[Human rhinosporidiosis of the nasal cavity in Argentina]. / Rinosporidiosis humana de cavidad nasal en Argentina.

Rhinosporidiosis is an infectious pathology caused by the aquatic protist pathogen Rhinosporidium seeberi that inhabits the fresh water of lakes or lagoons and in the soil. It is extremely rare, being endemic in India and Sri Lanka, although cases have been seen in countries in Europe, Africa and South America. It predominates in young males and affects both humans and animals. It is characterized by the formation of uni- or bilateral reddish polyps, mainly in the nasal cavity. Presenting airway symptoms include obstruction, epistaxis, rhinorrhea and foreign body sensation. We report the case of a 22-year-old man from Argentina who presented with nasal ventilatory insufficiency, rhinorrhea and epistaxis. Rhinoscopy revealed a polypoid formation arising from the septal septum and occupying the entire right nostril. Microscopically, the polyp was covered by respiratory and squamous epithelium, and at the stromal level it evidenced a granulomatous chronic inflammatory process with the presence of abundant thick-walled cystic structures between 100 and 500 υm (sporangia), which contained abundant endospores inside. The periodic acid Schiff and Grocott techniques highlighted these structures. Given the clinical-epidemiological background and the morphological picture, the diagnosis of rhinosporidiosis was reached.

La rinosporidiosis es una enfermedad infecciosa causada por el patógeno protista acuático Rhinosporidium seeberi que habita en agua dulce de lagos o lagunas y el suelo. Es sumamente infrecuente, siendo endémica en India y Sri Lanka, aunque se han visto casos en países de Europa, áfrica y Sudamérica. Predomina en jóvenes de sexo masculino y afecta tanto a humanos como animales. Se caracteriza por la formación de pólipos rojizos uni o bilaterales, principalmente en cavidad nasal. Los síntomas de presentación en la vía aérea incluyen obstrucción, epistaxis, rinorrea y sensación de cuerpo extraño. Reportamos el caso de un varón de 22 años nativo de Argentina que se presentó a la consulta por insuficiencia ventilatoria nasal, rinorrea y epistaxis. La rinoscopía exhibió una formación polipoide que surgía del tabique septal y ocupaba toda la fosa nasal derecha. Microscópicamente el pólipo estaba revestido por epitelio de tipo respiratorio y pavimentoso y a nivel estromal evidenciaba proceso inflamatorio crónico granulomatoso con presencia de abundantes estructuras quísticas de pared gruesa de entre 100 y 500 υm (esporangios), que contenían en su interior abundantes endosporas. Las técnicas de ácido peryódico de Schiff y Grocott resaltaron dichas estructuras. Dado los antecedentes clínicoepidemiológicos y el cuadro morfológico se arribó al diagnóstico de rinosporidiosis.

Rhinosporidiosis of the lacrimal sac in a tertiary care hospital of India - A retrospective case study.

Purpose: Though rhinosporidiosis of the lacrimal sac is a rare disease across the globe, the frequency with which these patients come to the outpatient department in western Odisha is quite alarming. This study was undertaken to upgrade the knowledge about the clinical profile and management of rhinosporidiosis of the lacrimal sac. Methods: This is a retrospective study comprising 32 clinically diagnosed and histopathologically proved cases of lacrimal sac rhinosporidiosis who were managed with dacryocystectomy with meticulous excision. Intraoperative copious irrigation with 5% povidone-iodine for 5 min and postoperative dapsone therapy for 3-6 months had been administered to all the patients. The mean follow-up period was 16.7 months. The study was conducted over 5 years from August 2015 to July 2020. Results: Rhinosporidium seeberi, an aquatic protistan parasite, was found to be the causative agent. Males and females were affected equally. Children less than 10 years of age comprised 56.2% (18 cases). History of pond bathing was found in 100% of cases. The most common presentation was boggy swelling over the lacrimal sac. The involvement was unilateral in all the cases. None of the patients were found to have nasal involvement. In 65.6%, the lesion was limited within the sac. Recurrence was noted in 25% of cases. Conclusion: Rhinosporidiosis of the lacrimal sac should be excluded in all patients presenting with boggy swelling of the lacrimal sac with a history of pond bath. The recurrence can be minimized by meticulous excision, intraoperative betadine, and postoperative dapsone therapy.

