|Year : 1969 | Volume
| Issue : 2 | Page : 59-63
Rhinosporidiosis manifesting as an unusual scleral staphyloma
PN Srinivasa Rao1, TT Ramalingam2
1 Kasturba Medical College, Manipal, India
2 Government Ophthalmic Hospital, Madras, India
|Date of Web Publication||8-Jan-2008|
PN Srinivasa Rao
Kasturba Medical College, Manipal
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Srinivasa Rao P, Ramalingam T T. Rhinosporidiosis manifesting as an unusual scleral staphyloma. Indian J Ophthalmol 1969;17:59-63
|How to cite this URL:|
Srinivasa Rao P, Ramalingam T T. Rhinosporidiosis manifesting as an unusual scleral staphyloma. Indian J Ophthalmol [serial online] 1969 [cited 2020 Jan 25];17:59-63. Available from: http://www.ijo.in/text.asp?1969/17/2/59/38432
Rhinosporidiosis is a specific fungus infection affecting mainly the mucous membrane and also the skin. Cases of rhinosporidiosis have been mainly reported from India, Ceylon and North American and occasionally from other countries. In India it is said to occur more commonly in the south, especially in the delta areas of Kerala and Tanjore. The lesions occur predominantly in the nasopharynx but it has been found at other anatomical sites also, including the ocular region. Lesions of lids, conjunctiva and lacrimal passages are frequently described.
A solitary scleral staphyloma is probably a rare manifestation of this disease. The following is the case report of a staphyloma like swelling of sclera presumably due to rhinosporidiosis and some of its peculiar features are discussed.
| Case report|| |
Patient A.R, a Muslim male 39 years old presented himself in the Kasturba Hospital, Manipal, on 17th September, 1960, with a history of a bluish swelling on the white of the right eye immediately outside the cornea. Apart from disfigurement, his other complaints were frequent attacks of mild redness in that eye associated with some irritation.
The swelling had a very insidious onset. It was of about 2 years' duration, very slowly increasing in size never receding, not associated with loss of vision, bleeding or pain. For the frequent attacks of mild redness and irritation, he was using hydrocortisone eye ointment with neomycin interruptedly and it used to afford him some relief.
The patient typically belonged to the rural population of Kasargod, the northern-most part of Kerala State (an endemic area for rhinosporidiosis) and of late he had migrated to an urban area for business purpose. He had a "fleshy" growth at the present site of swelling, which was removed surgically at Kasargod in 1947. He had another "fleshy swelling" in the same area which was removed in 1959 in Jaffna, Ceylon.
Local Examination:-There was an irregularly oval swelling in the sclera 7 mm from the limbus, mainly in the outer lower quadrant of sclera, between the 9.30 and 6.30 o'clock meridians. The swelling was bluish in colour, with a well defined border. The surface was faintly lobulated, and was of a consistency less firm than that of the sclera. The conjunctiva was mobile over the swelling, but adherent at places. Its dimensions were 14 x 11 mm and the maximum height was 8 mm. In addition, the swelling showed an extension above for a distance of about 7 mm. This area was slightly raised and was faintly bluish. On closing the lids the swelling could be easily seen moving under the lids.
The regional glands were not enlarged and the liver was not palpable.
At the upper inner corner of the swelling there was a reddish conjunctival mass over it. It was 6 mm x 4 mm in dimensions, with 6 to 7 yellowish dots over it, of 1-2 mm diameter. The red mass of the conjunctiva was freely mobile over the bluish swelling underneath.
The tension was 20 mm of Hg., the vision was 6/6 without glasses (same as in the left eye) the fundus normal and no other abnormality A the eye was detected.
Manteaux test done with 1 in 1,000 dilution of old tuberculin was mildly positive with no significance. The other tests done were urine analysis, total W.B.C. count, differential count, E.S.R., and V.D.R.L. which were all non-contributory.
There was no evidence of rhinosporidiosis anywhere else on the body. The fluid after syringing of the lacrimal passages did not show any spores. A general examination by a consultant physician did not reveal any general disease such as tuberculosis, leprosy or syphilis.
