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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 31  |  Issue : 3  |  Page : 274-279

Rhinosporidiosis of maxillary sinus: A case report


1 Department of Oral Medicine and Radiology, P. D. U. Dental College, Solapur, Pune, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, M. A. Rangoonwala College of Dental Scicence and Research Center, Pune, Maharashtra, India

Date of Submission05-Jul-2018
Date of Acceptance28-Feb-2019
Date of Web Publication30-Sep-2019

Correspondence Address:
Dr. Abdullah Zakaria Kazi
Department of Oral Medicine and Radiology, P. D. U. Dental College, Solapur - 413 255, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_114_18

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   Abstract 


Rhinosporidiosis is a chronic granulomatous inflammatory disease inducing polyps of the affected mucous membrane and is caused by Rhinosporidium seeberi. Seldom mimicking other more malignant and vigorous pathologies of the involved part. As rhinosporidiosis primarily affects orofacial and sino-nasal region, it is of great value to the oral physician, oral radiologist and oral surgeon and a necessary differential diagnosis to be kept in mind for sino-nasal masses. As the causative organism is an aquatic protistan parasite and cannot be cultured in vitro, the diagnosis depends on clinical and histopathological findings. Treatment is complete surgical excision but recurrence is quite common at 10% to 30%. Use of cryosurgery and electrocautery reduce the recurrence. Also, systemic dapsone therapy is used as an adjunct to surgical intervention and helps in preventing recurrences. In this case report, the clinical, radiological and pathological features of a case presenting in a 55-year old male has been described. The involvement, erosion and destruction of the various walls of maxillary sinus and maxillary osteomyelitis as in this case is very rare and only a few cases have been recorded in the literature. The surgical and pharmacologic management along with 18 months follow up has been discussed and a short review of literature has been made.

Keywords: Rhinosporidiosis, deep fungal infections, opportunistic infections, osteomyelitis


How to cite this article:
Kazi AZ, Kazi HZ, Channe RC, Kulkarni AS. Rhinosporidiosis of maxillary sinus: A case report. J Indian Acad Oral Med Radiol 2019;31:274-9

How to cite this URL:
Kazi AZ, Kazi HZ, Channe RC, Kulkarni AS. Rhinosporidiosis of maxillary sinus: A case report. J Indian Acad Oral Med Radiol [serial online] 2019 [cited 2019 Dec 13];31:274-9. Available from: http://www.jiaomr.in/text.asp?2019/31/3/274/268272




   Introduction Top


Rhinosporidiosis is a rare perplexing disease which has been recognized by the medical fraternity since the beginning of 20th century, following its first description by Sir Guillermo Seeber in Argentina. Seeber comprehensively described it in a 19 year-old farm worker in Buenos Aires whose breathing was impaired by a nasal mass. Seeber proposed that the etiology of the disease was an infection by a fungus, which was later isolated by Ashworth in 1923. Ashworth later described the life cycle of the organism and established the nomenclature Rhinosporidium seeberi.[1] Rhinosporidiosis is a chronic granulomatous inflammatory disease inducing polyps of the affected mucous membrane and recently the isolated organism has been reclassified as an aquatic protistan parasite.[2],[3] Rhinosporidiosis is not a fatal disease unless it affects tracheobronchial tree where it can cause obstruction of airway and lead to death. However, the disease is known to mimic (clinically and radiologically) other more malignant and vigorous pathologies of the involved part.[4] The disease is considered to be endemic in India, Sri Lanka and Brazil and is very sporadic in other countries.[5] The most common sites are nasal mucosa, nasopharynx, oropharynx and nasolacrimal duct.[6] Cutaneous lesions and involvement of other viscera usually occur as a part of disseminated Rhinosporidiosis. Transmission of the disease is not clear but the organism is thought to be contracted from infected soil and water. Habitual bathing in rivers and ponds has been strongly associated with rhinosporidiosis.[7] A statistically significant correlation could be found between male gender and rural residential status and incidence of rhinosporidiosis.[7]


   Case Report Top


A 55 years old male diabetic [Figure 1] was referred to our institute with complaints of pain and pus discharge from right infraorbital region and right maxillary buccal vestibule since 5 months. He had a history of fever, obstructed right nose, hemi-facial palsy of right side, ulceration on right ala of nose, blurred vision from right eye and loosening of his right maxillary teeth.
Figure 1: Portrait photograph of a patient

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On examination, extraoral sinus was present in the right infraorbital region which was communicating with the maxillary sinus [Figure 2]. Intraorally, the buccal cortical plate of right alveolar process of maxilla was grossly destructed, leaving buccal roots of the teeth in the region exposed and the teeth mobile. There was an oro-antral communication in the premolar region on right side, adjacent to which was an ulcer with exposed necrotic bone and debris at its floor [Figure 3]. There was expansion of the hard palate on right side and a sinus with frank pus discharge near the mid palatine raphe in the premolar region [Figure 4]. Electric and thermal (hot) pulp vitality tests were performed and revealed that 13, 14, 15, 17 were non vital.
Figure 2: Sinus with discharge present in the right infraorbital region