Equine rhinosporidiosis in Buenos Aires, Argentina.

Rhinosporidiosis is caused by Rhinosporidium seeberi, a parasitic organism of the family Rhinosporideacea family, class Micomycetozoa. The disease is endemic in India; however, some cases were reported in Europe, Africa, North America, and South America. The aim of the present study is to report three cases of rhinosporidiosis in wild horses in different cities of Buenos Aires province, Argentina. We confirm the presence of R. seeberi in the analyzed samples using histopathological and PCR sequencing techniques.

Rhinosporidiosis-Factors predicting disease recurrence.

BACKGROUND: Rhinosporidiosis is a chronic granulomatous disease of the nose caused by Rhinosporidium seeberi. The disease is largely non-amenable to medical therapy and shows high recurrence rates requiring patients to undergo multiple surgeries often resulting in increased morbidity. OBJECTIVE: To analyse the epidemiological, clinical, histopathological characteristics, treatment and outcome in rhinosporidiosis and to identify factors which predispose to recurrence of the disease. PATIENTS/METHODS: Retrospective analysis of data of all patients with a diagnosis of rhinosporidiosis confirmed by histopathology at a tertiary care hospital from 2015 to 2019. RESULTS: There were 42 patients, 40 males and two females, with a mean age of 37.37 years. Disease showed bilateral involvement in 17 (40.48%) patients. Nineteen (45.24%) patients had more than two sites involved at initial presentation. Most patients had nasal cavity involvement followed by nasopharynx. Among the 28 patients who had a follow-up, 12 showed recurrent disease. However, 21 patients were disease free following a revision excision. Involvement of more than two sites was an independent significant factor for recurrence. On univariate analysis, other factors which showed statistically significant odds of developing recurrence were previous surgery (p = .054), involvement of nasal septum (p = .022), middle turbinate (p = .024), nasopharynx (p = .049) and posterior pharyngeal wall (p = .05). Factors which showed significantly less likelihood of developing a recurrence included patients who had less than 12 months duration from first symptom to intervention (p = .016), involvement of less than two sites (p = .0003) and unilateral disease (p = .019). CONCLUSION: Early intervention in rhinosporidiosis especially when the disease is unilateral and involves less than two sites improves the outcome.

Rhinosporidiosis in the Americas: A Systematic Review of Native Cases.

Rhinosporidiosis is a chronic mucosal infection caused by Rhinosporidium seeberi, an aquatic protistan parasite. It presents as nasal or ocular polypoidal or vascularized masses. It is endemic in tropical and subtropical areas, especially in South Asia; R. seeberi´s endemicity in the Americas is often overlooked. The objective of this study was to describe the demographic and clinical characteristics of patients with rhinosporidiosis in the Americas, its management, and patient outcomes. This study is a systematic review of cases of human rhinosporidiosis in the Americas reported in the literature from 1896 to February 28, 2019. This review screened 1,994 reports, of which 115 were eligible for further analysis. The selected reports described 286 cases of human rhinosporidiosis between 1896 and 2019. Cases were diagnosed in Brazil (32.2%), Colombia (24.4%), Paraguay (12.6%), and the United States (11.9%). The majority of the cases (91%) occurred in geographic areas with altitudes < 1,000 m above sea level and in areas with median temperatures ≥ 25°C (67.3%). Most of the patients presented nasal (65%) and ocular involvement (35%). Surgical treatment was provided for 99.6% of patients, but 19.8% of them recurred. This review describes the under-recognized geographic distribution and clinical presentation of rhinosporidiosis in the Americas and highlights clinical differences to cases in Asia, specifically in reference to a higher prevalence of ocular disease and higher relapse rates.

Rhinosporidium seeberi: Is It a Fungi or Parasite?

