Rhinosporidiosis in Central India: A cross-sectional study from a tertiary care hospital in Chhattisgarh.

BACKGROUND AND AIMS: Chhattisgarh is an endemic region for rhinosporidiosis infection in Central India. This high prevalence can be explained by suitable hot tropical environment as well as social practice of common bathing of public with animals outdoor, mainly in ponds and river. There are more than 200 ponds in and around Bilaspur district, mainly in Ratanpur region. However, due to limited literature describing rhinosporidiosis from this part of India, it remains neglected and still not recognized as a serious health-care issue and any definite government or private measures/initiatives are not taken to counter this problem.
MATERIALS AND METHODS: It is a retrospective demographic study analyzing 113 consecutive biopsies received from different clinical departments in a tertiary care center with a histological diagnosis of rhinosporidiosis over a period of 7 years from January 2011 to December 2017.
RESULTS: Most of the patients presented in the otorhinolaryngology (86.7%) department, followed by ophthalmology (10.6%). The mean age of the patients was 23.4 years, with a range of 3-70 years. A male-to-female ratio of about 3:2 was noted. Nasal cavity was the most common site of involvement (90/113), followed by conjunctiva (7/113), nasal cavity with oropharynx (6/113), and eyelid (3/113). Patients' complaints showed a varying range from nasal block, nasal mass, bleeding, bleed on touch, difficulty in breathing, and conjunctival mass. Two of the cases presented as recurrent rhinosporidiosis.
CONCLUSION: Rhinosporidiosis is endemic and possesses a great health-care burden in Chhattisgarh. The socioeconomic status, outdoor bathing, and cultural practice in rural areas are the major factors contributing to newer cases. An urgent government initiative to decontaminate public water bodies and public health education through health-care workers and nongovernment organizations is the need of the hour to curtail the incidence of rhinosporidiosis. Copyright:

INTRODUCTION

Rhinosporidiosis is a chronic, granulomatous disease characterized by reddish mulberry-like polypoidal masses typically involving nasal cavity and nasopharynx. Other sites which are reportedly involved include maxillary sinus, ethmoid sinus, conjunctiva, lacrimal gland, oral cavity, and genitalia.[1] It is caused by a protist Rhinosporidium seeberi. Tropical hot climate is very favorable for this organism. Hyperendemicity is noted in Sri Lanka and South India. However, it is also highly prevalent in Central India, particularly Chhattisgarh state, due to hot tropical weather, large number of artificial water reservoir as well as social and cultural practice.[2] The organism flourishes in stagnant water of ponds, tanks, well, and river. It not only affects humans but also infects primates, mainly livestock. Transepithelial infection through breached epithelium is the most accepted mechanism proposed for its transmission.[3] Herein, we are describing the second largest regional cross-sectional study from a tertiary care center in Bilaspur district, Chhattisgarh, Central India, mainly pertaining to the sociodemographic profile and clinical presentations in rhinosporidiosis patients with review of literature.

MATERIALS AND METHODS

This is a retrospective demographic study analyzing 113 consecutive biopsies received from different clinical departments in a tertiary care center with a histological diagnosis of rhinosporidiosis over a period of 7 years from January 2011 to December 2017. Most of the patients presented in the otorhinolaryngology department, followed by ophthalmology, dentistry, and general surgery. A detailed clinical, social, and environmental history was taken with special emphasis to bathing habits. Routine examinations including hemogram, X-ray, blood grouping, and viral markers (hepatitis C, hepatitis B, and HIV) were performed in each patient. After preanesthetic clearance, all the patients were subjected to excision of the mass/polyp with electrocauterization of base. All the tissues submitted in 10% formalin in the department of pathology for histopathological examination. Sections of 5-μ thickness were stained in hematoxylin and eosin stain for microscopic examination.

RESULTS

Of the 113 cases, majority of the patients presented in the otorhinolaryngology (86.7%) department, followed by ophthalmology (10.6%), dentistry (1.7%), and general surgery (0.8%). The mean age of the patients was 23.4 years, with a range of 3–70 years. A male-to-female ratio of about 3:2 was noted. The site-wise distribution of cases [Table 1] showed the highest number of cases in nasal cavity with nasal block as the most common presenting symptom. Other clinical presentations are summarized in Table 2. In most of the cases, clinical diagnoses were straightforward; however, in some cases, other differentials such as hemangioma, antrochoanal polyp, angiofibroma, granulomatous inflammation, and malignant lesion were also considered. Two of the cases presented as recurrent rhinosporidiosis.

Table 1

Site-wise distribution of the rhinosporidiosis cases in the study

Site	Number of cases
Conjunctiva	7
Eye (orbit)	1
Eyelid	3
Lacrimal gland	1
Cervical lymph node	1
Nasal cavity	90
Nasal cavity with oropharynx	6
Nasal cavity with ethmoid sinus	1
Nasal cavity with ear	1
Nasal cavity with eyelid	1
Palate	1
Total	113

Table 2

Summary of the clinical presentation of the patients of rhinosporidiosis

Symptoms	Number of cases
Bleed on touch	14
Bleeding	19
Nasal block	40
Nasal block and bleeding	2
Nasal blocking and difficult breathing	3
Bleed on touch and difficult breathing	1
Difficulty in breathing	2
Difficulty in swallowing	1
Nasal mass	21
Eyelid mass	2
Eyelid swelling	1
Conjunctival mass	4
Orbital cellulitis	1
Cervical lymphadenopathy	1
Nasal pain	1
Total	113

DISCUSSION

Rhinosporidiosis was previously considered as a fungal disease endemic in some parts of world. However, currently, it is considered as protist classified under Mesomycetozoea. Stagnant contaminated water in artificial as well as natural water bodies serves as a main reservoir of the organism.

