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| CASE REPORT |
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| Year : 2023 | Volume
: 3
| Issue : 2 | Page : 268-270 |
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Conjunctival rhinosporidiosis: A diagnostic surprise
Agrima Bhatia1, Santosh Kumar1, Jeenu Varghese2, Roohie Singh3
1 Department of Ophthalmology, Command Hospital Air Force, Bangalore, Karnataka, India
2 Department of Pathology, Command Hospital Air Force, Bangalore, Karnataka, India
3 Department of ENT, Command Hospital Air Force, Bangalore, Karnataka, India
| Date of Submission | 21-Oct-2022 |
| Date of Acceptance | 16-Jan-2023 |
| Date of Web Publication | 28-Apr-2023 |
Correspondence Address:
Agrima Bhatia
Department of Ophthalmology, Command Hospital Air Force Bangalore, Karnataka - 560 007
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/IJO.IJO_2768_22
Rhinosporidiosis is a rare ocular infection and a chronic granulomatous disease caused by Rhinosporidium seeberi. The disease is endemic in India and Sri Lanka but has been known to occur even in colder regions of North America and Eastern Europe. It is most commonly seen as a soft polypoidal pedunculated mass affecting mucus membranes of the nose, pharynx, and conjunctiva. We present a case of a 23 years male who presented with a flat, pedunculated, highly congested mass with a bumpy surface attached at the inferior border of the tarsal conjunctiva. It was initially suspected to be an epithelialized impacted foreign body but when completely excised and sent for histopathological examination revealed rhinosporidiosis. Diagnosis is based solely on microscopic features. Even though a rare clinical entity, it should be borne as a differential in case of any pedunculated conjunctival mass.
Keywords: Granulomatous, rare, pedunculated, Rhinosporidiosis seeberi, tarsal conjunctiva
How to cite this article:
Bhatia A, Kumar S, Varghese J, Singh R. Conjunctival rhinosporidiosis: A diagnostic surprise. Indian J Ophthalmol Case Rep 2023;3:268-70 |
Rhinosporidiosis is a rare ocular infection and a chronic granulomatous disease caused by Rhinosporidium seeberi. It is an organism classified as a protist in its own taxonomical class “mesomycetozoea,” “meso” – in middle of, “–myceto”–fungi, and “–zoea”– animals.[1] The disease is endemic in India and Sri Lanka but is also known to occur even in colder regions of North America and Eastern Europe.[2] It is most commonly seen as a soft polypoidal pedunculated mass affecting mucus membranes of the nose, pharynx, and conjunctiva. Other sites of infection include maxillary sinuses, urethra, and penis. Ocular rhinosporidiosis can affect eyelids, limbus, caruncle, canthi, lacrimal sac, and nasolacrimal duct. Scleral ectasia, staphyloma formation, and scleral melt have been seen in rare cases.[3] In this communication, we report an unusual location of the focus of rhinosporidiosis, i.e., tarsal conjunctiva.
| Case Report | |  |
Twenty-three years old male, resident of Kerala, reported to our department with complaints of foreign body sensation and itching in the left eye (LE) for 2 months duration. It was associated with a sticky, whitish discharge. There was no history of redness, photophobia, or watering. The patient had no prior history of any ophthalmic surgery but gave a nonspecific history of something entering his eye while driving before the onset of symptoms. He was a known case of bronchial asthma for 10 years, controlled on a steroid inhaler. On initial examination, uncorrected visual acuity was 6/6 in both eyes. The anterior segment examination of the LE revealed upper tarsal conjunctiva congestion and follicles on lid eversion. It also showed a 5 × 4 mm sized, flat, pedunculated, highly congested mass with a bumpy surface attached at the inferior border of the tarsal conjunctiva [Figure 1]a. It had grayish dots overlying the surface [Figure 1]b. The rest of the anterior and posterior segment was within normal limits (WNL). The right eye was essentially normal. Intraocular pressure by noncontact tonometer was 16 mm Hg in both eyes. Based on clinical appearance and history, a differential diagnosis of an impacted foreign body and pyogenic granuloma was made. He was planned for surgical excision; however, 2 days before excision, there was an episode of pinpoint bleeding from the lesion on patient's eye rubbing. The lesion was excised under local anesthesia, and light cautery was performed to arrest bleeding. Mass sent for histopathological examination unraveled a diagnostic surprise of rhinosporidiosis. Hematoxylin-eosin stain showed focal ulceration of hypertrophied lining epithelium, multiple sporangia of which few were ruptured, lying within reactive mixed inflammatory cell infiltrate [Figure 2]a and [Figure 2]b. Advanced pathological staining, periodic acid Schiff, and Grocott's stain highlighted large thick-walled intact and ruptured sporangia with numerous endospores within [Figure 2]c and [Figure 2]d. Postoperative period was uneventful, with mild congestion of the tarsal conjunctiva [Figure 1]c. The patient was commenced on Tab Dapsone 50 mg twice a day after baseline blood investigations and physician consultation. An otorhinologist's opinion was sought to rule out any additional lesions, but it was WNL. MRI brain and orbit done to look for any further lesions was also WNL. | Figure 1: (a) 5 × 4 mm, flat, pedunculated, highly congested mass with a bumpy surface attached at the inferior border of the tarsal conjunctiva. (b) Grayish, pinhead-sized spots on the surface suggestive of mature sporangia. (c) First postoperative day revealing mildly congested tarsal conjunctiva. (d) Six months post operatively showing no recurrence of lesion
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 | Figure 2: (a and b) Photomicrograph of H and E 20× and 40×, respectively, shows different stages of sporangia maturation (red arrow: intact, blue arrow: ruptured, green arrow: intervening stages). (c and d) PAS stain (10×) and Grocott's methanamine silver stain (10×), respectively, highlight large thick-walled intact and ruptured sporangia with numerous endospores within (red arrow: intact, green arrow: ruptured)
Click here to view |
The patient has been followed up for 6 months, but there is no recurrence or any fresh lesion [Figure 1]d.