Rhinosporidium seeberi (R. seeberi) causes rhinosporidiosis, which is manifested as tumor-like polyps developing primarily in the nostrils and conjunctiva in human and animals. This disease is characterized by the presence of large, round-shaped mature stage and small endospores with resistance to culturing. R. seeberi was first reported in 1900 as a sporozoan parasite, but later classified as a lower fungi, although its morphological similarity with aquatic parasites were also noticed. According to 18S small-subunit ribosomal DNA sequencing, R. seeberi belongs to a group of fish parasite DRIP clade located between the animal and fungal divergence. Histological examination is thus necessary for the definitive diagnosis of rhinosporidiosis, and the first line of treatment is usually total surgical excision and electro-cauterization of the polyp base. Among the drug therapies attempted, remission has been reported in some patients who received only Dapson treatment. This disease is endemic across India, Pakistan and Sri Lanka and occurs sporadically in other parts of The World with a common history of patients bathing in stagnant water. An outbreak in Serbia during 1992-1995 and 5 rhinosporidiosis cases from Turkey have been reported until date. Considering that rhinosporidiosis is associated with exposure to water and the agent belongs to a branch of aquatic parasites, it has been proposed that aquatic animals are the natural hosts and that the mammalian hosts acquire infection by contacting contaminated water. Therefore, there is a need for the investigation of the infection in fish besides mammalian animals as reservoirs as well as to conduct screening of antiparasitic drugs with infected fish or infected cell lines with the nearest phylogenetic relatives of R. seeberi.

Evaluation of diagnosed cases of eye rhinosporidiosis in a public hospital of Maranhão, Northeast Brazil.

BACKGROUND: Oculosporidiosis (ocular rhinosporidiosis) accounts for 15% of cases of rhinosporidiosis, which is a chronic granulomatous disease and is endemic in India and Sri Lanka. In Brazil, the climatic and hydrographic similarities to these endemic areas and the presence of riverside populations contributes to an increase in the incidence of rhinosporidiosis particularly in the State of Maranhão. This study, therefore, aimed to identify the number of diagnosed cases of oculosporidiosis and describe its the clinical epidemiology, laboratory, histopathology, and therapeutic characteristics. METHODS: The study is descriptive, observational, and cross-sectional, and reports the prevalence and clinical epidemiological characteristics of oculosporidiosis in the State of Maranhão, Brazil. A retrospective analysis of the paper and electronic records for a period from 1999 to 2017 was conducted in the University Hospital of Federal University of Maranhão (HU-UFMA), located in the northeastern region of Brazil. RESULTS: Thirty patients were diagnosed with rhinosporidiosis, eight of them had oculosporidiosis and seven of these met the criteria to be included in the study. Of the cases (23.3% of all 30), five were men (71.4%) and two women (28.5%), with an average age of 16.4 ± 15.6 years. In terms of race, four patients (57.1%) declared themselves white and three (42.9%) as brown. The north of the state, the mesoregion, had the most diagnosed cases accounting for 57.1% of the total. Left eye was the most affected site, reported in six patients (85.7%), while the conjunctiva was affected in all patients. Rhinosporidiosis and papilloma were the predominant diagnostic hypotheses (28.5 and 28.5%, respectively), followed by chronic scleritis, granuloma, and chalazion (14.25, 14.25, and 14.25%, respectively). All these cases were treated with lesion excision, and only two patients (28.5%) progressed with recurrence. CONCLUSION: It was verified that there was a male predominance, with only one eye reported as an infected site, with no bilateral involvement. The younger age group (between 1 and 2 years of age) was more affected by oculosporidiosis, and histopathological examination was necessary for a conclusive diagnosis.

Cluster of Nasal Rhinosporidiosis, Eastern Province, Rwanda.

We report 4 recent cases of nasal rhinosporidiosis in Rwanda. All patients were boys or young men living in the same district (Gatsibo District, Eastern Province), suggesting a reservoir in the area. The recent reemergence of rhinosporidiosis in Rwanda might reflect increased availability of diagnostic services rather than emerging disease.

Lacrimal Sac Rhinosporidiosis and Surgical Management by Transnasal Endoscopic Excision: A Case Series.

