Case Report of Nasal Rhinosporidiosis in South Africa.

We describe a classic case of nasal rhinosporidiosis in a woman who resided in Johannesburg, South Africa, but originated from a rural area in Eastern Cape Province. We confirmed histologic diagnosis using PCR testing and compared details with those from records on 17 other cases from South Africa.

Tumoral cutaneous rhinosporidiosis: Case report and review of literature.

Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi commonly affecting nasal mucosa, conjunctiva, and urethra. Subcutaneous tumor nodule presentation is rare and often mimics as sarcoma. Such tumoral rhinosporidiosis has been reported rarely. This report describes a 60-year male who presented with a solitary, firm, nontender swelling in posterior aspect of right leg with an ulcer and mimicking clinically as soft tissue sarcoma. Histopathology was diagnostic. Surgical excision was found to be useful.

[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.

Lacrimal Sac Rhinosporidiosis: Clinical Profile and Gram Stain as a Method of Preoperative Diagnosis.

PURPOSE: To describe the clinical profile of lacrimal sac rhinosporidiosis and to introduce a method for preoperative microbial identification of rhinosporidiosis using Gram stain. METHODS: This is a prospective study, carried out from January 2016 to January 2022. This series included 18 patients with clinical suspicion of lacrimal sac rhinosporidiosis. All patients underwent a comprehensive eye check-up. Mucopurulent discharge collected by a sterile swab after applying pressure over the sac area and Gram staining was done. All patients underwent dacryocystectomy. The sac contents were sent for histopathology, and rhinosporidiosis was confirmed. RESULTS: A total of 18 patients over a period of 6 years with suspicion of lacrimal sac rhinosporidiosis were included. There were 11 (61.1%) male patients. Ten patients (55.5 %) had a history of either regular or occasional bathing in stagnant water. Nontender doughy swelling over the lacrimal sac region was the most common presentation. Gram staining of the mucopurulent discharge revealed thick-walled sporangia with endospores suggestive of rhinosporidiosis in all these cases. All patients underwent dacryocystectomy. Hematoxylin and eosin sections confirmed the diagnosis. Two patients had recurrence within 6 months after surgery. CONCLUSION: Regurgitation of pus mixed with whitish granular particles or blood is highly suspicious of rhinosporidiosis. In clinically suspected cases, microbial diagnosis using Gram stain is an economical, office procedure, which helps the surgeon to plan for the surgery and for better counseling.

Disseminated rhinosporidiosis masquerading as soft tissue round cell tumour diagnosed by fine needle aspiration cytology.

Rhinosporidium seeberi belongs to the eukaryotic class Mesomycetozoea and causes chronic granulomatous lesions known as rhinosporidiosis. Rhinosporidiosis frequently involves the nasal cavity and nasopharynx through transepithelial invasion. Atypical presentations of this disease at other body sites have been reported, including the subcutis, visceral organs, bones, and genitals. Only a few cases of cutaneous and subcutaneous involvement have been reported to date. This chronic granulomatous condition is known for its recurrence following autoinoculation unless the correct diagnosis and appropriate treatment are given. We describe a case of an immunocompetent adult who had undergone fine needle aspiration cytology (FNAC) of mass-like swellings in the right thigh and right calf at another healthcare centre and had been diagnosed with a small round blue cell tumour. FNAC at our centre confirmed a rare case of rhinosporidiosis that was clinically mimicking a soft tissue neoplasm of the lower extremity, and the erroneous interpretation of the prior cytology studies had resulted in misinterpretation of the individually dispersed pathogenic organisms as individual malignant cells. FNAC of rhinosporidiosis can lead to early diagnosis and prompt treatment of this pathogen when it presents at unanticipated body sites.

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.

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.

Lacrimal sac rhinosporidiosis.

Rhinosporidiosis is a chronic mucocutaneous granulomatous disease caused by Rhinosporidium seeberi, involving primarily the nose and nasopharynx. Very rarely, the disease can affect the lacrimal sac. Here we report a 35-year-old male patient who had rhinosporidial involvement of the nose 5 years ago, for which he underwent endoscopic nasal surgery. Five years after the excision of the nasal mass, he presented with lacrimal sac involvement. The clinical presentation and the management of lacrimal sac rhinosporidiosis are discussed here.

