Recurrent rhinosporidiosis in a young male: A case report and its taxonomical journey.

Rhinosporidiosis is a rarely encountered granulomatous infection caused by Rhinosporidium seeberi affecting both humans and animals. Although the disease has been reported worldwide, it is mainly endemic in tropical and subtropical countries. In the Indian subcontinent, it is endemic in some parts like Orissa, Tamil Nadu, Kerala, eastern Madhya Pradesh, and Chhattisgarh. It is a chronic granulomatous disease with varied controversial taxonomical history, but recently based on genetic sequencing and the nature of aquatics, it was later identified as an aquatic eukaryote. The mucous membranes are frequently impacted in humans, with a typical manifestation being the presence of a polypoidal mass. The occurrence of Rhinosporidiosis in nonendemic regions is uncommon. We report one such case of a young male with recurrent Rhinosporidiosis from India.

[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.

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

Resumen 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 ro jizos 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 epite lio 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 μm (esporangios), que contenían en su interior abundantes endosporas. Las técnicas de ácido peryódico de Schiff y Grocott resalta ron dichas estructuras. Dado los antecedentes clínico-epidemiológicos y el cuadro morfológico se arribó al diagnóstico de rinosporidiosis.

Abstract Rhinosporidiosis is an infectious pathology caused by the aquatic protist pathogen Rhinosporidium seeberi that in habits the fresh water of lakes or lagoons and in the soil. It is extremely rare, being endemic in India and Sri Lanka, al though 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 obstruc tion, 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 μm (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.

Equine rhinosporidiosis in Buenos Aires, Argentina

Abstract 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.

Resumen La rinosporidiosis es una enfermedad causada por Rhinosporidium seeberi, un organismo parasitario clasificado en la familia Rhinosporideacea, clase Micomycetozoa. Es una enfermedad endémica de la India, pero se notificaron algunos casos en Europa, África, América del Norte y América del Sur. El objetivo del presente estudio fue describir tres casos de rinosporidiosis en caballos de vida libre en diferentes ciudades de la provincia de Buenos Ares, Argentina. Confirmamos la presencia de R. seeberi en las muestras analizadas utilizando técnicas histopatológicas, PCR y secuenciación.

A new technique to resolve Nasal Rhinosporidiosis: A case report with review of literature.

INTRODUCTION AND IMPORTANCE: Rhinosporidiosis is a chronic, localized granulomatous infectious disease caused by Rhinosporidium seeberi that predominantly affects the mucosal membranes of the nose and nasopharynx, conjunctiva, and urethra. Rhinosporidium seeberi is a eukaryotic pathogen that spreads in certain geographical areas, particularly in tropical and subtropical areas, through aquatic exposure. CASE PRESENTATION: We present the case of a young man who had been suffering from a right nasal mass for four months, and whose diagnosis was confirmed after surgical excision and histopathological examination, which revealed distinct pathognomonic findings. Laser-assisted endoscopic excision, in combination with Dapsone, is recommended as a more effective treatment to prevent a recurrence. CLINICAL DISCUSSION: For clinicians, it has been advised to obtain a detailed case history of exposure in patients diagnosed with Rhinosporidiosis. Rhinosporidiosis can be diagnosed with a simple examination of H&E-stained histopathological sections. Because chemotherapy has not been proven to be effective, Laser-assisted endoscopic excision, in combination with Dapsone is the recommended treatment for Rhinosporidiosis. CONCLUSION: One of the differential diagnoses for Rhinosporidiosis in the nasal cavity is masses or abnormal growths without bleeding, which should be kept in mind by clinicians and pathologists.

Nasopharyngeal rhinosporidiosis with intracranial extension masquerading as juvenile angiofibroma: an unusual entity.

Rhinosporidiosis is a chronic fungal inflammatory disease prevalent in India and Sri Lanka. Its manifestations are mostly nasal and extranasal lesions are relatively rare. Occasional atypical presentations of this disease lead to diagnostic dilemma. Herein we report on a case of nasopharyngeal rhinosporidiosis having extensive involvement of paranasal sinuses along with intracranial extension which mimicked radiologically as juvenile nasopharyngeal angiofibroma. To our knowledge, this is the first reported case of rhinosporidiosis having intracranial extension. We discuss the pathology, treatment and briefly review the literature of this rare disease.

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.

A case of primary disseminated rhinosporidiosis and dapsone-induced autoimmune hemolytic anemia: A therapeutic misadventure.

Rhinosporidiosis, a chronic inflammatory disease, which is caused by the aquatic microorganism Rhinosporidium seeberi, is endemic in India and in many other regions of the tropics. It primarily infects mucocutaneous surfaces of nose, nasopharynx, and conjunctiva through transepithelial invasion. However, over the centuries, atypical involvement of other body parts, especially viscera, bone, subcutaneous layers, genitals, the tracheobronchial tree, and even the skull has been, though rarely, reported. This chronic granulomatous infection is notorious for its propensity for recurrence following autoinoculation and poor response to most of the anti-microbials except dapsone. Surgical excision followed by cauterization remains the treatment of choice when an operation is feasible. We herein report a case of an immunocompetent person with primary disseminated dermato-pulmonary rhinosporidiosis, which created significant diagnostic dilemma at the beginning, got complicated due to dapsone-induced direct anti-globulin test-positive autoimmune hemolytic anemia, and finally responded to prolonged multidrug therapy with liposomal amphotericin B, ketoconazole and cycloserine. This report establishes the importance of tissue diagnosis in rhinosporidiosis and even, in resource-poor set-ups, a simple histopathological diagnosis can promote an early and affordable accurate diagnosis, and subsequently, a proper therapeutic intervention.