On 13-9-1961, the red mass of conjunctiva was excised and the biopsy showed it to be rhinosporidiosis. The only benefit the patient got was relief from the recurrent attacks of redness and irritation. He was advised against the use of hydrocortisone with neomycin eye ointment and he found no necessity for the same now.
On 14-5-1963, the patient was reexamined. The vertical height was now about 10 mm. On lightly closing the lids, there was the danger of exposure. There was again the history of recurrent attacks of redness and irritation. Over the swelling, there reappeared a small conjunctival reddish mass 3 x 4 mm with yellowish dots over it. (Plate 1) Routine examination of the eye showed no other abnormality except that the vision R.E. was 6/9, with -0.75 Dcyl 20'=6/6. Vision L.E. was 6/6 (without glasses).
The fundus was normal in each eye even on repeated examination with dilated pupil.
The case was referred to the Government Ophthalmic Hospital, Madras. There the red conjunctival mass was excised and a conjunctival flapping was done to strengthen the staphyloma. (Plate 2). The biopsy of the excision specimen showed it to be rhinosporidiosis.
The patient was re-examined on 1-10-1963. The dimensions of the swelling were more or less the same as those during September 1960, and there was no fleshy conjunctival mass over it. The vertical height between scleral surface and apex of the staphyloma was about 8 mm.
| Discussion|| |
Rhinosporidiosis may be a sporozoa or phycomycetes. Attempts to culture the organism and inoculate it on animals have been mostly unsuccessful. The mode of infection is also not known. This may be either water-borne or air-borne. Macroscopically, the lesion appears as villous polypi, pedunculated or sessile, but with no induration at the base. Under the microscope, it is seen that the lesions are made up of granulation tissue containing plasma cells and giant cells in which are found cysts of mixed matter (sporangia), containing the fungus spherules. These morulas can sometimes be seen discharging into the surrounding granulation tissue.
The disease has a higher incidence in males than in females. It is also commonly found in children and young adults. It is said that the disease is more common in the Muslims. Most of the cases reported are from the nose, but nasopharynx, lacrimal sac, eyelids, maxillary antrum, urethra, conjunctiva, larynx, uvula, soft palate, jaw, ear, bronchus, glans penis, rectum and vagina may be affected sporadically. Patients may show multiple foci of infection. In the nose it assumes the form of a papillomatous mass of granulation tissue, pink or purple red in colour, with the surface dotted over with minute yellow or white spots which . are the sporangia. The growth bleeds easily to touch and there is usually a history of epistaxis. On the eye lids it appears as painless masses of exuberant villous polypi, pedunculated or sessile with no infiltration at the base. When the lacrimal sac is infected, it may be distended with creamy pus which gives rise to little pain and can be expressed into the nose. The punctum may be dilated. The lacrimal passage may be free. The fluid after syringing of the lacrimal sac may show the spores.
In the conjunctiva it may occur silently like granulation tissue, persisting after a spontaneous drainage from a chalazion. Often the tissue is subjected to removal several times before microscopic examination discloses the sporidial invader. It appears as a small pale pink granular nodule freely movable with the conjunctiva. The bulbar or the palpebral conjunctiva is usually involved. The lesion in the eye may be asymptomatic and may pass unnoticed. A growth of moderate size creates a foreign body sensation in the eye with or without infection of the conjunctiva, lacrimation or photophobia.
In the present case, the matter for discussion is the cause and nature of the scleral swelling. The cause of the scleral swelling may he (1) trauma, (2) inflammation, or (3) neoplasm.
(1). Undoubtedly trauma as a cause cannot be ruled out completely, since two operations were performed for fleshy growth in the same area where the present swelling is situated. However, trauma cannot be the sole factor. If it were so, usually one would expect the swelling to consist of thinned out sclera lined by atrophic choroid. In that case for such a huge staphyloma there must be structural damage inside the eye ball such as secondary glaucoma, atrophy of the uveal tract, iridodialysis, luxation of the lens, displacement of the cornea or hypotension. None of these were noticed, except a small degree of astigmatism. It may be that the trauma acted only as a contributory factor.
