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Table of Contents
CASE REPORT
Year : 2011  |  Volume : 1  |  Issue : 1  |  Page : 30-32

A rare case of laryngeal rhinosporidiosis


Department of ENT, Pt JNM Medical College, Raipur, Chhattisgarh, India

Date of Web Publication7-Feb-2011

Correspondence Address:
Digvijay Singh
Sr. MIG-17, Sector-4, Pt. Deendayal Upadhyay Nagar, Raipur, Chhattisgarh - 492010
India
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DOI: 10.4103/2230-9748.76135

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   Abstract 

Rhinosporidiosis is chronic granulomatous disease of likely fungal etiology which usually affects the nasal mucosa. Extranasal manifestations of rhinosporidiosis are relatively uncommon. Laryngeal and tracheobronchial tree involvement is extremely rare and only six cases have been reported till date. Here we report a case of laryngeal rhinosporidiosis, which occurred in a patient with coexisting nasal rhinosporidiosis and was successfully managed surgically.

Keywords: Rhinosporidiosis, Larynx


How to cite this article:
Daharwal A, Banjara H, Singh D, Gupta A, Singh S. A rare case of laryngeal rhinosporidiosis. J Laryngol Voice 2011;1:30-2

How to cite this URL:
Daharwal A, Banjara H, Singh D, Gupta A, Singh S. A rare case of laryngeal rhinosporidiosis. J Laryngol Voice [serial online] 2011 [cited 2014 Aug 22];1:30-2. Available from: http://www.laryngologyandvoice.org/text.asp?2011/1/1/30/76135


   Introduction Top


Rhinosporidiosis is chronic granulomatous disease of likely fungal etiology which usually affects the nasal mucosa. Laryngeal rhinosporidiosis is rare. Here we present a case of 36-year-old male who had recurrent nasal and nasopharyngeal rhinosporidiosis and found to have left vocal cord involvement. The occurrence at vocal cord poses a challenging problem for excision due to its location, bleeding, and high tendency to recurrence. Only six cases had been reported in the world literature, all of which have occurred in India.


   Case Report Top


A 36-year-old male patient presented with a 4-month history of change in voice. The patient had been diagnosed with nasal rhinosporidiosis 3 years earlier for which he had underwent excision of rhinosporidiosis mass and cauterization of base with diathermy twice under general anesthesia for which orotracheal intubation was done.

On laryngoscopic examination a pinkish mass was noted on the left vocal cord posteriorly. On videostroboscopic examination, the pinkish granular mulberry like mass was noted at posterior one third of left vocal cord on its superior surface, which did not move with respiration [Figure 1]. Right vocal cord was normal.
Figure 1: Photograph showing pinkish mulberry like rhinosporidiosis mass (in circle) at posterior third of left vocal cord

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Due to its laryngoscopic appearance and previous history of nasal rhinosporidiosis, a provisional diagnosis of laryngeal rhinosporidiosis was made. After premedical and preanesthetic checkup the patient was posted for microlaryngeal surgery excision under general anesthesia. The patient was intubated orally with a 6 mm internal diameter cuffed tube. Utmost precaution was taken to avoid injury to the involved site while intubation. The patient was positioned and the laryngoscope fixed, rest of the procedure was done under an operating microscope with 400 mm focal length lens. A cotton patty was kept behind the vocal cords to avoid spillage of spores and blood in the subglottic region. Mass was excised with proper margins with the help of microlaryngeal instruments. Very minimal bleeding observed which was well controlled by the adrenaline-soaked cotton patties. Cauterization of the base of the lesion with diathermy was not considered. The patient received oral antibiotics and steroidal drugs for 1 week.

On histopathological examination, the lesion showed the characteristic features of the rhinosporidiosis containing many globular cysts; each of these cysts represented a thick-walled sporangium containing numerous daughter spores in different stages of development [Figure 2]. The patient did not receive any drug therapy and on close follow up. At 11 months of follow up there is no sign of recurrence [Figure 3]. Patient's voice is normal.
Figure 2: Histopathological picture of rhinosporidiosis (H & E, 40×)

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Figure 3: Photograph showing site of lesion (in circle) after excision of mass at 11 months of follow up.

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   Discussion Top


The taxonomic relationship of Rhinosporidium seeberi with other organisms remained controversial for more than a century. Recently, molecular studies have suggested R. seeberi to be a protistal microbe in the newly described class Mesomycetozoea which lies between animals and fungi. Invasion by this fungus is universal, but it is endemic in India and Sri Lanka. [1]

Rhinosporidiosis mostly affects adult men and is possibly transmitted to human subjects by means of direct contact with spores of R. seeberi through dust, infected clothing, or fingers and through swimming in stagnant water contaminated with the spores. [2]

Rhinosporidiosis predominantly affects the mucous membranes of the nose and nasopharynx; it also occasionally involves the lips, palate, uvula, maxillary antrum, conjunctiva, lacrimal sac, epiglottis, larynx, trachea, bronchus, ear, scalp, skin, penis, vulva, and vagina. [3]

In the present case, the primary lesion was in nose and nasopharynx, for which the patient had undergone excision and cauterization of base twice. Even though endotracheal intubation was done for previous surgeries, in the present case laryngeal involvement could be due to the result of trauma and implantation of the spores during intubation or during episodes of bleeding for previous operation.