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.

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.

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.

Rhinosporidiosis in horses / Rinosporidiose em equinos

Rhinosporidiosis is a disease caused by Rhinosporidium seeberi, an aquatic protist of the class Mesomycetozoa. It primarily affects the nasal mucosa and transmission is associated with contaminated water contact. This report describes seven cases of rhinosporidiosis in horses in Rio Grande do Sul covering the period of 13 years. The disease predominantly affected Crioulo and thoroughbred horses. No apparent gender predisposition occurs, and age ranged from two to 25 years, with a median of 10 years. The gross aspects were characterized by unilateral (85.7%, 6/7) or bilateral (14.3%, 1/7) polyps. These were soft to friable, whitish to pink, cauliflower-like, with an irregular, sometimes ulcerated surface, measuring 2.5 to 6.0cm in diameter. There was a severe inflammatory infiltrate of the submucosa was observed, associated with moderate proliferation of the epithelium, and numerous rounded structures were identified compatible with sporangia of R. seeberi. Rhinosporidiosis should be included in the differential diagnosis of other conditions affecting the respiratory tract of horses, and it is important to perform histopathology for diagnosis.(AU)

A rinosporidiose é uma doença causada por Rhinosporidium seeberi, protista aquático da classe Mesomycetozoa. Acomete principalmente a mucosa nasal e a transmissão está associada ao contato com água contaminada. Este trabalho descreve sete casos de rinosporidiose em equinos no Rio Grande do Sul em um período de 13 anos. A doença afetou predominantemente cavalos de raça, como Crioulo e Puro Sangue Inglês, sem predisposição sexual evidente e a idade variou de dois a 25 anos, com a mediana de 10 anos. Macroscopicamente foram caracterizadas por pólipos unilaterais (85,7%; 6/7) ou bilaterais (14,3%; 1/7). Os pólipos eram macios a friáveis, esbranquiçados a róseos, com aspecto de couve flor e com superfície irregular, por vezes ulcerada, medindo 2,5 a 6,0cm de diâmetro. Havia infiltrado inflamatório piogranulomatoso acentuado na submucosa associado à moderada proliferação do epitélio e numerosas estruturas arredondadas compatíveis com esporângios de R. seeberi. A rinosporidiose deve ser incluída no diagnóstico diferencial de outras patologias que acometem o trato respiratório de equinos, sendo importante a realização da histopatologia para diagnóstico.(AU)

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.

Palatal rhinosporidiosis: an unusual case report and review of the literature.

Rhinosporidiosis is a chronic, granulomatous, mucocutaneous infection caused by Rhinosporidium seeberi . The infection is non-contagious and sporadic in humans. The site most commonly affected is the mucous membrane of the nose and nasopharynx, followed by the oropharynx, trachea, bronchi, ear, eye, and genitourinary tract. It can also spread to other areas through blood and lymph. Here, we report a case of rhinosporidiosis affecting the palate in a 60-year-old female patient.

Palatal rhinosporidiosis: an unusual case report and review of the literature

Rhinosporidiosis is a chronic, granulomatous, mucocutaneous infection caused by Rhinosporidium seeberi . The infection is non-contagious and sporadic in humans. The site most commonly affected is the mucous membrane of the nose and nasopharynx, followed by the oropharynx, trachea, bronchi, ear, eye, and genitourinary tract. It can also spread to other areas through blood and lymph. Here, we report a case of rhinosporidiosis affecting the palate in a 60-year-old female patient.

Disseminated Rhinosporidiosis with Conjunctival Involvement in an Immunocompromised Patient.

Rhinosporidiosis is a granulomatous infection of mucocutaneous tissue caused by Rhinosporidium seeberi that most commonly occurs in the nasal cavity. Ocular rhinosporidiosis affects primarily the conjunctiva. Diagnosis of rhinosporidiosis is based on strong clinical suspicion and is confirmed by histopathological examination. We report a rare case of conjunctival rhinosporidiosis in an immunocompromised patient (human immunodeficiency virus) with disseminated cutaneous rhinosporidiosis. A 44-year-old male presented with a swelling in the right upper eyelid for 6 months. Excision biopsy of the ocular lesion showed multiple thick-walled, variable-sized sporangia containing endospores within the subepithelium suggestive of rhinosporidiosis. A multidrug regimen of systemic cycloserine, ketoconazole, and dapsone was administered to treat disseminated rhinosporidiosis, in addition to antiretroviral therapy. There was good response with reduction in the swellings.