| Discussion | | |
It is extremely rare for rhinosporidiosis to present as a conjunctival mass. Suh et al.[4] reported a case where rhinosporidiosis presented as chronic follicular conjunctivitis in a 15 years old contact lens wearer., Yellowish pin head sized spots on the surface, representing underlying mature sporangia, may help in pointing toward a possible diagnosis of rhinosporidiosis, but histopathology establishes a definitive diagnosis. Also, the site of infection may decide the configuration of the lesion. The bulbar conjunctiva may have a flatter and sessile lesion in view of pressure exerted by the upper eyelid.[3] In our case, the mass was arising from the tarsal conjunctiva, but probably owing to lack of space to grow outward, it may have assumed a flatter configuration.
Traumatized epithelium makes transmission feasible when the former comes in contact with contaminated water/soil. Subsequently, mass of polypoid traits, which is pedunculated and associated with bleeding, pruritis, or foreign body sensation, may develop.[5] Leading questions revealed the use of the village swimming pool on multiple occasions during the last 6 months, which probably was contaminated. Frequent rubbing of the eyes due to chronic pruritis may have caused a breach of epithelium, inviting the infection.
On histopathological examination, multiple sporangia were seen in various stages of degeneration, consistent with rhinosporidiosis. Some histological findings associated with infection and quoted in literature cite squamous metaplasia and epithelial hyperplasia.[2] Rhinosporidiosis diagnosis is based solely on its microscopic features. Treatment is surgical excision with cauterization at the base of the lesion. The largest case series of 462 rhinosporidiosis cases in Southwestern India reported that the disease occurs in the nose and nasopharynx (81.1%), while the eyes were affected in 14.2%.[1]
A case report of disseminated rhinosporidiosis showed 40% eosinophilia.[6] Although a case of ocular rhinosporidiosis, eosinophilia was noted in our case as well with bronchial asthma as the confounder. However, eosinophilia settled significantly from 19 to 2%, possibly attributable to 10 days of dapsone treatment.
Dapsone as an adjunct prevents the uptake of para-aminobenzoic acid by the microorganism by competitively inhibiting enzyme folate synthetase. It interferes with the synthesis of folic acid, which is essential for the formation of DNA, thereby decreasing the formation and maturation of spores. It also promotes stroma fibrosis causing the lesion to shrink.[5] In our case, no adverse effects of pharmacotherapy were noted. Ocular recurrences are typically rare, with a rate of 1% and between 2 and 40% for conjunctiva and lacrimal sac, respectively.[7] Causes of relapse are incomplete excision of lesion and seeding of endospores to adjacent sites during surgery. Ineffective cauterization and failure to use dapsone in the postoperative period may make the lesion more susceptible to relapses.[8] Though benign, it may be locally invasive and devastating.[9]
The initial differential of pyogenic granuloma was considered because of overlying grayish dots and that of an impacted foreign body was a consideration in view of possible epithelialization of the same over a period of time. Differential diagnosis in such cases must include conjunctival papilloma, hemangioma, arteriovenous malformation, and pyogenic granuloma.
| Conclusion | | |
Regardless of geographic location and the fact that it is a rare clinical entity, the possibility of rhinosporidiosis must always be borne in mind when evaluating any pedunculated or polypoidal conjunctival mass.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Acknowledgement
Dr. Shivapriya Manivannan for the acquisition of the initial pictures of the patient.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
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