Abstract
Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi. It usually presents as sessile or pedunculated granular polyp, red in colour with multiple yellowish pin-head sized dots. Atypical presentations due to involvement of extra-nasal sites may produce diagnostic dilemma. Very high incidence of rhinosporidiosis and that too with atypical presentations in the rural western part of West Bengal, encourages us to undertake this study. The present study was conducted in a tertiary care hospital in the rural western part of West Bengal from July 2013 to December 2016. Patients presenting with rhinosporidiosis in extra-nasal sites and with atypical presentations were included in the study. Rhinosporidiosis confined to nose and patients who lost follow up were excluded from the study. All patients were treated with wide excision and cauterization of base. Among total 114 patients of rhinosporidiosis, 16 had atypical presentations (14.04%). Nine patients (56.25%) presented with a mass hanging in the oropharynx, some mimicking oropharyngeal malignancy. Two patients (12.50%) presented with acute respiratory distress and stridor. One patient (6.25%) presented with disseminated rhinosporidiosis with involvement of the skin, subcutaneous tissue, muscle, bone, penis and urethra. Recurrence was noted in only two patients (12.50%) in nasopharynx. This chronic disease may present with different acute presentations. Proper clinical eye may avoid pre-operative biopsy which may lead to extensive bleeding. Recurrence can be reduced with meticulous and complete removal. Regular post-operative follow-up with endoscopy is must to detect and treat early recurrence.
Keywords: Rhinosporidiosis, Atypical presentations, Disseminated, Lacrimal sac, Tonsil, Larynx, Skin
Introduction
Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi [1]. The most frequent site is nose. Extra-nasal infection involve conjunctiva, lacrimal sac, nasopharynx, oropharynx, skin, urethra, rectum and rarely larynx and even bones [2]. It usually presents as sessile or pedunculated granular polyp, red in colour with multiple yellowish pin-head sized dots. It is painless, friable and bleeds on touch [3]. Atypical presentations due to involvement of extra-nasal sites may produce diagnostic dilemma. The disease is endemic in India and Sri Lanka, with only few cases being reported from Africa, South America and other parts of the world. In India highest number of cases are reported from southern states of Kerala and Tamil Nadu which are on either sides of Western Ghats [4]. We came across a very high incidence of rhinosporidiosis and that too with atypical presentations in the rural western part of West Bengal, which encourages us to undertake this study.
Aims and Objectives
The objectives of our study were to
Study distribution of atypical presentations of rhinosporidiosis according to age, sex and presenting features.
Study distribution of atypical presentations of rhinosporidiosis according to site of origin and compare them with previous reports.
Describe clinical dilemma of atypical presentations of rhinosporidiosis.
Describe the surgical technique to tackle those atypical presentations and reduce the recurrence.
Materials and Methods
The present study was conducted in a tertiary care hospital in the rural western part of West Bengal from July 2013 to December 2016. The study conforms to the declaration of Helsinki and was approved by the Institutional ethical committee. Patients presenting with rhinosporidiosis in extra-nasal sites and with atypical presentations were included in the study. Rhinosporidiosis confined to nose and patients who lost follow up were excluded from the study. Written informed consent was taken from each participant or their guardian. We treated total 119 patients of rhinosporidiosis over 31/2 years. Five patients were lost in follow up, so we excluded them from the study. Among 114 patients 16 had atypical presentation (14.04%) (Figs. 1, 2, 3, 4, 5). A detailed history including age, sex, area of residence, chief complains and personal habits were recorded. Clinical examination including diagnostic nasal endoscopy and fibreoptic laryngoscopy were done to know the site of origin and extent of lesion. CT scan of nose, paranasal sinuses and CT scan of neck where needed were done. All patients were operated under general anaesthesia after doing routine investigations. Two patients presented to emergency with acute respiratory distress and stridor. Emergency tracheostomy was done in both of them. In another two patients having huge rhinosporidiosis in the oropharynx, elective tracheostomy was done prior to removal of mass due to fear of difficult intubation and bleeding. Rhinosporidiosis in nasopharynx and oropharynx were removed trans-orally in tonsillectomy position. Mouth was opened with Boyle–Davis mouth gag with tongue blade. Four handed technique was used for removal of rhinosporidiosis in nasopharynx. Rubber catheter was introduced through nostril to retract soft palate and uvula. With the help of 0° endoscope, placed through the nose, base of the mass in nasopharynx was visualized after gently pulling the mass with Negus tonsillar artery replacement forceps. The mass was removed after cauterization of the base with the help of curved bipolar cautery. Rhinosporidiosis attached to tonsil was removed by doing tonsillectomy using bipolar cautery. Rhinosporidiosis attached to faucial pillars and vallecula was removed by cauterizing the base after pulling them gently with artery forceps. Using an