|Year : 2019 | Volume
| Issue : 1 | Page : 84-86
Devendra Tiwari1, Satvinder Singh Bakshi2, Soumyajit Das2, Suriyanarayanan Gopalakrishnan1
1 Department of ENT and Head and Neck Surgery, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth, Puducherry, India
2 Department of ENT and Head and Neck Surgery, All India Institute of Medical Sciences, Guntur, Andhra Pradesh, India
|Date of Web Publication||12-Jun-2019|
Dr. Satvinder Singh Bakshi
House 1A, Selvam Apartments, 71 Krishna Nagar Main Road, Krishna Nagar, Puducherry - 605 008
Source of Support: None, Conflict of Interest: None
Rhinosporidiosis is a rare granulomatous disease of mucous membranes caused by Rhinosporidium seeberi. Although the taxonomy is still debatable, it has recently been concluded that it belongs to a novel group of fish parasite referred to as the “DRIP” clade (Dermocystidium, rosette agent, Ichthyophonus, and Psorospermium). It usually involves the nasal mucosa, but the incidence of extranasal (atypical) sites such as lip, palate, uvula, larynx, trachea, buccal cavity, lacrimal sac, scalp, skin, penis, urethra, vulva, and bone has been reported. Simultaneous involvement of both nasal and extranasal sites, particularly larynx and trachea, is rare. Only a few such cases have been reported, mostly from South India and Chhattisgarh. We report such a case of a 46-year-old male with extensive laryngotracheal rhinosporidiosis, which was managed successfully with surgery.
Keywords: Extranasal site, laryngeal rhinosporidiosis, nasal surgery, rhinosporidiosis
|How to cite this article:|
Tiwari D, Bakshi SS, Das S, Gopalakrishnan S. Disseminated rhinosporidiosis. Arch Med Health Sci 2019;7:84-6
| Introduction|| |
Rhinosporidiosis is a rare chronic granulomatous condition endemic in South Asia. The causative agent is Rhinosporidium seeberi which is taxonomically placed in “DRIP” clade (Dermocystidium, Rosette agent, Ichthyophonus, and Psorospermium) which has been renamed as the Mesomycetozoea. It is more common in males., Although polypoidal nasal mass is the most common clinical manifestation, mass in the nasopharynx and lower aerodigestive tract is also reported. The mainstay of treatment is surgery, but excision of laryngotracheal lesions poses a challenging problem due to its friable nature, location, and recurrence.
| Case Report|| |
A 46-year-old diabetic male complained of bilateral nasal obstruction with progressive hoarseness of voice for 3 months. The patient had a past history of similar illness which was removed surgically and was diagnosed as rhinosporidiosis on histopathology. On anterior rhinoscopy, reddish granular polypoidal masses with yellowish spots were seen, filling both nasal cavities, which bled on touch [Figure 1]. Fiber-optic laryngeal examination showed left false vocal cord hypertrophy with bilateral mobile vocal cords and multiple tracheal satellite lesions [Figure 2]. A preoperative computed tomography neck with thorax revealed a soft-tissue density at the level of the carina and tracheal satellite lesion along with irregularity at the level of the glottis and subglottis. The patient was further evaluated with rigid bronchoscopy under general anesthesia, and the carinal lesion was excised with radiofrequency ablation, followed by radiofrequency-assisted excision of the laryngeal lesions. The nasal and nasopharyngeal lesions were later excised by conventional method. Histopathology revealed characteristic features of rhinosporidiosis, containing many cysts; each of these cysts consists of thick-walled sporangium containing numerous daughter endospores in various stages of development [Figure 3]. The patient was started on dapsone 100 mg once a day for 3 months. The patient was asymptomatic at 4 months of follow-up, with no evidence of recurrence.
|Figure 3: Histopathological picture showing sporangia filled with sporangiospores surrounded by inflammatory cells (H and E, ×40)|
Click here to view
| Discussion|| |
Rhinosporidiosis is a granulomatous disease caused by R. seeberi which belongs to Mesomycetozoea group. Risk factors include bathing or working in contaminated stagnant water harboring the infectious agent. It was thought to be a zoonotic disease, which is transmitted to humans via traumatized epithelium. Endospore penetrates the nasal cavity mucosa and matures into sporangium in the submucosa. No person-to-person transmission is noted. The disease is prevalent mainly in hot tropical climates, though mostly reported in South India and Sri Lanka. According to Sudarshan et al., it is also endemic in Central India in Chhattisgarh.
Rhinosporidiosis is more common in males (M:F = 4:1), with most patients presenting between 10 and 40 years of age. Nasal cavity and nasopharynx are the most common sites of infection, followed by the eye. Occurrence is rare in the larynx and trachea. The laryngeal lesions can be explained by direct inoculation of the organism into the mucosa of the larynx and trachea during episodes of bleeding from the nasal lesions. Nasopharyngeal involvement, particularly in adolescent males, can mimic juvenile angiofibroma and produce a clinical dilemma. Rhinosporidiosis can also rarely affect the lung, urethra, genitalia, lacrimal sac, and bones., Disseminated rhinosporidiosis is seen in immunocompromised patients and can have high mortality. Tracheal involvement in rhinosporidiosis can present diagnostic and therapeutic challenges. This is due to the vascular and friable nature of the mass which makes removal difficult and also compromises with the airway caliber as well as visualization. In the present case, the tracheal lesion was excised first so as to enable securing the airway before excision of the nasal mass.
The treatment for rhinosporidiosis has been a gray area in otolaryngology practice. This is due to high recurrence rates following surgery. The standard treatment is wide local excision with cauterization of the base. The use of CO2 and KTP laser provides a good alternative to achieve a bloodless field but has no additional advantages in the prevention of recurrence. Dapsone has been used for medical treatment with doubtful results.
| Conclusion|| |
Although some cases of nasal and nasopharyngeal rhinosporidiosis are reported from South Asian countries, the incidence of laryngotracheal rhinosporidiosis is rare. There is no standardized treatment protocol. Excision of lesion from these sites is difficult and poses a challenge to the surgeons. The use of novel technology such as lasers can decrease postoperative complications and recurrence.
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 understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3]