An unusual cause of recurrent bloody tear.

We describe a female patient who presented with watering followed by swelling in the left infraorbital area of 5 years duration. She had previously been prescribed topical antibiotics on several occasions with no improvement. On pressure over the swelling, there was blood-tinged discharge from the left eye and nostril. Magnetic resonance imaging revealed an enhancing, well-defined mass lesion in the inferomedial aspect of the left orbit, likely of nasolacrimal origin. Computed tomography dacryocystogram with three-dimensional reconstruction showed a well-circumscribed mass with an irregular surface, originating from the lacrimal sac. Gram staining and potassium hydroxide mount from the regurgitant fluid revealed thick-walled cysts with sporangia suggestive of Rhinosporidium seeberi infection. Excision biopsy of the lesion confirmed R. seeberi as the causative agent. The patient has been put on long-term dapsone therapy to prevent a recurrence and has been asked to follow-up 6 months later.

Rinosporidiosis: a propósito de 5 casos / Rhinosporidiosis: A propos of five cases

Introducción: la rinosporidiosis es una enfermedad crónica granulomatosa poco frecuente, causada por el microorganismo Rhinosporidium seeberi, parásito acuático que afecta seres humanos y animales. Se describió por primera vez en 1900 por Guillermo Seeber, a partir de alteraciones observadas en un pólipo nasal, de ahí el origen de su nombre. Se presenta clínicamente como un pólipo friable de color rosado en la cavidad nasal o en la conjuntiva, aunque ha sido descrita la entidad en otros sitios. Los pacientes afectados presentan entre 20 y 40 años de edad, con predilección por el sexo masculino. Sri Lanka, el sur de la India y el sureste de Asia constituyen las áreas geográficas de mayor prevalencia. Presentación de caso: se realiza el reporte de 5 casos en el Hospital de Mbarara, Uganda, en pacientes que acuden a consulta, cuatro de ellos, por lesión papilomatosa en conjuntiva y uno con historia de obstrucción nasal. La información sobre la historia de la enfermedad fue recogida de la historia clínica de cada paciente. Resultados:después de la biopsia excisional, el estudio histológico de las lesiones demostró que todos los casos cumplieron con los criterios histopatológicos para el diagnóstico de la entidad. Conclusiones: la mucosa nasal y conjuntival son los sitios más afectados; se considera una patología de interés por su baja incidencia, la edad de presentación así como la clínica. Macroscópicamente puede ser confundido con un pólipo nasal secundario a procesos alérgicos o tumores vasculares. La excisión de la lesión suele ser suficiente para la cura del paciente(AU)

Background: Rhinosporidiosis is a rare, chronic granulomatous disease caused by the organism Rhinosporidium seeberi, aquatic protistan parasite. First described in 1900 by Guillermo Seeber, because of histological finding in a nasal polyp, that was the origin of the disease's name. Generally presents as swollen, pink or red polyps in the nasal cavity or the ocular conjunctivae but other sites of infection have been report. It is more frequently present in patient between 20 and 40 years old, with predilection inmale. It has high prevalent in southern India, Sri Lanka, and Southeast Asia. Clinical case: This report describes a five case of Rhinosporidiosis, four of them in conjunctiva and one in nasal cavity with history of nasal obstruction. Clinical outcome information was obtained from hospital files. Results: After surgical excision of the lesions, the microscopic finding corresponded in all patients with Rhinosporidiosis. Conclusions: Despite the nose and the conjunctiva are the most common sites of infection, it has been considerable interest due to age of presentation, the low incidence of the disease, and the clinical presentation that can be confuse with allergic nasal polyp, or vascular tumors. Surgical excision of the polyps is often successful in treating the disease(EU)

Rinosporidiosis: a propósito de 5 casos / Rhinosporidiosis: A propos of five cases