operating microscope with 400 mm focal length lens, laryngeal lesions were removed with proper margins with the help of microlaryngeal instruments. A cotton patty soaked in adrenaline was kept behind the vocal cords to avoid spillage of spores and blood in the subglottic region. The base was cauterized with the help of sucker cum cautery. Rhinosporidiosis of lacrimal sac was removed via subciliary incision. Involved lacrimal sac along with some part of nasolacrimal duct was removed and the surrounding tissue was cauterized. Patient with disseminated rhinosporidiosis was treated with excision of skin lesions, enucleation of subcutaneous lesions, partial amputation of radius with wide margin and partial amputation of penis. Nasal rhinosporidiosis was removed with the help of endoscope after preparing the nose with cotton patty soaked with 4% lidocaine hydrochloride with epinephrine in a ratio of 4:1. Dapsone 100 mg twice daily for 6 months was given in all recurrent cases and cases having multifocal attachments. Patients were followed up with endoscope at 2 weeks, 1, 3, 6 months and 1 year.
Fig. 1.
Photographs of patient serial no 1. a Clinical photograph of cutaneous rhinosporidiosis. b Subcutaneous rhinosporidiosis being operated. c Rhinosporidiosis of radius being removed via partial amputation
Fig. 2.
Photographs of patient serial no 6. a Clinical photograph of rhinosporidiosis in left nostril and oropharynx. b CECT scan of nose and PNS axial section showing rhinosporidiosis in left nasal cavity extending to nasopharynx. c Intra-operative photograph of rhinosporidiosis of left lacrimal sac. d Intra-operative photograph showing huge oropharyngeal rhinosporidiosis being removed
Fig. 3.
Photographs of patient serial no 9. a Rhinosporidiosis attached to left tonsil mimicking malignancy. b CT scan of neck coronal section showing rhinosporidiosis attached to left tonsil. c Intra-operative photograph of rhinosporidiosis attached to left tonsil. d Specimen of rhinosporidiosis along with left tonsil immediately after operation
Fig. 4.
Photographs of patient serial no 11. a Clinical photograph of patient with tracheostomy showing rhinosporidiosis in left nostril, oropharynx. b CECT scan of nose and PNS axial section showing rhinosporidiosis in left nasal cavity extending to nasopharynx. c Intra-operative photograph of oropharyngeal rhinosporidiosis being removed in tonsillectomy position. d Intra-operative photograph of rhinosporidiosis attached to left vocal cord
Fig. 5.
Photographs of patient serial no 16. a Clinical photograph of patient with rhinosporidiosis over right eyelid, left nostril and swelling over left medial canthus. b CECT scan of nose and PNS axial section showing rhinosporidiosis in left nasal cavity extending to left lacrimal sac. c Intra-operative photograph showing removal of rhinosporidiosis from left nasal cavity and left lacrimal sac in continuity
Results
Among total 114 patients of rhinosporidiosis over three and half years period, 16 had atypical presentations (14.04%). Patients were from 7 to 46 years of age, being predominantly in the 2nd and 3rd decades of life (Table 1). Among 16 cases there were 11 males (68.75%) and 5 females (31.25%), with male:female ratio being 2.2:1 (Table 1). All of them came from rural area and gave history of bathing in pond where cattle were also cleaned. Nasal obstruction and nasal discharge (93.75% each) were most common symptoms followed by epistaxis (87.50%), sensation of something coming out from nose (50.00%), foreign body sensation in throat (43.75%), difficulty in swallowing and change in voice (25.00% each), swelling at medial canthus (18.75%), respiratory distress on exertion and acute respiratory distress with stridor (12.50% each), (Table 2). Nose (81.25%) was the most common site of attachment of rhinosporidiosis. Other sites in decreasing order of frequency were oropharynx (43.75%), nasopharynx (37.50%), lacrimal sac (18.75%), larynx and skin (12.50% each), hypopharynx, subcutaneous tissue, bone, penis (6.25% each) (Table 3). There was one case of disseminated rhinosporidiosis with involvement of the skin, subcutaneous tissue, bone, penis and urethra with long-standing primary lesion in the nose (Fig. 1). There were small, non-tender nodules on the face and back. Involvement of the radius presented as almost painless swelling over the wrist joint with restriction of movement. The skin covering the swelling got ulcerated and grew to form a huge fungating ulcero-proliferative mass. The involvement of penis presented as discrete pink polypoidal mass with granular surface, protruding from the urethral meatus involving the glans penis. Seven patients (43.75%) had previous history of excision of rhinosporidiosis. Tracheostomy was needed in total four patients, emergency in two patients and elective in two patients. All patients were treated with wide local excision and electrocautery of the base under general anaesthesia. Severe intra-operative haemorrhage was encountered in two cases (12.50%) where blood transfusion was needed. Histopathological examination of the specimens showed sporangia in different stages of maturation and stroma with predominance of plasma cells, lymphocytes and scarce neutrophils. We noticed recurrence or residual mass in two patients up to 1 year follow-up, both of them were in the nasopharynx. We treated them with endoscopic cauterization. No recurrence was noted in any other site. Synechia was noted in two cases between septum and inferior turbinate and in one case between septum and middle turbinate.
