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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 14  |  Issue : 4  |  Page : 205-207

Rhinosporidiosis reinfection after 20 years – A case report from United Arab Emirates


1 Department of ENT, Ishaq Bin Omran Medical Center, Sharjah; Department of ENT, MedStar Daycare Center, Dubai; Department of ENT, Zulekha Hospital Sharjah, Sharjah, United Arab Emirates
2 Department of Medicine, Ishaq Bin Omran Medical Center, Sharjah, United Arab Emirates
3 Department of Medicine, Medcare Hospital Sharjah, Sharjah, United Arab Emirates

Date of Submission01-Sep-2021
Date of Decision20-Sep-2021
Date of Acceptance11-Oct-2021
Date of Web Publication11-Jan-2022

Correspondence Address:
Irshad Mazhar Mohiuddin
Department of ENT, Ishaq Bin Omran Medical Center, Sharjah; Department of ENT, MedStar Daycare Center, Dubai; Department of ENT, Zulekha Hospital Sharjah, Sharjah
United Arab Emirates
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/hmj.hmj_56_21

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  Abstract 


Introduction: Rhinosporidiosis is a rare, endemic, chronic granulomatous infection caused by protistan parasite Rhinosporidium Seeberi. It affects upper airways, mostly nasal mucosa and sometimes skin and manifests as lesions that appear like strawberry pulp. It is known for post excision recurrences attributed to residual spores or re-inoculation during excision. Beyond the endemic zones it's rare in the developed world. Case Report: We report nasal Rhinosporidiosis in a case from an endemic area in India, who previously suffered from the disease and was successfully treated 20 years ago and was symptom free until his current reinfection and treatment. Conclusion: Though rare, awareness of this disease is important for clinicians in places with a large migrant population and in cases of travel to an endemic area. The disease can recur on visiting an endemic area even after 20 years of previous treatment and cure as might have happened in our case.

Keywords: Dapsone therapy, electro cautery, recurrent, reinfection, rhinosporidiosis, Rhinosporidium seeberi, surgical excision


How to cite this article:
Mohiuddin IM, Burud S, Harriss M. Rhinosporidiosis reinfection after 20 years – A case report from United Arab Emirates. Hamdan Med J 2021;14:205-7

How to cite this URL:
Mohiuddin IM, Burud S, Harriss M. Rhinosporidiosis reinfection after 20 years – A case report from United Arab Emirates. Hamdan Med J [serial online] 2021 [cited 2022 Jan 20];14:205-7. Available from: http://www.hamdanjournal.org/text.asp?2021/14/4/205/335385




  Introduction Top


Rhinosporidiosisis a rare chronic granulomatous infection caused by an aquatic protistan parasite Rhinosporidium seeberi (R. seeberi)[1] that manifests as characteristic polypoidal mucosal lesions.[2] Its endemic in South Asia (India and Sri-Lanka) and South America (Brazil and Argentina),[3] whereas 90% infections are from Asia and South America accounts for 5% of cases.[3] Although reported from more than 70 countries, most infections in Middle East occur in expat Indians who may have got infected in their native lands.[4] In India, its endemic in Tamil Nadu, Kerala, Pondicherry, Andhra Pradesh, West Bengal and Chhattisgarh.[3]

Since Seeber's descriptions in 1900s, R. seeberi remained enigmatic with a debatable taxonomy. First considered a sporozoon then protozoan then fungus and later classified as a novel clade of aquatic protistan parasites branching in the evolutionary tree near animal-fungal divergence called the DRIPs clade (dermocystidum, rosette agent, ichthyophonus and psorospermium).[5] The infection is associated with rural areas, agricultural workers, contaminated soil/sand, stagnated water bathing (lakes and ponds) and low socioeconomic status.[2],[6]

The association with water and findings that R. seeberi belongs to a clade of aquatic parasites support a hypothesis that fish or other aquatic animals are natural hosts of R. seeberi and humans get infected when they contact water containing these fish and their parasites.[5] Human transmission is by contact with the organism's spores through dust, infected fingers or cloths and stagnant water bathing.[3] The presumed mode of infection from its natural habitat is across traumatised epithelium (trans-epithelial infection) mostly in the nose.[4] Although infective, it is not infectious as transmission across family members is not documented.[4]

Clinically, it manifests as nasal, ocular, cutaneous, disseminated form with classical pedunculated or sessile lesions. In humans, it triggers an immune response but evades host immunity by immune suppression, immune distraction, immune deviation and host immunoglobulin binding which explains its chronicity, recurrence and dissemination.[1]

We describe the case of an expat Indian in United Arab Emirates (UAE) who suffered from Rhinosporidiosis 20 years ago. His first exposure was treated and he was diseases free for the two decades until his current episode of reinfection was identified and managed. There are sporadic reports from Middle East and only one case report from UAE. Although the disease is known for post-excision recurrences usually within a couple of years, we could not find any reported recurrence after the decades of cure from the disease hence this is a reinfection.