Introducción: la rinosporidiosis es una enfermedad crónica granulomatosa poco frecuente, causada por el microorganismo Rhinosporidium seeberi, parásito acuático que afecta seres humanos y animales. Se describió por primera vez en 1900 por Guillermo Seeber, a partir de alteraciones observadas en un pólipo nasal, de ahí el origen de su nombre. Se presenta clínicamente como un pólipo friable de color rosado en la cavidad nasal o en la conjuntiva, aunque ha sido descrita la entidad en otros sitios. Los pacientes afectados presentan entre 20 y 40 años de edad, con predilección por el sexo masculino. Sri Lanka, el sur de la India y el sureste de Asia constituyen las áreas geográficas de mayor prevalencia. Presentación de caso: se realiza el reporte de 5 casos en el Hospital de Mbarara, Uganda, en pacientes que acuden a consulta, cuatro de ellos, por lesión papilomatosa en conjuntiva y uno con historia de obstrucción nasal. La información sobre la historia de la enfermedad fue recogida de la historia clínica de cada paciente. Resultados: después de la biopsia excisional, el estudio histológico de las lesiones demostró que todos los casos cumplieron con los criterios histopatológicos para el diagnóstico de la entidad. Conclusiones: la mucosa nasal y conjuntival son los sitios más afectados; se considera una patología de interés por su baja incidencia, la edad de presentación así como la clínica. Macroscópicamente puede ser confundido con un pólipo nasal secundario a procesos alérgicos o tumores vasculares. La excisión de la lesión suele ser suficiente para la cura del paciente(AU)

Background: Rhinosporidiosis is a rare, chronic granulomatous disease caused by the organism Rhinosporidium seeberi, aquatic protistan parasite. First described in 1900 by Guillermo Seeber, because of histological finding in a nasal polyp, that was the origin of the disease's name. Generally presents as swollen, pink or red polyps in the nasal cavity or the ocular conjunctivae but other sites of infection have been report. It is more frequently present in patient between 20 and 40 years old, with predilection inmale. It has high prevalent in southern India, Sri Lanka, and Southeast Asia. Clinical case:This report describes a five case of Rhinosporidiosis, four of them in conjunctiva and one in nasal cavity with history of nasal obstruction. Clinical outcome information was obtained from hospital files. Results: After surgical excision of the lesions, the microscopic finding corresponded in all patients with Rhinosporidiosis. Conclusions: Despite the nose and the conjunctiva are the most common sites of infection, it has been considerable interest due to age of presentation, the low incidence of the disease, and the clinical presentation that can be confuse with allergic nasal polyp, or vascular tumors. Surgical excision of the polyps is often successful in treating the disease(EU)

Lesión polipoide en la conjuntiva / Polypoid lesion on the conjunctiva

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Cytologic identification of immature endospores in a dog with rhinosporidiosis.

An 8-year-old, intact, male Labrador Retriever was presented to the Boren Veterinary Medical Teaching Hospital at Oklahoma State University with a 2-month history of severe sneezing episodes that resulted in epistaxis and bilateral sanguineous discharge. Rhinoscopy revealed a small polypoid mass, and specimens were obtained for histopathology. Microscopic examination of formalin-fixed tissue specimens revealed organisms consistent with Rhinosporidium seeberi. The mass was surgically excised and impression smears were made for cytology examination. Smears revealed high numbers of endospores, typical of those previously described for R seeberi. In addition, numerous smaller structures, presumed to be immature endospores, were noted. The immature endospores were morphologically distinct from mature endospores and have not been described previously. Recognition of immature forms of Rhinosporidium may help prevent misidentification of the organism or misdiagnosis of a dual infection.

Detection of 16S rRNA gene in round bodies isolated from polyps of rhinosporidiosis.

Polyps of rhinosporidiosis contain microscopic round bodies, believed to be causative agents of the disease. The source and identity of these round bodies has been debated ever since the first case was described in the year 1900. Most patients have a history of exposure to pond water. Collection and microscopic analysis of pond water samples where patients had been dipping led to the idea that a cyanobacterium could possibly develop into round bodies after entering the patient's nose. Molecular genomic studies based on PCR, cloning, and sequencing were, therefore, carried out using purified DNA, extracted from individually separated round bodies from polyp samples. Parallel investigations were also conducted on DNA extracted from cyanobacterium Microcystis isolated from pond water samples. The results of these experiments presented here provide evidence of the presence of 1458 bp 16S rRNA gene in round bodies. This is the first molecular study indicating the prokaryotic identity of round bodies in rhinosporidiosis.