OBJECTIVES: To study the clinical profile and the route of spread of lacrimal sac rhinosporidiosis. To device a standard transnasal endoscopic surgical procedure in the treatment of lacrimal sac rhinosporidiosis. METHODS: This is a retrospective analysis of a case series on lacrimal sac rhinosporidiosis. Thirteen cases of lacrimal sac rhinosporidiosis reported in our institution from July 2003 to July 2016 were included. Workup included diagnostic nasal endoscopy and lacrimal syringing. All patients underwent transnasal endoscopic excision of rhinosporidiosis. RESULTS: Lacrimal sac rhinosporidiosis was more prevalent among males and in the 31 to 40 years age group. All patients had a history of bathing in stagnant water bodies. The most common clinical presentation was a doughy swelling medial to the medial canthus. Lacrimal sac rhinosporidiosis invariably presented with involvement of the nasolacrimal duct. Following the endoscopic excision of lacrimal sac rhinosporidiosis, all patients were followed up for a mean period of 16 months, and only one patient had recurrence. CONCLUSION: A unique aspect of our case series is that patients presented with features pertaining to isolated lacrimal system involvement rather than sinonasal involvement. Involvement of the nasolacrimal duct and nasal cavity without involvement of the eyes in all cases confirms the retrograde spread of rhinosporidiosis from the nasal cavity into the lacrimal sac via the nasolacrimal duct as the most common route of spread of infection. We recommend that endoscopic dacryocystorhinostomy, along with nasolacrimal duct excision used for surgical management in our case series with superior results, must become the standard of care in treatment of this condition. LEVEL OF EVIDENCE: 4 Laryngoscope, 128:2693-2696, 2018.

Rhinosporidiosis in Colombia: case series and literature review.

Rhinosporidiosis is a chronic granulomatous disease that affects mucosal surfaces. Its epidemiology and clinical presentation in Colombia are not well-known. We therefore reviewed all 58 reported cases between 1964 and 2015 to raise awareness among clinicians in a non-endemic area. Of the patients, 64% were male (median age = 15 years) and 57% had ocular and 43% nasal manifestations; there were no disseminated cases of the disease. All lesions were surgically removed.

Rhinosporidiosis: the largest case series in Brazil.

INTRODUCTION: Rhinosporidiosis is a chronic infection of the mucous membrane and is caused by Rhinosporidium seeberi, an aquatic mesomycetozoan. The mode of infection is probably transepithelial penetration. The large number of rivers and lakes and the strong presence of riparian populations in the State of Maranhão are strong predisposing factors for rhinosporidiosis. METHODS: A 5-year retrospective study was conducted in a tertiary medical center situated in Maranhão, Northeast Brazil. Twenty-five Maranhense patients diagnosed with rhinosporidiosis were analyzed. RESULTS: Most of the patients were children, adolescents and young adults (age range: 7-24 years, mean age: 14 years). The majority of the participants were male (84%), brown (76%), and students (92%). All lesions involved the entire nasal cavity and presented with a vascular polypoid mass. All patients were treated by surgical excision of the lesions. CONCLUSIONS: Rhinosporidiosis affects younger age groups, especially students from the countryside and the outskirts of urban areas. This study will aid and guide physicians in diagnosing and treating this infection in endemic areas.

Rhinosporidiosis: the largest case series in Brazil

Abstract: INTRODUCTION: Rhinosporidiosis is a chronic infection of the mucous membrane and is caused by Rhinosporidium seeberi, an aquatic mesomycetozoan. The mode of infection is probably transepithelial penetration. The large number of rivers and lakes and the strong presence of riparian populations in the State of Maranhão are strong predisposing factors for rhinosporidiosis. METHODS: A 5-year retrospective study was conducted in a tertiary medical center situated in Maranhão, Northeast Brazil. Twenty-five Maranhense patients diagnosed with rhinosporidiosis were analyzed. RESULTS: Most of the patients were children, adolescents and young adults (age range: 7-24 years, mean age: 14 years). The majority of the participants were male (84%), brown (76%), and students (92%). All lesions involved the entire nasal cavity and presented with a vascular polypoid mass. All patients were treated by surgical excision of the lesions. CONCLUSIONS: Rhinosporidiosis affects younger age groups, especially students from the countryside and the outskirts of urban areas. This study will aid and guide physicians in diagnosing and treating this infection in endemic areas.