  Case Report Top


A 51-year-old Indian male, native of Kerala presented with sudden onset of profuse bleeding from left nostril since a couple of days which stopped on its own in 10 min. The second episode of sudden profuse bleed followed few hours later and stopped with local nasal pressure. No previous history of recurrent nasal bleeds. He was operated for septum and turbinate surgery 6 years back.

On examination, the left nasal cavity was filled with clots with no active ante or post-nasal bleeding. On cleaning the clots, nasal endoscopy revealed strawberry like lesions in the left inferior meatus with abrasions over them that bled to touch. The lesions extended 1.5 cm over the left inferior turbinate and meatus [Figure 1]a. Other otorhinolaryngological and systemic examinations were unremarkable.
Figure 1: (a) Lesion in the left nose with strawberry like appearance. (b) Appearance after surgical excision and cautery of lesion base on follow up. R: Rhinosporidiosis lesion, IT: Inferior turbinate

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Computerised tomographic scan of paranasal sinuses (CT PNS) shows a localised lesion with no bone erosion or sinusitis. An excisional biopsy and cautery of base at the site was performed. Histopathology confirmed rhinosporidiosis [Figure 2].
Figure 2: Histological appearance of the patient's biopsy specimen with oval sporangia containing hundreds of endospores being easily identified under the microscope

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A retrospective history revealed that rhinosporidiosis was diagnosed and surgically excised 20 years back. The patient remained symptom-free since then until his current disease was confirmed as rhinosporidiosis.

He was put on dapsone DDS 100 mg once daily for 1 year and was symptom free on follow-ups at 1 year and 6 months [Figure 1]b. He was advised to avoid contact with reservoirs of infection to prevent future recurrence.

This case demonstrates that rhinosporidiosis in the endemic areas can reinfect after successful treatment of previous episode if adequate precautions to avoid contact from the reservoirs of infection are not taken.


  Discussion Top


Rhinosporidiosis is extremely rare in the Middle East and literature review with crossref, Pubmed and Google Search shows only one case report from UAE, two reports form Qatar, one report from Kuwait, six cases from Saudi Arabia and one report from Bahrain and Egypt. Practising physicians in UAE may encounter it in expat population from the endemic areas of the Indian subcontinent. From within densely endemic areas, a survey of school children in Pallam, India suggests a disease prevalence of 1.4%.[5]

Rhinosporidiosis was first identified in 1892 by Malbran.[5],[7] Guillermo Seeber in 1900 published the first report of rhinosporidiosis.[5] Ashworth in 1923 described the organism and its life cycle.[7] Karunaratne in 1964 published a complete review 'Rhinosporidiosis in man'.[7],[8]

Nasal and nasopharyngeal rhinosporidiosis is commonly seen in 70% of cases, conjunctivae and associated structures (including lacrimal apparatus) are involved in 15% of cases.[9] Disease affecting skin, ear, larynx, trachea, bronchi, genitals, and rectum has also been described. Disseminated and multisite disease is reported but rare.[10]

The common clinical presentations are nasal obstruction, epistaxis, nasal discharge, or nasal mass.[11] The most common nasal attachment sites include septum, inferior turbinate and inferior meatus in that order.[6] Extranasally, nasopharyngeal, ocular, dermal and laryngeal involvement is seen in that order.[12] The lesions are friable, vascular, pedunculated or sessile polyps, having tiny white surface dots (from spores beneath the epithelium) giving a raspberry or strawberry-like appearance.[2] Although CT PNS scan and magnetic resonance imaging determines disease extent giving a moderate to intense contrast enhancement, biopsy remains the gold standard for definitive diagnosis.[13] In histological sections, the organism is seen in all the stages of development with oval sporangia containing hundreds of endospores being easily identified under the microscope [Figure 2].