Socio-demographic Correlates of Rhinosporidiosis: A Hospital-Based Epidemiologic Study in Purulia, India.

The present paper attempts to explore the socio-demographic profile of patients with rhinosporidiosis in an endemic area. A cross-sectional study was carried out in a tertiary-care hospital in Purulia district, India, including consecutive patients with histologically-proved rhinosporidiosis. Their socio-demographic profiles were obtained through a pre-designed proforma with given epidemiologic parameters. Data was statistically analyzed with inputs from literature review. Of the 39 patients included, 87 % were fresh/new cases. The age-group of 10-20 years was mostly involved, with multiple peaks around 50. About 82 % were from rural background, commonly involved in cattle farming and agriculture, with a universal habit of pond-bathing. There was a male preponderance; however women were being increasingly affected. Nasal cavity was the predominant site involved; nasal obstruction and epistaxis were the primary complaints. About 13 % had recurrent lesions that were statistically related to higher age-group (≥15 years) and occupation (agriculture, labor). Rhinosporidiosis is predominantly the disease of young rural adults engaged in field activities and habituated to pond-bathing. A bimodal age distribution was noticed. The present article provides an update on the socio-demographic perspectives of rhinosporidiosis in an endemic zone. It also summarizes the factors that would identify the vulnerable population and help formulate preventive measures.

Rinosporidiosis nasal: reporte de un nuevo caso y revisión de la literatura / Nasal rhinosporidiosis: new case report and review

La rinosporidiosis es una enfermedad granulomatosa rara producida por el microorganismo Rhinosporidium seeberi. A pesar de ser considerada una infección endémica en algunas zonas de Asia, en nuestro país es una enfermedad extremadamente rara. Se presenta el caso clínico de un escolar de 10 años que consulta por aumento de volumen en fosa nasal izquierda de 1 mes de evolución, de crecimiento progresivo, con epistaxis autolimitada, presentando al examen físico una lesión polipoídea, en la cual, no existiendo sospecha previa, se determinó mediante histopatología la presencia de rinosporidio-sis. Se discute la epidemiología de la enfermedad, sus mecanismos de diseminación, alternativas de tratamiento y principales complicaciones.

Rhinosporidiosis is a rare granulomatous disease produced by the microorganism Rhinosporidium seeberi. Despite being considered an endemic infection in some areas of Asia, in our country it is an extremely rare disease. We present the case of a 10 year-old boy who consult for increased volumen in the left nostril of 1 month evolution, with progressive growth, self-limited epistaxis, and a physical examination with a polypoid lesion, in which there were no prior suspicion, histopathology determined the presence of rhinosporidiosis. Epidemiology of the disease, its dissemination mechanisms, treatment options and major complications are discussed.

A Retrospective Epidemiological Study of Rhinosporidiosis in a Rural Tertiary Care Centre in Pondicherry.

INTRODUCTION: Rhinosporidiosis is an age old endemic scourge which has affected various parts of the world, most notably India and Sri Lanka. 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. AIM: The aim of this study was to find out the site, presentation and postoperative recurrence rate in patients with rhinosporidiosis. MATERIALS AND METHODS: This was a retrospective study involving operated cases of rhinosporidiosis in the Department of Ear, Nose and Throat of a rural tertiary care referral centre over a period of 3 years. The behavioural pattern of the disease, its epidemiology, rate of recurrence and outcomes of surgical management were analysed. RESULTS: A statistically significant correlation could be found between male gender and rural residential status and incidence of rhinosporidiosis. Bathing in rivers and ponds were found to be strongly associated with rhinosporidiosis (p=0.005). Almost all patients were found to be of low socioeconomic status. Nasal obstruction and epistaxis were the most common presenting complaints. CONCLUSION: Rhinosporidiosis is strongly associated with male gender, young and middle aged adults, agricultural occupations, rural residential status, a history of bathing in ponds and rivers and a low socioeconomic status. Post-surgical recurrence of rhinosporidial masses can be avoided with careful and complete clearance of the mass and cauterization of the base. Appropriate and consistent public health education helps to reduce the incidence of rhinosporidiosis in endemic communities.

Elusive treatment for human rhinosporidiosis.

OBJECTIVES: The aim of this study was to clarify the contentious taxonomic classification of Rhinosporidium seeberi, the cause of human rhinosporidiosis, which may have treatment implications. METHODS: PCR was used to amplify the internal transcribed spacer (ITS)-2 region from the genomic DNA of the aetiological agent obtained from a sample of human rhinosporidiosis lesions. The amplicon was sequenced and the organism identified using the Basic Local Alignment Search Tools (BLAST). RESULTS: Phylogenetic analysis revealed that the aetiological agent clustered along with the R. seeberi isolated from humans and also with Amphibiocystidium ranae from frogs. This organism is a member of the order Dermocystida in the class Mesomycetozoea. A patient with disseminated rhinosporidiosis did not respond to conventional therapy with dapsone and surgical excision, and treatment with amphotericin B also proved futile. CONCLUSION: An effective treatment for R. seeberi-a eukaryote belonging to the class Mesomycetozoea-is still elusive.