Rhinosporidiosis in southwest Bengal.

Rhinosporidiosis is a non-contagious chronic granulomatous disease that is prevalent in southern India and Sri Lanka. It has been known for centuries, but the details of the disease and the precise manner of its transmission have, until recently, remained unknown. Our institution sees many cases of this disease and we investigate the management protocol and its recent advances and include a review of the published literature. A total of 152 patients who were treated at Bankura Sammilani Medical College were studied between 2005 and 2011. The most common age group affected were those aged between 11 and 20 years of age and the male-to-female ratio was 1.9:1. Three patients suffered recurrent disease - one experienced it on the same site and the others on distant sites. Eleven patients with inadequate excision in which the margins were not free from disease were treated with dapsone therapy without any reported recurrence. It is a common disease in southwestern West Bengal. Surgical excision with electrocoagulation of the base is the main treatment, and dapsone therapy is recommended in order to prevent recurrences in multiple sites of affection and inadequate surgically excised cases. Although the disease occurs sporadically in most parts of the world, we see many patients in our area.

Ocular and adnexal rhinosporidiosis : the clinical profile and treatment outcomes in a tertiary eye care centre.

INTRODUCTION: Rhinosporidiosis may mimic a burst chalazion or a simple conjunctival polyp. OBJECTIVE: To study the demography, histopathological evaluation, treatment modalities, their outcomes and recurrence rates in patients clinically and histopathologically diagnosed as ocular and adnexal rhinosporidiosis. PATIENTS AND METHODS: The study was conducted in the Department of Orbit, Oculoplasty and Oncology at a tertiary eye care centre in South India. Fifty patients were included who were diagnosed with ocular and adnexal rhinosporidiosis. The conjunctival and lid mass underwent complete excision. All patients with rhinosporidiosis of the lacrimal system under went dacryocystectomy (DCT) with care taken to avoid spilling the spores. Diagnosis was confirmed histopathologically. RESULTS: The mean age at presentation was 30.42 years, standard deviation( SD) being 16.89 (1 - 70 years). The mean follow-up was 14.2 months (12 months - 18 months) . The most common site was conjunctiva (n = 26 eyes, 52 %), followed by the lacrimal sac ( n = 13, 26 %) and lids (n = 11, 22 %). All the cases were treated by surgical excision with cautery at the base. There was a single recurrence (2 %) involving the lacrimal sac. Endonasal polypectomy combined with exploration in the sac region was done. There were no further recurrences during subsequent follow-up. CONCLUSION: Rhinosporidiosis is an ocular disorder with high recurrence rates reported. We recommend histopathological examination in every case following excision biopsy for conclusive diagnosis. Recurrence rates can be very low if a complete meticulous excision coupled with cauterization of the lesion is performed.

The regional sero-epidemiology of rhinosporidiosis in Sri Lankan humans and animals.

No data is available in the world literature on serum anti-rhinosporidial antibody levels in animals, and as far as we aware this is the first report. Although rhinosporidiosis in farm and domestic animals has been widely reported from other countries, rhinosporidiosis in animals has not been reported in Sri Lanka, though this country has the highest world-wide prevalence of human rhinosporidiosis on a unit-population basis. Serum IgG titres in 6 species of Sri Lankan animals (buffalo, cat, cattle, dog, goat, horse; total 291) were assayed by the Immuno blot (dot-ELISA) method on nitrocellulose paper and were compared with serum IgG titres in normal Sri Lankan human subjects (total 211) in different geographical areas, and in human Sri Lankan patients with rhinosporidiosis as reference values (total 36). Sensitization to rhinosporidial antigen(s) was detected in all 6 species of animals and the highest titres (1/3200) were found in cats, and free-grazing horses. Cattle showed higher levels of antibody than buffaloes. The titres in these animals are compared with world reports on overt rhinosporidiosis in these species, and with titres in normal Sri Lankan humans. Human, but not animal titres showed variations compatible with the regional prevalence of rhinosporidiosis. The variations in titres in animals especially horses, were probably more related to their mode of feeding, while in humans the titres in normal persons were probably related to the rhinosporidial-endemicity of their respective regions. No conclusions from sero-positivity in animals could be made regarding the absence of reports on rhinosporidiosis as an overt disease in these Sri Lankan animal species but the possibility of a genetically-determined insusceptibility to rhinosporidiosis in Sri Lanka, is considered. Rhinosporidium seeberi-specific PCR positive reactions were obtained with nasal scrapings from cattle that microscopically showed PAS+ bodies that were compatible with rhinosporidial sporangia. Sequence-analysis of the reactions products from five positive R. seeberi-specific PCR samples (four in this study and 1 in a previous study) gave results confirmatory of R. seeberi.