Tropical dermatology: fungal tropical diseases.

UNLABELLED: Fungal infections are common in tropical countries and can have an important impact on public health. Lobomycosis is a common fungal infection in the tropical rain forest of South America, and paracoccidioidomycosis (South American blastomycosis) is a widespread and sometimes severe illness. Penicilliosis marneffei is an opportunistic infection of AIDS patients in southeast Asia. Chromoblastomycosis and mycetomas are causes of morbidity around the world. Sporotrichosis is a worldwide subcutaneous mycosis with a high incidence in tropical countries and is an important illness in immunocompromised patients. Rhinosporidiosis was classed as a fungal infection but is now considered a protistan parasite that belongs to the class Mesomycetozoea. It is included in this review because of its historical classification. In the past, most of these mycoses were restricted to specific geographic areas and natural reservoirs. There are, however, situations in which people from other regions come in contact with the pathogen. A common situation involves an accidental contamination of a traveler or worker who has contact with a tropical mycosis. Even minor trauma to the skin surface or inhalation of the fungal conidia can infect the patient. Thus recognition of the clinical symptoms and the dermatologic findings of the diseases, as well as the geographic distribution of the pathogens, can be critical in diagnosis of the tropical mycoses. This review discusses some of the more common tropical subcutaneous and systemic mycoses, as well as their signs, symptoms, methods of diagnosis, and therapies. LEARNING OBJECTIVE: At the completion of this learning activity, participants should be able to recognize the clinical and histologic presentations of tropical fungal diseases with cutaneous manifestations and be familiar with the appropriate therapies.

Human anti-rhinosporidial antibody does not cause metabolic inactivation or morphological damage in endospores of Rhinosporidium seeberi, in vitro.

This report describes the use of the MTT-reduction and Evan's blue-staining tests for the assessment of the viability and morphological integrity, respectively, of rhinosporidial endospores after exposure to sera from rhinosporidial patients with high titres of anti-rhinosporidial antibody. Sera from three patients, with nasal, ocular and disseminated rhinosporidiosis respectively were used, with human serum without anti-rhinosporidial antibody for comparison, with or without added fresh guinea pig serum as a source of complement. All four sera tested, with or without guinea-pig serum, had no effect on the morphological integrity or the viability of the endospores and it is suggested that anti-rhinosporidial antibody has no direct protective role against the endospores, the infective stage, in rhinosporidiosis. This finding is compatible with the occurrence of chronicity, recurrence and dissemination that are characteristic of rhinosporidiosis despite the presence of high titres of anti-rhinosporidial antibody in patients with these clinical characteristics. The possible occurrence of humoral mechanisms of immunity that involve anti-rhinosporidial antibody with cells such as leucocytes and NK cells, in vivo, cannot yet be discounted, although the presence of high titres of anti-rhinosporidial antibody in patients with chronic, recurrent and disseminated lesions might indicate that such antibody is non-protective in vivo.

Molecular evidence for multiple host-specific strains in the genus Rhinosporidium.

The taxonomic relationship of Rhinosporidium seeberi with other organisms remained controversial for over a century. Recently, molecular studies have shown R. seeberi to be a protistal microbe in the newly described class Mesomycetozoea at the animal-fungal boundary. Phylogenetic analyses of R. seeberi using 18S small-subunit (SSU) rRNA genes from several hosts suggested Rhinosporidium as a monotypic genus. To test this hypothesis, the internal transcribed spacer 1 (ITS1), 5.8S, and ITS2 from eight humans, two swans, and a dog with rhinosporidiosis were sequenced. The ITS regions were amplified by PCR using a primer designed from a unique region of R. seeberi's 18S SSU rRNA genes in combination with the ITS4 universal primer. In addition, the universal ITS4 and ITS5 primers were also used. R. seeberi's ITS sequences showed differences in the numbers of nucleotides among strains. For instance, the eight human ITS sequences were uniformly similar with only a few mismatches and approximately 1,060 bp long. In contrast, sequences from one of the swans and the dog were 1,356 bp and approximately 1,147 bp long, respectively. Clustal analysis of all of the ITS sequences showed multiple 50- to 60-bp gaps and several mismatches among them. Parsimony analysis placed the Rhinosporidium ITS sequences in three well-supported sister groups according to the hosts' identities. This analysis strongly suggests that the genus Rhinosporidium may possess multiple host-specific strains. No correlation was found between this finding and the phenotypic features of R. seeberi in the studied samples.

