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).

Three unusual cases of parasites in eye.

A 17-year-old male patient presented with cellulitis and mass in the eye noticed approximately 3 months back. The mass was about 1 cm in size and situated at the limbus. All preoperative routine investigations were normal. Surgical exploration revealed a sub-conjunctival cystic mass near the lateral rectus muscle about 1 cm in diameter; the mass was excised. Gross pathological examination revealed a thin-walled cystic mass. There was a hard nodule in the center. Microscopy revealed a wall of cysticercosis. Scolex was also seen. Surrounding tissue revealed sparse acute and chronic inflammatory cells. The case was confirmed by CDC, Atlanta, and was also included in their departmental presentation as an interesting case. A 60-year-old lady presented with complaints of itching over the forehead and right eye for 5 days. She was prescribed steroid eyedrops and antihistaminics. The itching aggravated with eyedrops along with watering and foreign body sensation. On revisit, the ophthalmologist noticed a worm in the right upper subconjunctival space. The worm was carefully removed in toto and sent to the laboratory for identification. The worm was thin, cylindrical, 8-10 cm long and white in color. After microscopic and gross examination of the worm, it was identified as Dirofilaria spp. CDC (Atlanta) confirmed the diagnosis of Dirofilaria. The patient was treated with antihistaminics and was relieved of symptoms without recurrence. A 45-year-old male patient had a painless mass in the eye for the last 3 months. He had no systemic illness. He gave a history of swimming pool use during that time. The mass was excised and submitted for histopathology. Numerous globular cysts representing thick-walled sporangia containing numerous spores diagnostic of Rhinosporidiosis were seen.

C3a receptor antagonism as a novel therapeutic target for chronic rhinosinusitis.

Chronic rhinosinusitis with nasal polyps (CRSwNP) is an inflammatory disease with an unknown etiology. Recent studies have implicated the complement system as a potential modulator of disease immunopathology. We performed proteomic pathway enrichment analysis of differentially increased proteins, and found an enrichment of complement cascade pathways in the nasal mucus of individuals with CRSwNP as compared to control subjects. Sinonasal mucus levels of complement 3 (C3) correlated with worse subjective disease severity, whereas no significant difference in systemic C3 levels could be determined in plasma samples. Given that human sinonasal epithelial cells were the predominate sinonasal source of C3 and complement anaphylatoxin 3a (C3a) staining, we focused on their role in in vitro studies. Baseline intracellular C3 levels were higher in CRSwNP cells, and following exposure to Aspergillus fumigatus (Af) extract, they released significantly more C3 and C3a. Inhibition of complement 3a receptor (C3aR) signaling led to a decrease in Af-induced C3 and C3a release, both in vitro and in vivo. Finally, we found in vivo that C3aR deficiency or inhibition significantly reduced inflammation and CRS development in a mouse model of Af-induced CRS. These findings demonstrate that local sinonasal complement activation correlates with subjective disease severity, and that local C3aR antagonism significantly ameliorates Af-induced CRS in a rodent model.

Disseminated Cutaneous Rhinosporidiosis.

A 41-year-old man from the state of Chhattisgarh, India, had a previous history of nasopharyngeal rhinosporidosis for 27 years, for which he had surgical intervention seven times, most recently endoscopically 2 months earlier. For the last 8 months, he had noticed skin lesions on the shoulders and forehead. Of interest was a history of bathing in ponds as a child. Examination revealed a soft, skin-colored, strawberry-shaped papule on the forehead (Figure 1A), a papule with a hemorrhagic crust on the left shoulder (Figure 1B), and a crusted friable plaque on the right shoulder (Figure 1C). Endoscopy revealed a 1 cm × 1 cm red polypoidal mass on the lateral wall of the nasopharynx. A diagnosis of disseminated cutaneous rhinosporidiosis with nasal-nasopharyngeal rhinosporidiosis was considered.

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.

Comments on 'novel multidrug therapy for disseminated rhinosporidiosis, refractory to dapsone - case report'.

Disseminated rhinosporidiosis can be successfully treated with multidrug therapy. Orally administered drugs can take longer to reach the affected areas in the presence of haemorrhage, oedema and inflammation. Wherever possible, surgical excision followed by drug therapy would be more effective.

Laryngotracheal rhinosporidiosis.

Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi. It usually affects mucous membranes of the nose, nasopharynx, and ocular conjunctiva. Cutaneous, laryngeal, tracheal, genital, and bony dissemination is rare. Laryngotracheal involvement poses many diagnostic and therapeutic challenges. A 45-year-old South Indian man presented with complaints of a mass in both nostrils for 2 years, associated with progressive hoarseness of voice and difficulty in breathing for 6 months. Rhinosporidial lesions were seen bilaterally in the nasal cavity. Telescopic and fiberoptic laryngoscopic examinations showed reddish, strawberry-like masses with whitish spots on their surface involving the larynx and trachea. Computed tomography of the head and neck revealed soft-tissue mass lesions involving the bilateral nasal cavities and nasopharynx, extending to the oropharynx and involving the larynx and trachea. A preliminary tracheostomy was performed, followed by direct laryngoscopic excision of the laryngeal lesions and rigid-bronchoscopy-guided excision of the tracheal lesions. The patient was prescribed dapsone and advised to take it for 2 years. At 2 years of follow-up, there was no recurrence.

Novel multidrug therapy for disseminated rhinosporidiosis, refractory to dapsone ­ case report.

Rhinosporidiosis is a chronic granulomatous disorder, caused by Rhinosporidium seeberi endemic in India and Sri Lanka. The most common sites are the nasal mucosa and the nasopharynx and cutaneous lesions usually occur as a part of disseminated rhinosporidiosis. Dapsone has been frequently used in treating disseminated disease in immunocompetent individuals. Here we report a case of disseminated rhinosporidiosis in an immunocompromised individual on antiretroviral drugs, non-responsive to Dapsone and therefore treated with a multidrug therapy of Cycloserine, Dapsone and Ketoconazole with good response.

Cost-effectiveness of mometasone furoate nasal spray in the treatment of acute rhinosinusitis.

BACKGROUND: Acute rhinosinusitis is a common disease with an increasing incidence rate. It causes substantial costs to the individual and to society through healthcare consumption and absence from work. The use of antibiotics is widespread in the treatment of acute rhinosinusitis, but increasing bacterial resistance is an argument for restricting excessive use of antibiotics. AIMS: The aim of this study was to analyse the cost-effectiveness of mometasone furoate nasal spray (MFNS) compared with amoxicillin or non-active treatment of mild to moderate acute rhinosinusitis in a Swedish setting. METHODS: A cost-effectiveness model was developed to capture the costs and health-related quality of life (HRQoL) over a 15-day period. Acute rhinosinusitis was modelled as changes in the Major Symptom Score. The model takes on a societal perspective in a Swedish setting. Efficacy data were taken from a randomised clinical study. The model has three treatment arms: (A) MFNS 200 µg twice daily, (B) amoxicillin 500 mg three times daily, and (C) placebo. Information about resource utilisation and HRQoL was taken from a recent observational study. RESULTS: Costs were reduced and quality-adjusted life years were increased with MFNS 200 µg twice daily compared with amoxicillin 500 mg three times daily. MFNS was cost-saving or cost-effective compared with amoxicillin or non-active treatment in the sensitivity analyses regardless of the HRQoL measurement used. CONCLUSIONS: This study shows that treatment with MFNS 200 µg twice daily results in lower costs and improved HRQoL in acute rhinosinusitis compared with amoxicillin or self-medication.

Rhinosporidiosis in southwest Bengal.

Rhinosporidiosis is a non-contagious chronic granulomatous disease that is prevalent in southern India and Sri Lanka. It has been known for centuries, but the details of the disease and the precise manner of its transmission have, until recently, remained unknown. Our institution sees many cases of this disease and we investigate the management protocol and its recent advances and include a review of the published literature. A total of 152 patients who were treated at Bankura Sammilani Medical College were studied between 2005 and 2011. The most common age group affected were those aged between 11 and 20 years of age and the male-to-female ratio was 1.9:1. Three patients suffered recurrent disease - one experienced it on the same site and the others on distant sites. Eleven patients with inadequate excision in which the margins were not free from disease were treated with dapsone therapy without any reported recurrence. It is a common disease in southwestern West Bengal. Surgical excision with electrocoagulation of the base is the main treatment, and dapsone therapy is recommended in order to prevent recurrences in multiple sites of affection and inadequate surgically excised cases. Although the disease occurs sporadically in most parts of the world, we see many patients in our area.

Disseminated cutaneous with nasopharyngeal rhinosporidiosis: Light microscopy changes following dapsone therapy.