Table 1.
Distribution of patients with atypical presentation, according to age, sex, H/O previous operation and symptoms
| Serial number | Age (in years) | Sex | H/O previous operation | Symptoms |
|---|---|---|---|---|
| 1 | 26 | Male | Once 16 years ago | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 15 years with involvement of the skin, subcutaneous tissue, bone, penis and urethra |
| 2 | 46 | Male | 9 times in last 14 years | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 6 months |
| 3 | 21 | Male | Once 11/2 years ago | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 1 year |
| 4 | 27 | Male | 3 times in last 6 years | Acute respiratory distress and inspiratory stridor, bilateral nasal obstruction and nasal discharge for 2 years, foreign body sensation in throat for 11/2 years, difficulty in swallowing for 1 year, change in voice for 6 months |
| 5 | 11 | Male | No | Right nasal obstruction, nasal discharge and occasional mild epistaxis for 10 months, sensation of something coming out from nose for 6 months |
| 6 | 14 | Female | No | Left nasal obstruction, nasal discharge and occasional mild epistaxis for 1 year, sensation of something coming out from nose and foreign body sensation in throat for 8 months, difficulty in swallowing, change in voice and respiratory distress on exertion for 6 months, swelling near left medial canthus for 6 months |
| 7 | 7 | Male | No | Right nasal obstruction, nasal discharge and occasional mild epistaxis for 7 months, sensation of something coming out from nose for 4 months |
| 8 | 23 | Female | Twice in last 5 years | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 11/2 years |
| 9 | 16 | Female | No | Foreign body sensation in throat for 6 months, difficulty in swallowing for 4 months, change in voice and respiratory distress on exertion for 11/2 months |
| 10 | 15 | Male | Once 21/2 years ago | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 2 years, sensation of something coming out from nose for 8 months, foreign body sensation in throat for 6 months |
| 11 | 25 | Female | No | Acute respiratory distress and inspiratory stridor, left nasal obstruction, nasal discharge and occasional mild epistaxis for 10 years, foreign body sensation in throat for 5 years, sensation of something coming out from nose for 3 years, difficulty in swallowing for 2 years, change in voice for 5 months |
| 12 | 27 | Male | No | Right nasal obstruction, nasal discharge and occasional mild epistaxis for 1 year, swelling near right medial canthus for 8 months |
| 13 | 19 | Female | Once 3 years ago | Bilateral nasal obstruction, nasal discharge and occasional mild epistaxis for 2 years, sensation of something coming out from nose for 9 months |
| 14 | 17 | Male | No | Left nasal obstruction, nasal discharge and occasional mild epistaxis for 3 years, sensation of something coming out from nose and foreign body sensation in throat for 11/2 years |
| 15 | 29 | Male | No | Right nasal obstruction, nasal discharge and occasional mild epistaxis for 11/2 years, foreign body sensation in throat for 1 year |
| 16 | 11 | Male | No | Left nasal obstruction, nasal discharge and occasional mild epistaxis for 9 months, swelling at right lower eyelid and near left medial canthus for 4 months, sensation of something coming out from nose for 4 months |
Table 2.