Rhinosporidiosis: what is the cause?

PURPOSE OF REVIEW: Significant advances in knowledge on rhinosporidiosis and Rhinosporidium seeberi were made in 1999, 2000, 2003 and 2004. These advances are reviewed on account of the continuing sporadic occurrence of the disease universally, and because of the availability of new approaches that could resolve persisting enigmas of both the disease and its causative pathogen. RECENT FINDINGS: R. seeberi, the pathogen that causes rhinosporidiosis, has been definitively classified using molecular biological tools in a new clade - the Mesomycetozoea, along with 10 parasitic and saprobic microbes. The controversial spherical bodies of the endospores have been shown to comprise both lipid/protein nutritive bodies and other spherical bodies that are metabolizing units that reduce MTT (3-[4,5-dimethyl-2-thiazolyl]-2,5-diphenyl-2H-tetrazolium bromide). This indicates the viability of these spherical bodies, provisionally identified as the electron dense bodies that have also been shown to contain nucleic acids. MTT reduction as an indicator of viability has been used to determine the sensitivity of rhinosporidial endospores to biocides, antimicrobial drugs, and to specific antibodies. Genetic heterogeneity has been identified in strains from humans and animals. Cell-mediated and humoral immune responses have been demonstrated in human patients and in mice. Several mechanisms of immune evasion by R. seeberi have been identified. SUMMARY: These findings are applicable in both clinical and laboratory practice, while the basic advances have implications in further work on experimental pathogenicity, the biology of R. seeberi, and on the epidemiology and pathogenesis of rhinosporidiosis.

Rhinosporidiosis in Egypt: a case report and review of literature.

Rhinosporidiosis is an infection caused by Rhinosporidium seeberi that frequently presents as a polypoidal nasal lesions. Here, we report the first indigenous case of tumoral rhinosporidiosis in Egypt. In this case, a 25-year-old male patient from a rural background of Assuit City presented with epistaxis and a nasal polyp. The patient had not traveled abroad. The diagnosis was established on the morphological basis by the identification of 5- to 10-microm endospores and 50- to 1000-microm sporangia. The clinicopathological and immunologic features were discussed and the literature was reviewed. To the best of our knowledge this is the first case of this disease to be reported in Egypt in the human literature.

Lacazia loboi and Rhinosporidium seeberi: a genomic perspective.

In the past five years, with the use of molecular strategies the phylogenetic affinities of the two more resilient pathogens studied in medical mycology, Lacazia loboi and Rhinosporidium seeberi were finally deciphered. These studies found that L. loboi was the sister taxon to Paraccidioides brasiliensis, and R. seeberi was closely related to protistan spherical aquatic fish pathogens, located at the point were animals diverged from the fungi, in the class Mesomycetozoea. These initial studies indicated that a molecular strategy was the ideal approach to further understand these anomalous pathogens. However, the limited amount of information gathered so far from few DNA sequences, although crucial to place these organisms in the tree of life and to take a glance to their ecological preferences, did not provide answers to other important traits. In the following pages we discuss a genomic perspective for both pathogens and the benefit that such information could generate to understand more about these two uncultivated pathogens.

Lacazia loboi and Rhinosporidium seeberi: a genomic perspective

In the past five years, with the use of molecular strategies the phylogenetic affinities of the two more resilient pathogens studied in medical mycology, Lacazia loboi and Rhinosporidium seeberi were finally deciphered. These studies found that L. loboi was the sister taxon to Paraccidioides brasiliensis, and R. seeberi was closely related to protistan spherical aquatic fish pathogens, located at the point were animals diverged from the fungi, in the class Mesomycetozoea. These initial studies indicated that a molecular strategy was the ideal approach to further understand these anomalous pathogens. However, the limited amount of information gathered so far from few DNA sequences, although crucial to place these organisms in the tree of life and to take a glance to their ecological preferences, did not provide answers to other important traits. In the following pages we discuss a genomic perspective for both pathogens and the benefit that such information could generate to understand more about these two uncultivated pathogens.