We report a case of disseminated cutaneous and nasopharyngeal rhinosporidiosis in a 48-year-old man. The patient complained of a 6-month history of six subcutaneous skin-coloured swellings on the body and a 5-year history of a pea-sized swelling on the inner aspect of the left lower eyelid. Histopathological examination from one of these lesions showed multiple globular cysts packed with endospores that were typical of rhinosporidiosis. We report this case because of its rarity and to describe the morphological effects of dapsone in promotion of a host inflammatory response to the organism.

Tectonic corneal graft for conjunctival rhinosporidiosis with scleral melt.

A 16 year old girl presented with irritation and watering of the right eye for 3 months. On examination, the superior perilimbal sclera was ectatic with incarcerated uveal tissue covered by conjunctiva. The conjunctiva showed discreet, yellow white mucoid spots. Excision biopsy of the conjunctiva showed subepithelial spherules of sporangia containing numerous endospores, suggestive of rhinosporidiosis. Diathermy was applied to flatten the staphyloma. The ectatic area was covered with a corneal patch graft. The patient was started on prednisolone acetate eye drops and oral dapsone for 6 months. Corneal graft was well incorporated and conjunctivalized by 3 months. Since the graft was not seen within the palpebral aperture, there was good cosmetic result. The corneal graft had the added advantage of transparency which allowed visualization of the underlying tissue to diagnose early recurrence. There was no recurrence at 6 months.

Patterns of rhinosporidiosis in Sri Lanka: comparison with international data.

One hundred forty-three cases of rhinosporidiosis, confirmed by smear or biopsy, treated in two major General Hospitals in Sri Lanka over a 14 year period (1995-2009) were analyzed in regard to their epidemiological, clinical, clinicopathological, immunological and microbiological features. Regional variations in incidence, age and sex distribution, bathing history, and histopathology were seen. Lacustrine waters were the commonest probable source of infection (84%). Rivers were a source of Rhinosporidium seeberi in Sri Lanka (11%) and domestic well water was a probable source in 5%. The epidemiological features, clinical presentations and histopathology were similar to those in other series. The antirhinosporidial antibody (mean) titers were IgM--142.1 and IgG--178.5, compatible with rhinosporidiosis of long duration. Mantoux positivity to PPD was found in 65% of normal Sri Lankans, by only 35% of patients with rhinosporidiosis. No outbreaks have been reported in Sri Lanka or India. No animal cases of rhinosporidiosis have been reported in Sri Lanka, although rhinosporidiosis in animals has been repeatedly documented in India.

Conjunctival rhinosporidiosis presenting as a cystic mass-an unusual presentation.

Rhinosporidiosis is endemic in India and Sri Lanka and parts of East Africa and South America. Cases of ocular rhinosporidiosis have also been reported from Nepal. Ocular rhinosporidiosis usually involves the conjunctiva and the lacrimal sac. It usually presents as a polypoidal mass. Cystic mode of presentation has not yet been reported in the literature. We herein report an unusual mode of presentation of ocular rhinosporidiosis presenting as a huge conjunctival cystic mass.

Primary lacrimal sac rhinosporidiosis with grossly dilated sac and nasolacrimal duct.

Rhinosporidiosis is a chronic infection of the mucous membranes of the upper respiratory tract. The authors report a case of primary lacrimal sac rhinosporidiosis with a grossly dilated sac and nasolacrimal duct as seen on CT dacryocystography. Despite a chronic infection of 12 years' duration, there was no involvement of conjunctiva, nasal, or nasopharyngeal mucosa.

Nasal rhinosporidiosis in a mule.

A mass was removed from the nostril of a mule that exhibited unilateral epistaxis and nasal discharge. Impression smears revealed oval structures consistent with spores of Rhinosporidium seeberi. Microscopically, the mass was composed of fibrovascular granulomatous tissue containing sporangia R. seeberi. Surgical excision and antifungal treatment proved curative.

Disseminated cutaneous rhinosporidiosis.

Rhinosporidiosis is a chronic granulomatous disorder caused by Rhinosporidium seeberi. It frequently involves the nasopharynx and occasionally affects the skin. We herewith report a 55-year-old man who has disseminated cutaneous rhinosporidiosis. He presents with multiple reddish lesions over the nose of 10 year's duration. In the past year, he develops skin lesions over the right arm and over back. Histopathological examination of the skin biopsy specimen from the representative cutaneous lesions shows hyperplastic epithelium with numerous globular cysts of varying shape, representing sporangia in different stages of development. His serology for HIV infection by ELISA is negative. On the basis of these clinical and histopathological findings, a diagnosis of nasal rhinosporidiosis with cutaneous dissemination is made.