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Figure 3: Oro-antral fistula, with areas of exposed necrotic bone and debris

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Figure 4: Swelling on the right side of hard palate and sinus with discharge, near the mid palatine raphe

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A PA Water's projection showed diffuse destruction of the bony walls of right maxillary sinus. Additionally, a CT scan was performed with the provisional diagnosis of Maxillary osteomyelitis secondary to odontogenic infection and was interpreted as an erosive mass of maxillary sinus. There was gross destruction of the walls of the right maxillary sinus, with a soft tissue mass in the sinus [Figure 5] and 6]. There was inflammation and thickening of the nasal mucosa along with detrimental effect on the regional nasal anatomy of right side [Figure 6]. Pus culture with antibiotic sensitivity was done and the results depicted an infection by gram negative bacilli which were highly sensitive to Norfloxacin, Ofloxacin, Tinidazole and Ornidazole. Incisional biopsy taken from the edge of sinus in the right buccal vestibule was inconclusive due to limited access to the core of lesion in the maxillary sinus, and exhibited chronic non-specific inflammation. Excisional Biopsy was done, which demonstrated multiple large irregular bony trabeculae with empty lacunae which were intermingled with fibrous connective tissue [Figure 7], [Figure 8], [Figure 9] which were suggestive of osteomylitis. The surrounding region comprised of moderate inflammatory cells, chiefly lymphocytes and plasma cells. Some areas demonstrated fungal colonies, which appeared to be sporangia, containing ovoid or round endospores that are pathognomonic and are characteristic of Rhinosporidiosis Seeberi [Figure 10].
Figure 5: PA-Waters view, showing the destruction of bony walls of right maxillary sinus (Radiograph is inverted laterally)

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Figure 6: CT Computed Tomography Sections and 3D Reconstruction (a and b), Axial Sections showing destruction of antero-lateral, postero-lateral, and medial walls of right maxillary sinus. Furthermore, increased thickness of the maxillary sinus lining mucosa can be appreciated on the right side. (c and d), Coronal sections showing the destruction of floor of orbit, lateral wall of nose and the zygomatic process of the maxilla on the right side. (e and f), Sagittal sections through the right maxillary sinus. (g and h), 3D reconstruction showing destructive lesion of right maxilla

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Figure 7: Intra- operative

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Figure 8: Excised Specimen

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Figure 9: H and E Photomicrograph (low power) showing empty lacune and chronic inflammatory cells suggestive of Osteomyelitis

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Figure 10: H and E Photomicrograph (low power) showing parasitic colonies, which appeared to be sporangia, containing ovoid or round endospores

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Prior to excision of the lesion patient was placed on Ofloxacin, Ornidazole and analgesics for 7 days. Routine blood work was performed and blood glucose level was monitored 3 days before surgery. Furthermore an elastomeric impression was made for the maxillary arch to facilitate the fabrication of post-surgical maxillary obturator [Figure 11]. The lesion was excised using aseptic techniques and under local anesthesia. The lining of the maxillary sinus was curated and the entire right maxilla was debrided until oozing of fresh blood was observed. It was followed by through irrigation of the region by povidone solution and suturing. Post-surgical obturator and instructions were dispensed to the patient, and recalled after 7 days for suture removal.
Figure 11: Post-surgical Obturator

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Following the diagnosis of Rhinosporidiosis Seeberi, it was decided to start the patient on long term Dapsone therapy to prevent recurrence of the lesion. Regimen was Dapsone 100 mg/day for six months, along with monthly follow up appointments. As of now, 24 months post operatively, the patient has responded well to the therapy and no local or distant recurrences have been observed [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16].
Figure 12: 3 months post-operative

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Figure 13: 18 months post-operative

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Figure 14: 18 months post-operative (another perspective) Kazi, et al.: Rhinosporidiosis of maxillary sinus: A case report Journal

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Figure 15: Final Prosthesis, which is covering the entire defect

