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.

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.

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.

Calvarial involvement in disseminated rhinosporidiosis - A case report and literature review.

Rhinosporidiosis is a chronic granulomatous infection caused by Rhinosporidium seeberi and mainly involves nasal and ocular mucosa. Bony involvement in rhinosporidiosis is very rare. A young male, previously operated for nasal rhinosporidiosis, presented with two bony swellings on the forehead and multiple subcutaneous lesions on the right lower limb. The diagnosis of disseminated cutaneous rhinosporidiosis with frontal bone involvement was made with the help of fine needle aspiration cytology (FNAC), histopathology, and computed tomography (CT) scan head. Wide excision of the bony lesion was performed. To the best of our knowledge, this is the first radiologically proven case of frontal bone involvement in disseminated rhinosporidiosis. Early diagnosis can be established with a good clinicopathological and radiological correlation. It also emphasizes the importance of CT scan for the evaluation of any subcutaneous skull lesion.

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.

Long-term use of Le Fort I osteotomy for the management of nasopharyngeal rhinosporidiosis: A case series.

Rhinosporidiosis is a rare, chronic, granulomatous infection of the mucous membranes that mainly involves the nose and nasopharynx; it occasionally involves the pharynx, conjunctiva, larynx, trachea and, rarely, the skin. The characteristic clinical features of this disease include the formation of painless polyps in the nasal mucosa or the nasopharynx that bleed easily on touch. At our center, excision of the lesion with a Le Fort I osteotomy is carried out in patients (1) in whom two or more previous attempts at excision of biopsy-proven rhinosporidiosis arising from the nasal mucosa was carried out or (2) in whom the rhinosporidiosis arises from the nasophayrngeal mucosa and/or extranasal sites. In this article we retrospectively present 7 cases in which, according to our inclusion criteria, complete excision of the lesion was carried out with a Le Fort I osteotomy. Excellent visualization of the entire maxillary and ethmoidal air cells after the down-fracture of the maxilla helped in the total removal of the lesions. Most of these lesions had multiple points of origin through the nasal, maxillary, and ethmoidal mucosa; the excellent visualization enabled direct cauterization of all these points of origin. The mean follow-up period was 7.96 years, and all patients were disease-free by the time the study was prepared. This article presents details of the treatment protocol and technique followed at our center for the treatment of nasopharyngeal rhinosporidiosis and the details of long-term follow-up. Through this study we hope to prove the efficacy of Le Fort I osteotomy in the definitive management of nasopharyngeal rhinosporidiosis.

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.

Partial regression of large anterior scleral staphyloma secondary to rhinosporidiosis after corneoscleral graft - a case report.

BACKGROUND: Rhinosporidiosis is a rare chronic infection of the mucous membranes caused by the Rhinosporidium seeberi. Approximately 15% of cases of rhinosporidiosis are ocular, occurring mainly in the tarsal conjunctiva. There are only 11 cases of scleral melt with staphyloma formation associated with bulbar conjuctival oculosporidiosis and none of them was associated with partial regression of the scleral ectasia after a corneoscleral tectonic graft. CASE PRESENTATION: a 13-year-old girl with a progressively increasing black mass in the upper nasal part above the cornea of the left eye. The biomicroscopy revealed an oval, bluish mass measuring 10x10x5 mm with congestion of the overlying conjunctiva. Conjunctival biopsy showed sporoblasts of Rinosporidium seeberi. Treatment was conducted by conjunctival resection and tectonic corneoscleral graft (13x13mm) over the staphyloma. Within 1 year of follow-up the patient presented a partial staphyloma reduction, 9x9x2.5 mm, and the patch detached from the lesion. A novel surgical approach was done reducing the corneal patch and no recurrence was seen after 9 months. CONCLUSIONS: This case is one of the largest anterior scleral staphylomas secondary to rhinosporidiosis described in the literature. Scleral anterior staphyloma partial regression is an unusual outcome after a tectonic corneoscleral graft. Infection resolution and graft covering of thinned area contributed to scleral reepithelization.

Rare case of disseminated rhinosporidiosis with chronic osteomyelitis of the calcaneum treated by a simple technique of negative pressure wound therapy.

Occurrence of nasal, cutaneous and disseminated rhinosporidiosis in the same patient is uncommon and involvement of calcaneum is rare. Rhinosporidial osteomyelitis is treated surgically, but multiple recurrences and local spread are common. Many a times, surgical ablation or amputation of the involved area of the limb is carried out to prevent recurrences. We present a case of rhinosporidial osteomyelitis of the calcaneum with a chronic discharging sinus and large lytic lesion in the calcaneum treated by an alternative surgical option in the form of debridement and negative pressure wound therapy.

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.