Studies by Kutty et al. and Guru and Pradhan in their series reported that the maximum number of cases lies in the age group of 21–30 years.[45] However, in this study, the highest number of cases (43.3%) was represented in 11–20 years' age group, followed by 28.3% of cases in 21–30 years' age group. This can be explained by frequent engagement of young children in outdoor bathing in rural areas. A higher male prevalence of 1.5:1 was noted which is similar to the previous reports of Karthikeyan et al.[3] and Arseculeratne et al.[6] Satyanarayana suggested that estrogen in females might be protective against infection by R. seeberi.[7] However, the cultural and social practice of lesser exposure of females for bathing in outdoor water bodies may be an important factor for low level of occurrence of disease.

Krishnamoorthy et al. in their article claimed a preliminary report of culture of R. seeberi from nasal swab of a patient.[8] This explains that droplet infection can also act as an important route of infection for transmission of the organism from person to person in crowded, poor hygiene communities among developing countries. Furthermore, the soil and sand near the water bodies such as river and pond is contaminated with the organism and its spores lies remain dormant which becomes active in contact with living tissue. Hence, the source of infection can be contaminated water, soil as well as air droplets.

In the literature, different routes of spread in the body are described for R. seeberi including contiguous spread, autoinoculation, lymphatic,[9] and hematogenous.[10] The contiguous spread of disease is explained by simultaneous involvement of close proximity sites such as nasal cavity to different sinuses, oropharynx, and conjunctiva. Although systemic dissemination is very uncommon, few case reports describing widespread subcutaneous nodular lesions and involvement of lungs are reported.[1112]

Grossly, the typical lesion of rhinosporidiosis is hyperplastic, fleshy, reddish granular, polypoidal, sessile/pedunculated mass and studded with yellowish-white pinhead dots representing sporangia over the surface of lesion [Figure 1].[13] Microscopically, it shows large (100–450 μm), thick-walled chitinous sporangia filled with innumerable numbers of endospore, each measuring about 6–10 μm, accompanied by a mixed inflammatory infiltrate [Figure 2]. In addition, frequent squamous metaplasia is also noted in the overlying lining epithelium. A granulomatous response occurs in case of rupture of the sporangia.

Figure 1

Representative clinical images of the rhinosporidiosis cases: (a) a patient with a typical red mulberry-like polypoidal mass in the left nasal cavity, (b) a young patient with oropharyngeal rhinosporidiosis, (c) A case of conjunctival rhinosporidiosis

Figure 2

Representative histological images of rhinosporidiosis: (a) multiple sporangia filled with innumerable number of endospores and granulomatous reaction including multiple foreign body-type giant cells (H and E, ×100), (b) chronic inflammatory cell infiltrate surrounding sporangia and comprising mainly of plasma cells and lymphocytes (H and E, ×100), (c) necrotic tissue around spores (H and E, ×100), (d) skin involvement by rhinosporidiosis (H and E, ×100)

Gomori's methenamine silver stain, periodic acid–Schiff stain, and Verhoeff–Van Gieson stain can be used to further highlight endospores and spherules. Histological differentials considered are cryptococcosis, Coccidioides immitis, Blastomyces dermatitidis, and myospherulosis.

Although the typical look of rhinosporidiosis, a fleshy mulberry-like mass with dotted sporangia, is usually sufficient to arrive at a clinical diagnosis, delayed presentation and extranasal involvement of multiple anatomically unrelated sites sometimes make the diagnosis difficult.[14] The clinical presence of typical nasal cavity lesion, with multiple lesions in the other parts of respiratory tract or elsewhere in the body is enough to make a correct diagnosis.

A remarkable variation is reported in the postoperative recurrence in different studies which may be attributed to many factors such as operative procedure, type of instrument (cautery/laser) used for excision, site of involvement, and re-exposure to the organism. Although a large body of literature exists regarding this problem, postoperative recurrence rates continue to vary, and the effect of public health education in the eradication of this disease has not been taken into account.[15]

More importantly, this study highlights the effect of public health education regarding susceptible habits in the transmission of rhinosporidiosis, which has helped reduce the incidence of this disease in the previously endemic communities of Pondicherry region.[16]

Diathermy excision is considered to be the treatment of choice. Diathermy excision is an acceptable treatment modality in cases of nose and nasopharyngeal presentations. However, when there is involvement of the tracheobronchial tree, a more radical approach may be considered. Now, as LASER is available at some centers, the choice of treatment may be cauterization with LASER through a bronchoscope.[17] However, LASER is still not freely available in India and Sri Lanka where rhinosporidiosis is endemic.

CONCLUSION

Rhinosporidiosis is an infectious disease with a high prevalence in tropical hot region of Central India. It is strongly associated with farming and animal husbandry in rural part of Chhattisgarh where outdoor public bathing is a common practice. Bilaspur district is one of the hot spots due to large number of contaminated ponds and water bodies. A concrete urgent government-aided public health awareness program is the need of the hour in this region to combat with rhinosporidiosis.

Limitations

It is a cross-sectional epidemiology study highlighting the regional prevalence of rhinosporidiosis, clinical presentation, local factors, and pathogenesis. However, no water sampling or any microbiological/genetic analysis is performed.

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.