Nasal rhinosporidiosis in humans: new interpretations and a review of the literature of this enigmatic disease.

Rhinosporidiosis is a disease caused by Rhinosporidium seeberi which primarily affects the mucosa of the nose, conjunctiva and urethra. While it is endemic in some Asian regions, isolated cases are reported in other parts of the world as a result of the socio-cultural phenomenon of the migration. Its manifestation is a polypoid mass growing inside the affected cavity and the only treatment is surgical excision. Rhinosporidiosis is a condition which both clinicians and microbiologists should keep in mind when managing patients with nasal masses even those from non endemic areas. It is critical in such cases to follow the clinical course to ensure against recurrence of the disease. This study describes the clinical features, diagnosis, and treatment of rhinosporidiosis of the nose and nasopharynx in a series of three cases in East Delhi, India.

Human nasal rhinosporidiosis: a case report from Malawi.

Rhinosporidiosis is a rare chronic granulomatous disease, characterised by polypous lesions of the mucous membrane. Commonly affects the mucous membrane of the naso-pharynx, conjunctiva and palate. Its causative agent is Rhinosporidium seeberi. The disease is more prevalent in the Indian subcontinent, but remains quite rare in our environment. We hereby present a case description of a 70 year old native Malawian male with a polypoid nasal rhinosporidiosis. Patient presented with long standing history of nasal obstruction and intermittent epistaxis for three years. Diagnosis was confirmed by histopathological examination and he was successfully treated by complete surgical excision. This was a very unusual cause of nasal masses in our setting. Nasal rhinosporidioss lesions may largely mimic other ordinary nasal polyps, it is crucial therefore for clinicians in our region to consider rhinosporidiosis as a differential diagnosis when assessing patients presenting with nasal swellings.

Patterns of rhinosporidiosis in Sri Lanka: comparison with international data.

One hundred forty-three cases of rhinosporidiosis, confirmed by smear or biopsy, treated in two major General Hospitals in Sri Lanka over a 14 year period (1995-2009) were analyzed in regard to their epidemiological, clinical, clinicopathological, immunological and microbiological features. Regional variations in incidence, age and sex distribution, bathing history, and histopathology were seen. Lacustrine waters were the commonest probable source of infection (84%). Rivers were a source of Rhinosporidium seeberi in Sri Lanka (11%) and domestic well water was a probable source in 5%. The epidemiological features, clinical presentations and histopathology were similar to those in other series. The antirhinosporidial antibody (mean) titers were IgM--142.1 and IgG--178.5, compatible with rhinosporidiosis of long duration. Mantoux positivity to PPD was found in 65% of normal Sri Lankans, by only 35% of patients with rhinosporidiosis. No outbreaks have been reported in Sri Lanka or India. No animal cases of rhinosporidiosis have been reported in Sri Lanka, although rhinosporidiosis in animals has been repeatedly documented in India.

Rhinosporidiosis in Delhi, north India: case series from a non-endemic area and mini-review.

We report three cases of rhinosporidiosis from migrant population of Delhi. Three male patients had sino-nasopharyngeal, nasopharyngeal and nasal rhinosporidiosis, respectively. One patient gave a history of bathing in stagnant water. The diagnosis was made by clinical presentation and microscopic observation of characteristic sporangia of Rhinosporidium seebri in mycological and histopathological investigations. All the patients were successfully treated with complete surgical excision of lesions and cauterization of base. There were no recurrences.