Anti-rhinosporidial antibody levels in patients with rhinosporidiosis and in asymptomatic persons, in Sri Lanka.

The only report hitherto, from India in 1982, on anti-rhinosporidial antibody levels in patients with rhinosporidiosis recorded that antibody was not detected in Indian patients. The present report describes the use of the dot-ELISA assay of serum anti-rhinosporidial IgG, IgM and IgA and salivary sIgA in patients with diverse clinical presentations, in rural asymptomatic persons who had bathed in ground waters that probably harboured the causative pathogen, Rhinosporidium seeberi, and in laboratory persons who were exposed to R. seeberi. Ultrasonic extracts of purified endospores and sporangia of R. seeberi were used as antigen. The geometric mean (reciprocal) titres of serum antibody detected in patients were IgM 142.1, IgG 178.5, IgA 84.6, with ranges of 0-640, 30-960 and 0-160 respectively, salivary sIgA titres ranged from 0 to 18 with a mean of 4.6. The levels of antibody had no correlation with the site, the number of sporangia, duration and recurrence of the disease. Asymptomatic persons from the same endemic area as patients showed mean titres of IgM 89.6, IgG 69.1, IgA 95.5, with salivary sIgA titres of 3.1. Asymptomatic personnel who had been working in a laboratory where rhiniosporidial work was being done, showed mean titres of 169.6 IgM, 62.8 IgG, and 6.5 salivary sIgA. These results indicate that an anti-rhinosporidial antibody response occurs in rhinosporidial patients, as well as in asymptomatic persons who were exposed to R. seeberi in the environment. Anti-R. seeberi antibody does not appear to be protective in rhinosporidiosis since appreciable titres were present in patients with recurrent, single, multiple or disseminated lesions of long duration.

Ocular rhinosporidiosis.

Rhinosporidium seeberi, the causative organism causing rhinosporidiosis, also affects extranasal sites. A 1 5-year-old male presented with a conjunctival fleshy swelling near the inner canthus of the left eye. His visual acuity was within normal range. Conjunctival papilloma was the provisional diagnosis for which he was operated on. Histopathology proved to be a case of ocular rhinosporidiosis. Six months follow-up of the case showed no recurrence.

The assessment of the viability of the endospores of Rhinosporidium seeberi with MTT (3-[4, 5-dimethyl-2-thiazolyl]-2, 5-diphenyl-2H-tetrazolium bromide).

This report describes tests with Evan's Blue and MTT (3-[4, 5-dimethyl-2-thiazolyl]-2, 5-diphenyl-2H-tetrazolium bromide) for the assessment of the viability of rhinosporidial endospores. MTT stained a proportion of the spherical bodies that we regard as the Electron Dense Bodies (EDBs), and the cytoplasm of freshly prepared endospores or ones that were stored at 4 degrees. Slow-freezing at -20 degrees C, exposure to 10% formalin, or 0.1% sodium azide of the endospores abolished MTT-staining in both sites. Evan's Blue stained the EDBs and cytoplasm of fresh endospores or those stored at 4 degrees, and of sodium azide-treated or frozen (-20 degrees)-thawed endospores. TMRE (tetramethyl-rhodamine ethyl ester) specifically labelled the spherical bodies, supporting the conclusion that these spherical bodies have a mitochondrial-like structure. TMRE-staining was however retained in endospores after their treatment with formalin, sodium azide and slow-freezing while MTT-staining was abolished in all these treated endospores. These results indicate that EvB and TMRE were capable of revealing the morphological integrity of endospores but failed to identify the metabolic inactivation of the endospores after treatment with formalin, sodium azide or slow-freezing. Only MTT was capable of identifying metabolically active endospores and hence their viability and could prove to be of value in standardizing models of infection.