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