Antimicrobial drugs with in vitro anti-rhinosporidial activity used in human and animal are amphotericin B, dapsone, ketoconazole, trimethoprim-sulphadiazine and sodium stibogluconate.[14] Cycloserine was recently found to be effective in 12 trials.[14]

Topical amphotericin B in corneal and nasal rhinosporidiosis has been found to be successful; however, intravenous drug was ineffective in the treatment of disseminated disease.[14] A significant reduction of recurrence rates from 93% to 39% was noted in dapsone untreated and treated patients, respectively.[14] In only one report, systemic ketoconazole, topical clotrimazole and surgery were effective treatment of nasal rhinosporidiosis.[14] Except for their in vitro activity timethoprim-sulphadiazine or sodium stibogluconate have apparently not been used clinically in rhinosporidiosis.[14] Cycloserine has shown some promise in multidrug treatment with other medication used for rhinospoidiosis.[14]

Surgical excision preferably by electrocautery is the recommended treatment.[4] Recurrences are conceivably due to spillage of spores in adjacent mucosa, hence surgical excision with cautery of base followed by adjuvant medical treatment with dapsone has been reported to prevent recurrences.[4],[14] There are no reports of dapsone resistance, the single most commonly used drug treatment for rhinosporidiosis and no new reported treatment innovations since surgery and dapsone were introduced.[14]

In most upper respiratory rhinosporidiosis, the natural history of illness is a couple of years as opposed to ocular or disseminated disease where it may range from 8 to 10 years.[6] Recurrences, usually post-excision are seen in disseminated (100%), upper respiratory (38%) and ocular disease (15%).[6] The history of illness in cases of post-excision recurrences in upper-respiratory rhinosporidiosis is a couple of years, usually at the same site of excision.[15]

Given that natural history of upper respiratory rhinosporidiosis and recurrences post-excision have a duration of illness of a couple of years, this infection in our case after two decades is clearly a reinfection of the disease and not a recurrence.

Hence, reinfection on subsequent visits to an endemic area can happen even after successful treatment and cure from the previous infection 20 years ago as may have happened in our case.


  Conclusion Top


Although rare in the developed world, awareness of rhinosporidiosis is of utmost importance for clinicians in places (like UAE) with a large migrant population that may have also travelled to endemic areas of the disease. The disease can potentially reinfect on visiting endemic areas even after 20 years of previous infection and successful treatment as might have happened in this case.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name 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

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Mathew S, Arora RD, Prabha N, Kamble P, Satpute SS, Nagarkar NM. Retroanalytical Study of Epidemiological Factors of Rhinosporidiosis. Int Arch Otorhinolaryngol 2020;25:e504-e508. doi: 10.1055/s-0040-1718526. PMID: 34737820; PMCID: PMC8558948.  Back to cited text no. 1
    
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Das S, Kashyap B, Barua M, Gupta N, Saha R, Vaid L, et al. Nasal rhinosporidiosis in humans: New interpretations and a review of the literature of this enigmatic disease. Med Mycol 2011;49:311-5.  Back to cited text no. 2
    
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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. 5
    
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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.  Back to cited text no. 6
    
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Kumar S, Mathew J, Cherian V, Rozario R, Kurien M. Laryngeal rhinosporidiosis: Report of a rare case. Ear Nose Throat J 2004;83:568, 570.  Back to cited text no. 7
    
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Karthikeyan P, Vijayasundaram S, Pulimoottil DT. A retrospective epidemiological study of rhinosporidiosis in a rural Tertiary Care Centre in Pondicherry. J Clin Diagn Res 2016;10:MC04-8.  Back to cited text no. 8
    
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Mishra LK, Gupta S, Pradhan SK, Baisakh MR. Lacrimal sac rhinosporidiosis. Plast Aesthet Res 2015;2:353.  Back to cited text no. 9
    
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Agrawal S, Sharma KD, Shrivastava JB. Generalized rhinosporidiosis with visceral involvement; report of a case. AMA Arch Derm 1959;80:22-6.  Back to cited text no. 10
    
11.
Bandopadhyay SN, Jana U, Bandopadhyay G, Majhi TK, Sen S, Das S, et al. Rhinosporidiosis: Various presentations and different sites. Bengal J Otolaryngol Head Neck Surg 2015;23:48-56.  Back to cited text no. 11
    
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Guru RK, Pradhan DK. Rhinosporidiosiswith special reference to extra nasal presentation. J Evol Med Dent Sci 2014;3:6189-99.  Back to cited text no. 12
    
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Prabhu SM, Irodi A, Khiangte HL, Rupa V, Naina P. Imaging features of rhinosporidiosis on contrast CT. Indian J Radiol Imaging 2013;23:212-8.  Back to cited text no. 13
[PUBMED]  [Full text]  
14.
Arseculeratne SN. The chemotherapy of rhinosporidiosis: A review. J Infect Dis Antimicrob Agents 2009;26:21-7.  Back to cited text no. 14
    
15.
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. 15
    


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