(2) Inflammation:-It is quite likely for the swelling to be a product of chronic inflammation. That the vision, apart from some astigmatism, the tension and the fundus were normal suggest that the structural damage to the inside of the eye was minimal. Such an event can occur only if the thinning of the sclera is associated with proliferative change in the sclera and probably in the outer part of the choroid. In addition, the swelling showed some regression in the third examination on 1-10-1963, favouring an inflammatory basis. It is also possible that there was proliferation of pigment cells and pigment accounting partly for the blue colour. The general examination and the special investigations done do not reveal any syphilitic or tuberculosis basis. On the other hand, the excised specimen of the conjunctival mass which appeared twice, showed rhinosporidiosis. Hence, it is reasonable to attribute the scleral staphyloma to the same. If the history is correct, the original infection must have occurred earlier than 1947 and the excision specimen of June 1963 has again shown the fungus. It means a persistence of the rhinosporidial infection for more than 16 years which is highly interesting. It is to be noted that for a prolonged period before September 1960, the patient had indiscriminately used hydrocortisone eye ointment and how far it has contributed to the local pathology is anybody's guess.
(3) Neoplasm:-A Neoplastic basis for the swelling was ruled out, since the swelling has shown definite regression as seen in the third examination on 1-10-1963. A malignant melanoma was suggested in 1960, but now it is ruled out since the swelling is localised, has been growing extremely slowly not showing any tendency for bleeding or new vessel formation or pain.
Thus the general impression is that the swelling is a scleral staphyloma due to rhinosporidiosis in which trauma might have played a part. Lack of intraocular damage was explained by the proliferative reaction occurring in the sclera and outer layers of the choroid. However, the matter can be settled ultimately only by a microscopic examination of the affected area. However, at present it is not possible since the vision is very good in that eye and any procedure taken in that direction may result in permanent damage of the eyeball.
| Summary|| |
The clinical features of an unusual scleral staphyloma are described and discussed. It is presumed that the cause of the same is rhinosporidiosis with probably trauma as a secondary factor.
| References|| |
SORSBY: Systemic Ophthalmology, 1958. Butterworth & Co., London, 273-274.
Anderson, W., and Byrnes, T.: A Case of Rhinosporidium of the Conjunctiva, Amer. J. Ophthal. 22:1383-1388, 1939.
Arnold, R., and Whildin, J.: Rhinosporidiosis of Conjunctiva Amer. J. Ophthal. 25:1227-1230, 1942.
Barnshaw, H., and Read, W.: Rhinosporidiosis of Conjunctiva, Arch Ophthal 24: 357, 1940.
Duke Elder, S. W.: Text book of Ophthal., Vol. II, 1946, pages 1.9591960 page 2045-2047.
Duggan: British J. of Ophthal. XII 526, 1928.
Elliot & Ingram: Ophthalmoscope X, 428, 1912.
Elles, N. B.: Rhinosporidium Seberi Infection in the Eye, Arch Ophthal 25: 969, 1941.
Griffey, E. W.: Rhinosporidiosis: A Case report, Amer. J. Ophthal. 22: 1389-1390, 1939.
Ingram, A. C.: Lancet 2:726, 1910. 430, 1912.
Kirkpatrick H.: Ophthalmoscope, 10:
Kirkpatrick: Ophthalmoscope XIV, 477. 1916.
Kurup: Proc. All India O. S. II, 104, 1916.
Rao N. and V.: Proc. All India O.S. II, 109, 1931.
Reddy, D. and Lakshminarayana, C.: Investigation into Transmission, Growth and Serology in Rhinosporidiosis Ind. J. Med. Res. 50: 363-137, (May) 1961.
Satyanarayana, C.: Rhinosporidiosis with a Record of 255 cases, Acta Otolaryng (Stockholm) 51: 348--366 (March) 1960.
[Figure - 1], [Figure - 2]