Distribution of patients according to symptoms
| Symptom | Number of patients | Percentage (%) |
|---|---|---|
| Nasal obstruction | 15 | 93.75 |
| Nasal discharge | 15 | 93.75 |
| Epistaxis | 14 | 87.50 |
| Sensation of something coming out from nose | 8 | 50.00 |
| Foreign body sensation in throat | 7 | 43.75 |
| Difficulty in swallowing | 4 | 25.00 |
| Change in voice | 4 | 25.00 |
| Respiratory distress on exertion | 2 | 12.50 |
| Acute respiratory distress and stridor | 2 | 12.50 |
| Swelling at medial canthus | 3 | 18.75 |
| Skin lesions | 2 | 12.50 |
| Subcutaneous swelling | 1 | 6.25 |
| Bony swelling | 1 | 6.25 |
| Penile mass | 1 | 6.25 |
Table 3.
Distribution of patients with atypical presentation, according to sites of attachment and whether tracheostomy was needed or not
| Serial number | Attachment sites | Tracheostomy needed or not |
|---|---|---|
| 1 | Floor of left nasal cavity, posterior wall of nasopharynx, skin, subcutaneous tissue, bone, penis and urethra (Fig. 1) | No |
| 2 | Floor of left nasal cavity, posterior end of right side of nasal septum and posterior wall of nasopharynx | No |
| 3 | Floor of right nasal cavity, left side of nasal septum, anterior end of left middle turbinate going to oropharynx, soft palate and bilateral faucial pillars | No |
| 4 | Posterior wall of nasopharynx, soft palate, bilateral posterior faucial pillars, vallecula and left vocal cord | Yes, emergency |
| 5 | Floor and inferior meatus of right nasal cavity going to oropharynx | No |
| 6 | Left side of nasal septum and left inferior turbinate going to oropharynx, left lacrimal sac (Fig. 2) | Yes, elective |
| 7 | Floor of right nasal cavity going to oropharynx, soft palate and right posterior faucial pillar | No |
| 8 | Posterior wall of nasopharynx, soft palate and bilateral posterior faucial pillars | No |
| 9 | Left tonsil (Fig. 3) | Yes, elective |
| 10 | Floor and inferior meatus of left nasal cavity, posterior wall of nasopharynx, soft palate and bilateral posterior faucial pillars | No |
| 11 | Floor of left nasal cavity going to oropharynx, soft palate, bilateral posterior faucial pillars, left vocal cord (Fig. 4) | Yes, emergency |
| 12 | Right inferior meatus going to oropharynx and right lacrimal sac | No |
| 13 | Left side of nasal septum, posterior wall of nasopharynx | No |
| 14 | Left inferior turbinate going to oropharynx | No |
| 15 | Posterior end floor of right nasal cavity going to oropharynx | No |
| 16 | Left inferior meatus, left lacrimal sac, right eyelid (Fig. 5) | No |
Discussion
Rhinosporidiosis was first reported from Argentina by Guillermo Seeber in 1900 [5]. O’Kinealy [6] reported the first Indian case from Medical College, West Bengal. The causative organism was considered as a fungus, when Asworth [7] described its life cycle establishing the nomenclature R. seeberi. However, the most accepted hypothesis today is that, R. seeberi belongs to a novel group of fish parasites (Mesomycetozoea) located phylogenitically between fungal and animal divergence [8]. Definite mode of transmission, host and natural reservoirs are largely unknown but the disease is presumed to be transmitted by direct contact with spores through dust, infected clothing or fingers and bathing in stagnant water [9, 10]. This explains the predilection of infection for mucosal sites, where the organism gains access through traumatized epithelium [9]. Ascending infection from the nose is implicated for involvement of the lacrimal sac. In the present study, all patients came from rural area with habit of pond bathing. The most frequent site is nose [2]. The typical look of rhinosporidiosis is usually sufficient for diagnosis [11]. It usually presents as sessile or pedunculated granular polyp, red in colour with multiple yellowish pin-head sized dots. It is painless, friable and bleeds on touch [3]. But extra-nasal infections, involvement of multiple anatomically unrelated sites and delayed presentation make the diagnosis difficult. The present study was compared with previous series (Table 4). Nine patients (56.25%) presented with a mass hanging in the oropharynx mimicking juvenile nasopharyngeal angiofibroma or oropharyngeal malignancy. One patient (6.25%) presented with unilateral tonsillar mass without involvement of any other site. Three patients (18.75%) with rhinosporidiosis of lacrimal sac presented with acute dacryocystitis. Two