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Figure 16: Final Prosthesis

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   Discussion Top


Rhinosporidiosis is an enigmatic disease which has been known to medicine since the beginning of the 20th century, following its first description by Guillermo Seeber in Buenos Aires, Argentina. Guillermo Seeber proposed that the etiology of the disease was infection by a fungus, which was later isolated by Ashworth in 1923, who in turn described the life cycle of the organism and established the nomenclature Rhinosporidium seeberi.[2] Rhinosporidiosis is a chronic infectious granulomatous disease of the upper respiratory tract characterized by the formation of polypoid masses. The organism has been classified as a cyanobacterium or an aquatic protistan parasite, infrequently mimicking other more dreaded and vigorous pathologies of the involved part.[3],[4] Rhinosporidiosis is not a fatal disease unless it involves the tracheobronchial tree where it may impede airway and lead to death. The disease is considered to be endemic in India and Sri Lanka and is very infrequent in other countries.[6] An outbreak that began in 1992 affected 17 individuals in Yugoslavia and was the first recorded epidemic in Europe. All the 17 patients had been exposed to the same source of stagnant water.[8] Habitual bathing in rivers and ponds has been strongly associated with rhinosporidiosis. Rhinosporidiosis affects all ages but is most common in the third and fourth decades. The most common sites are nasal mucosa, nasopharynx, oropharynx, and nasolacrimal duct. The most common symptoms are nasal obstruction, epistaxis, and rhinorrhea. Other sites of involvement may include the larynx, tracheobronchial tree, esophagus, conjunctiva, and ears.[5] Cutaneous lesions and involvement of other viscera usually occur as a part of disseminated disease. Rhinosporidiosis is characterized histologically by the presence of mucosal and submucosal cysts (sporangia) ranging from 10 to 300 μm in diameter. Sporangia contain innumerable endospores seen with hematoxylin-eosin; organisms also stain with Periodic Acid–Schiff, mucicarmine and Gomori methenamine silver stains.[5] Since it is primarily a disease affecting the orofacial region, it is of great value to the oral physician, oral radiologist and oral surgeon, and a necessary differential to be kept in mind for sino-nasal masses. As the causative organism is an aquatic protistan parasite and cannot be cultured in vitro, the diagnosis depends on clinical and histopathological Rhinosporidiosis is primarily a disease findings. Treatment is a complete surgical excision, but recurrence is quite common at 10% to 30%.[9] The use of laser surgery, cryosurgery, coblation, harmonic scalpel and electrocautery reduce the recurrence.[10],[11],[12] Surgery with inadequate hemostasis can cause autoinoculation in adjacent traumatized mucosa, giving rise to disease recurrence. Also, systemic dapsone therapy is used as an adjunct to surgical intervention and helps in preventing recurrences in immunocompetent patients and is known to prevent reoccurrence by arresting the maturation of sporangia and promoting fibrosis in adjacent tissues [7, 13, 14].

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
1 Karunaratne WA. Rhinosporidiosis in man. London: Athlone Press; 1964. Pp. 14-18  Back to cited text no. 1
    
2.
Muhammad MN, Moallam FA. Rhinosporidiosis. Journal of the College of Physicians and Surgeons--Pakistan: JCPSP. 2012 Oct;22(10):671-2.  Back to cited text no. 2
    
3.
Morelli L, Polce M, Piscioli F, Del Nonno F, Covello R, Brenna A, Cione A, Licci S. Human nasal rhinosporidiosis: an Italian case report. Diagnostic pathology. 2006 Dec;1(1):25.  Back to cited text no. 3
    
4.
Roy FH, Fraunfelder FW, Fraunfelder FT, editors. Roy and Fraunfelder's current ocular therapy. Elsevier Health Sciences; 2008. (Section 1 Page 74)  Back to cited text no. 4
    
5.
Weidner N, Cote RJ, Suster S, Weiss LM. Modern Surgical Pathology E-Book. Elsevier Health Sciences; 2009 Jul 8. (Chapter 10). Page 189.  Back to cited text no. 5
    
6.
Sinha A, Phukan JP, Bandyopadhyay G, Sengupta S, Bose K, Mondal RK, Choudhuri MK. Clinicopathological study of rhinosporidiosis with special reference to cytodiagnosis. Journal of Cytology/Indian Academy of Cytologists. 2012 Oct;29(4):246.  Back to cited text no. 6
    
7.
Das S, Kashyap B, Barua M, Gupta N, Saha R, Vaid L, Banka A. Nasal rhinosporidiosis in humans: new interpretations and a review of the literature of this enigmatic disease. Medical mycology. 2011 Apr 1;49(3):311-5.  Back to cited text no. 7
    
8.
Vukovic Z, Bobic-Radovanovic A, Latkovic Z, Radovanovic Z. An epidemiological investigation of the first outbreak of rhinosporidiosis in Europe. The Journal of tropical medicine and hygiene. 1995 Oct;98(5):333-7.  Back to cited text no. 8
    
9.
Thompson LD. Head and neck pathology E-book: a volume in the series: foundations in diagnostic pathology. Elsevier Health Sciences; 2012 Aug 30.  Back to cited text no. 9
    
10.
Nichlani S, Jagade MV, Ganeshan AP, Sayeed S, Borade A. Endoscopic resection of nasal rhinosporidiosis with diode Laser. Bombay Hospital Journal. 2011;53(1):96-8.  Back to cited text no. 10
    
11.
Chery J, Bacskai C, Mendoza E. Recurrent rhinosporidiosis. Journal of Medical Cases. 2014 Jan 28;5(2):58-61.  Back to cited text no. 11
    
12.
Rachmanidou A, Modayil PC. Coblation resection of paediatric laryngeal papilloma. The Journal of Laryngology & Otology. 2011 Aug;125(8):873-6.  Back to cited text no. 12
    
13.
Arseculeratne SN. Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian journal of medical microbiology. 2002 Jul 1;20(3):119.  Back to cited text no. 13
    
14.
Sonkhya N, Singhal P, Mishra P. Naso-oropharyngeal rhinosporidiosis: endoscopic removal. Indian Journal of Otolaryngology and Head and Neck Surgery. 2005 Oct 1;57(4):354-6.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16]



 

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