Long-term follow-up of laryngeal Rhinosporidium seeberi diagnosed by PCR and treated with laser ablation and voriconazole nebulization in a retired thoroughbred polo horse.

A 21-year-old retired polo Argentinian thoroughbred horse from a teaching herd was presented for a routine bronchoalveolar lavage demonstration, during which an incidental finding of a granulomatous mass on the dorsal aspect of the epiglottis was made. Rhinosporidium seeberi was suspected from a histological section obtained from an initial biopsy, and the mass was removed via laser surgery for cytology and PCR. Sequencing of the PCR amplicons confirmed the diagnosis of R. seeberi. A treatment protocol of nebulized voriconazole for 10 d postoperatively was used. Long-term follow-up required 2 more laser surgeries plus oral fluconazole to resolve the remaining fungal spores. However, 2.5 y later, there was no evidence of remaining fungal spores. Key clinical message: Horses from endemic regions can potentially be exposed to R. seeberi. Based on its travel history, this horse may have contracted the infection in South America, California, or Alberta. Treatments administered, including diode laser resection, voriconazole antifungal nebulization, and oral fluconazole administration, were successful but required repeated interventions.

Suivi à long terme du Rhinosporidium seeberi laryngé diagnostiqué par PCR et traité par ablation au laser et nébulisation au voriconazole chez un cheval de polo thoroughbred pur-sang à la retraiteUn cheval thoroughbred argentin de polo retraité de 21 ans, issu d'un troupeau d'enseignement, a été présenté pour une démonstration de lavage broncho-alvéolaire de routine, au cours de laquelle une découverte fortuite d'une masse granulomateuse sur la face dorsale de l'épiglotte a été faite. Rhinosporidium seeberi a été suspecté à partir d'une coupe histologique obtenue à partir d'une biopsie initiale, et la masse a été retirée par chirurgie au laser pour cytologie et PCR. Le séquençage des amplicons PCR a confirmé le diagnostic de R. seeberi. Un protocole de traitement au voriconazole nébulisé pendant 10 jours après l'opération a été utilisé. Le suivi à long terme a nécessité 2 autres interventions chirurgicales au laser et du fluconazole oral pour éliminer les spores fongiques restantes. Cependant, 2,5 ans plus tard, il n'y avait aucune trace de spores fongiques restantes.Message clinique clé:Les chevaux des régions endémiques peuvent potentiellement être exposés à R. seeberi. D'après ses antécédents de voyage, ce cheval pourrait avoir contracté l'infection en Amérique du Sud, en Californie ou en Alberta. Les traitements administrés, notamment la résection au laser à diode, la nébulisation antifongique au voriconazole et l'administration orale de fluconazole, ont été efficaces mais ont nécessité des interventions répétées.(Traduit par Dr Serge Messier).

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.

[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.

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.

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.

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.

Naso-Bronchial Rhinosporidiosis.

Rhinosporidiosis is a chronic mucocutaneous granulomatous disease caused by Rhinosporidium seeberi, commonly affecting the nose and nasopharynx. Endobronchial involvement is of rare occurrence but can pose challenging problems for diagnosis, surgical excision and anaesthetic management. We report a 40-year-old man with a history of recurrent nasal rhinosporidiosis who presented with unilateral nasal obstruction, cough, shortness of breath and a radiological feature of left lung collapse. Eight years since the last surgery, he presented with a recurrent lesion in the nose with concurrent endobronchial involvement. The patient underwent excision of the nasal and the endobronchial lesion successfully under general anaesthesia without any complication and good symptomatic improvement. The clinical presentation and the management of endobronchial rhinosporidiosis are discussed here. The surgical difficulties faced during the procedure are highlighted.

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.

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.