Only six cases of laryngeal rhinosporidiosis had been reported till date, all of which had occurred in India. [4],[5],[6],[7],[8]

In the first report, published in 1974, Pillai described two patients who presented with nasal and laryngeal masses. Both were excised under local anesthesia. [4]

In 2004 Kumar et al. described a new case of laryngeal rhinosporidiosis, which occurred in a patient with coexisting nasal rhinosporidiosis who presented with inspiratory stridor. Both lesions were completely excised under general anesthesia with a CO 2 laser without the need for preliminary tracheostomy. [6]

Recently Mathew et al. reported a case of laryngeal rhinosporidiosis and highlighted their technique in the management of the airway using a combination of fiberoptic bronchoscope and an endoscope. The technique was atraumatic and also avoided the potential for autoinoculation, which is a frequent cause of recurrence of this disease. [7]

Madana et al. reported a case of nasal rhinosporidiosis with progressive hoarseness of voice for 1 year. Fiberoptic laryngoscopic examination revealed reddish mass with whitish surface specks, involving the glottis, subglottis, and trachea. Direct laryngoscopic and rigid bronchoscopic-guided excision of the laryngeal and tracheal lesions were performed. [8]

In the present case, due to unavailability of laser, microlaryngeal surgery was planned. The patient was intubated orally with a 6 mm internal diameter cuffed tube. Mass was excised with proper margins in totality. Very minimal bleeding was observed which was well controlled by the adrenaline-soaked cotton patties. Base cauterization with diathermy was not considered being very close to the vocal cords. No recurrence was noted in 1 year follow up.

The mainstay of treatment for nasal and nasopharyngeal rhinosporidiosis is surgical excision by laser or electric diathermy. In a study done by Kameswaran et al. on management of nasal and nasopharyngeal rhinosporidiosis, usage of KTP-532 laser showed numerous advantages over the conventional treatment modality. It enabled the surgeon to obtain a better clearance margin with improved visibility and also significantly decreased total amount of blood loss. It also helped in reducing the chances of recurrence. This could also attribute to the fact that treatment with laser can be achieved without direct contact of infected granulomata thus avoiding tissue contamination. [9]

Medical treatment has been proposed by some authors for the prevention of recurrence, but the results are not convincing. The only drug having clinical promise is Dapsone. Antimicrobial therapy is ineffective and the disease may recur after months and years. [10],[11],[12] In the present case, the patient did not receive any drug therapy for prevention of recurrence.

Treatment standards have not been established for the laryngeal rhinosporidiosis, owing to the rarity of these lesions though surgery remains the mainstay of treatment. Usage of a CO 2 /KTP laser could be a safe and effective substitute for conventional microlaryngeal surgery due to its advantages of less chances of trauma, avoids implantation of spores, and preservation of good voice. However, conventional microlaryngeal surgery may suffice for small lesions as in the present case.


   Conclusion Top


Laryngotracheal involvement of rhinosporidiosis poses many diagnostic and therapeutic challenges, due to the potential risk of bleeding, aspiration, and recurrence. Surgery is the mainstay of treatment and can safely be carried out using microlaryngeal surgery after expert intubation. To the best of our knowledge and literature review, laryngeal rhinosporidiosis is a very rare case and this is the seventh case reported in the world literature.

 
   References Top

1.Silva V, Pereira CN, Ajello L, Mendoza L. Molecular evidence for multiple host-specific strains in the genus Rhinosporidium. J Clin Microbiol 2005;43:1865-8.  Back to cited text no. 1
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2.Thappa DM, Venkatesan S, Sirka CS, Jaisankar TJ, Gopalkrishnan, Ratnakar C. Disseminated cutaneous rhinosporidiosis. J Dermatol 1998;25:527-32.  Back to cited text no. 2
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3.Kerr AG, Scott-Brown's Otolaryngology. Oxford: Butterworth-Heinemann; 1997:39-40  Back to cited text no. 3
    
4.Pillai OS. Rhinosporidiosis of the larynx. J Laryngol Otol 1974; 88:277-80.  Back to cited text no. 4
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5.Banerjee SB, Sarkar A, Mukherjee S, Bhowmik A. Laryngeal rhinosporidiosis. J Indian Med Assoc 1996;94:148-50.   Back to cited text no. 5
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6.Kumar S, Mathew J, Cherian V, Rozario R, Kurien M. Laryngeal rhinosporidiosis: Report of a rare case. Ear Nose Throat J 2004;83:568-70.  Back to cited text no. 6
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7.Mathew JS, Padhy S, Lata S, Balachander H, Gopalakrishnan S. Case report: Telelaryngoscopy-guided flexible fiberoptic intubation for laryngeal rhinosporidiosis. Anesth Analog 2010;110:1066-8.  Back to cited text no. 7
    
8.Madana J, Yolmo D, Gopalakrishnan S, Saxena SK. Rhinosporidiosis of the upper airways and trachea. J Laryngol Otol 2010:1-3.  Back to cited text no. 8
    
9.Kameswaran M, Anand Kumar R S, Murali S, Raghunandan S, Jacob J. KTP-532 laser in the management of rhinosporidiosis. Indian J Otolaryngo Head Neck Surg 2005;57:298-300.  Back to cited text no. 9
    
10.Nair KK. Clinical trial of diaminodiphenylsulfone (DDS) in nasal and nasopharyngeal rhinsporidiosis. Laryngoscope 1979;89:291-5.  Back to cited text no. 10
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11.Job A, Venkateswaran S, Mathan M, Krishnaswami H, Raman R. Medical therapy of rhinosporidiosis with dapsone. J Laryngol Otol 1993;107:809-12.  Back to cited text no. 11
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12.Venkateswaran S, Date A, Job A, Mathan M. Light and electron microscopic findings in rhinosporidiosis after dapsone therapy. Trop Med Int Health 1997;2:1128-32.  Back to cited text no. 12
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    Figures

  [Figure 1], [Figure 2], [Figure 3]


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