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.

Nasal Rhinosporidiosis: Clinical Presentation, Clinical Findings, and Outcome in Dogs.

The purpose of this study was to describe the clinical presentation, imaging findings, and outcome in 10 dogs diagnosed with Rhinosporidium seeberi infections. Histopathology and cytology records were searched at a veterinary teaching hospital and a veterinary diagnostic laboratory to identify dogs with rhinosporidiosis. Medical records were reviewed for clinical, imaging, endoscopic, and surgical findings. Outcome was determined via evaluation of records and, where possible, telephone conversation with the primary care veterinarian and/or owner. Young to middle-aged large-breed dogs with an approximately equal sex distribution were represented. Unilateral signs predominated. Diagnosis was confirmed by histopathology in 9 cases, and cytology was diagnostic in only 1 of 3 cases. Histopathology was superior to cytology. Masses were soft tissue and contrast enhancing with no evidence of bony lysis on computed tomography (2 dogs). Direct or rhinoscopic (2 dogs) visualization revealed white to yellow pinpoint foci. Surgical resection (4 dogs) can result in long-term disease-free periods (up to 2659 days), although repeat surgery can be required. Dapsone was well tolerated in 1 dog, and relapse was not noted despite incomplete surgical resection (follow-up 749 days). Visualization of pale foci on a rostral intranasal mass in an endemic region should prompt consideration of rhinosporidiosis.

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.

Case Report: Rhinosporidiosis Literature Review.

Rhinosporidiosis is caused by Rhinosporidium seeberi, a pathogen currently considered a fungus-like parasite of the eukaryotic group Mesomycetozoea. It is usually a benign condition, with slow growth of polypoid lesions, with involvement of the nose, nasopharynx, or eyes. The clinical characteristics of a painless, friable, polypoid mass, usually unilateral, can guide the diagnosis, but the gold standard for diagnosis is histopathological findings. This article reviews the epidemiology, pathobiology, clinical manifestations, diagnostic strategies, and treatment approach for rhinosporidiosis.

An unexpected host in a soft-tissue lesion of thigh.

Rhinosporidiosis is an enigmatic entity and poses a major health problem in the developing countries of South-East Asia. A soft friable polypoid nasal mass is the most common presentation, while sparse literature is available on extranasal involvement. We describe the case of a 35-year-old female patient who presented with a slow-growing soft-tissue swelling with ulceration over the thigh. On clinical and radiological examination, a provisional diagnosis of soft-tissue neoplasm was made. After resection, histopathological sections showed a closely packed cyst with innumerable endospores. The present case report documents the rare occurrence of an incidentally detected cutaneous rhinosporidiosis causing diagnostic difficulty.

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.

External dacryocystorhinostomy for isolated lacrimal sac rhinosporidiosis - A suitable alternative to dacryocystectomy.

Purpose: To describe the outcome of external dacryocystorhinostomy (DCR) as a suitable alternative to dacryocystectomy (DCT) in cases of isolated lacrimal sac rhinosporidiosis. Methods: This was a retrospective, interventional case series. Institutional review board approval was obtained for the study. The chart review of 13 patients who underwent external DCR surgery for isolated lacrimal sac rhinosporidiosis between July 2012 and May 2018 was performed. Demographic details, clinical presentation, preoperative and intraoperative findings, surgical technique used, postoperative management, surgical outcome and duration of follow up were reviewed for each patient. Results: Thirteen patients were included in the study. Nine (69.2%) were male and four (30.8%) were female. The mean age was 22.1 years (range: 8-46 years). Involvement was unilateral in all cases. The commonest presenting complaint was epiphora with discharge seen in 10 cases (76.9%). An intrasac granuloma was grossly identified intraoperatively in 12 out of the 13 patients. Mean follow up was 26.9 months (range: 1.5-68 months). Till the last follow up, all cases were symptom free and did not have any evidence of recurrence of infection. Conclusion: From this series of cases reported by the authors, external DCR with appropriate precautions to prevent recurrence appears to be a suitable alternative to conventional DCT with excellent long-term outcomes without disease recurrence.