patients (12.50%) presented with acute respiratory distress and stridor due to laryngeal rhinosporidiosis. Both of them underwent emergency tracheostomy. One of them had history of excision of nasal rhinosporidiosis thrice in last 6 years. Other had rhinosporidiosis in multiple sites including nose for 10 years. Only eight cases of laryngeal rhinosporidiosis had been reported till date [12–18]. There was one patient of disseminated rhinosporidiosis with involvement of the skin, subcutaneous tissue, muscle, bone, penis and urethra with long-standing primary lesion in the nose. Only a few cases of disseminated rhinosporidiosis have been reported [2, 9, 19–21]. Cutaneous rhinosporidiosis may be misdiagnosed as wart, verrucous tuberculosis or granuloma pyogenicum [21]. Rhinosporidiosis of soft tissue and bone are often misdiagnosed as sarcoma or other soft tissue tumours [18]. Dissemination to anatomically unrelated sites is mainly attributed to haematogenous spread. Direct implantation by contaminated nasal secretions either during trauma or surgery can also implant the spores or cause secondary lesions [22]. The treatment of choice of rhinosporidiosis is wide local excision and cauterization of the base. Recurrence is the rule rather than the exception [23]. Literature review suggests a residual or recurrence rate between 10 and 70% [23]. Most of the recurrences are thought to be due to incomplete removal of mass due to excessive bleeding or auto-inoculation by surgical trauma [22]. Measures to control bleeding during operation are hypotensive anaesthesia, decongestion with 4% lignocaine plus 1:2000000 adrenaline in case of nasal cavity, bipolar diathermy with different angulations and malleable suction cum cautery to reach remote area. Different angled endoscopes and microscope help to detect the base of the lesion thereby removing the entire mass under vision with minimal handling of surrounding normal tissue. Blood loss also is less due to limited manipulation of the mass and the operative time is shortened. We need to transfuse blood in only two patients. In the present study we observed recurrence in two cases (12.50%). Both of them were in the nasopharynx. We found that clearance of rhinosporidiosis from nasopharynx is the most difficult job because of multiple attachments and difficulty in exposure and instrumentations. Medical treatment is described in the literature. However it is without useful results when used as the only modality of treatment. Dapsone has been used by some authors as an adjuvant to the surgical treatment to reduce the recurrence of disease. It appears to arrest the maturation of the sporangia and promotes fibrosis in the stroma when used as an adjunct to surgery [24]. We used Dapsone 100 mg twice daily for 6 months for all patients.
Table 4.
Distribution according to anatomical sites compared with other series
| Anatomical site | Allen and Dave [25] (n = 60) | Satyanarayana [26] (n = 255) | Sharma et al. [27] (n = 56) | Saha et al. [28] (n = 98) | Present study (2016) (n = 114) |
|---|---|---|---|---|---|
| Nose and Nasopharynx | 56 | 250 | 51 | 78 | 111 + 6 |
| Oropharynx and tonsil | 0 | 1 + 1 | 0 | 1 | 6 + 1 |
| Hypopharynx | 1 | 3 | 1 | 1 | 1 |
| Larynx | 0 | 0 | 0 | 0 | 2 |
| Trachea | 0 | 1 | 0 | 0 | 0 |
| Conjunctiva | 4 | 0 | 4 | 18 | 0 |
| Lacrimal sac | 0 | 3 | 0 | 2 | 3 |
| Urethra and Penis | 0 | 0 | 0 | 0 | 1 |
| Skin | 0 | 0 | 0 | 1 | 2 |
| Subcutaneous tissue | 0 | 0 | 0 | 0 | 1 |
| Bone | 0 | 0 | 0 | 1 | 1 |
Conclusion
Rhinosporidiosis is a disease of dubious aetiology. Nasal mucosa is the most common site. The typical red, granular friable polypoid appearance may not be evident in extra-nasal rhinosporidiosis making the diagnosis difficult. Proper clinical eye may avoid pre-operative biopsy which may lead to extensive bleeding which is not self-limiting. This chronic disease may also present with acute presentation like respiratory distress, epistaxis or acute dacryocystitis. Recurrence can be reduced with meticulous and complete removal. The population at risk should be educated to avoid bathing in ponds and rivers open to animals. Regular postoperative follow-up with endoscopy is must to detect and treat early recurrence.
Compliance with Ethical Standards
Conflict of interest
The authors declare that they have no conflict of interest.
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