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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 14-16

Conjunctival oculosporidiosis-associated ciliary staphyloma, managed with scleral patch graft using box sutures


1 Department of Cornea, Refractive, Ocular Surface Services, Sankara Nethralaya, Kolkata, West Bengal, India
2 Department of Cornea, Refractive, Ocular Surface Services, Batra Hospital and Medical Research Centre, New Delhi, India
3 Consultant, School for Excellence for Eye, Indore, Madhya Pradesh, India
4 Department of Uveitis and Ocular Pathology, Sankara Nethralaya, Chennai, Tamil Nadu, India

Date of Submission06-Mar-2021
Date of Acceptance08-Jul-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Mona Bhargava
Aditya Birla Sankara Nethralaya, 147, Mukundapur, EM Bypass, Kolkata - - 700 099, India
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_532_21

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  Abstract 


To report four cases of oculosporidiosis associated with scleral thinning and staphyloma formation, including recurrent bulbar conjunctival oculosporidiosis, retrospective reviews of case records of four patients with bulbar conjunctival oculosporidiosis were undertaken. All cases were managed with excision of growth followed by diathermy and scleral patch graft. Box sutures were used for securing the graft in place, followed by oral dapsone therapy. This case series describes successful management of oculosporidiosis and associated scleral ectasia even in a recurrent case. It also highlights the utility of box sutures for securing scleral patch grafts.

Keywords: Box suture, recurrence, rhinosporidiosis, oculosporidiosis, staphyloma


How to cite this article:
Bhargava M, Johri A, Ghosh AD, Paul RS, Gupta R, Biswas J. Conjunctival oculosporidiosis-associated ciliary staphyloma, managed with scleral patch graft using box sutures. Indian J Ophthalmol Case Rep 2022;2:14-6

How to cite this URL:
Bhargava M, Johri A, Ghosh AD, Paul RS, Gupta R, Biswas J. Conjunctival oculosporidiosis-associated ciliary staphyloma, managed with scleral patch graft using box sutures. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Jun 29];2:14-6. Available from: https://www.ijoreports.in/text.asp?2022/2/1/14/334956



Rhinosporidiosis is a rare chronic infection of the mucous membrane caused by a Mesomycetozoa, Rhinosporidium seeberi. The commonly affected sites are the nose and nasopharynx. Approximately 15% of cases of rhinosporidiosis are ocular, with tarsal and bulbar conjunctiva being the most common site, followed by the lid and lacrimal gland.[1] History of frequent pond baths in endemic areas is a major risk factor with the presumed mode of infection being from the aquatic habitat through the traumatized epithelium.[2]

Bulbar oculosporidiosis usually presents as polypoidal and vascular masses. Definitive diagnosis is achieved by microbiological scrapings and histopathological examination (HPE) of resected tissues. Scleral melting and staphyloma formation associated with bulbar conjunctival rhinosporidiosis has been seldom reported in the literature.[1],[3],[4]


  Case Reports Top


Description of four oculosporidiosis cases with similar clinical presentations and surgical outcomes are enumerated under the following headings.

A. History:

We report four cases presenting with complaints of uniocular diminution of vision and a history of progressively increasing ocular mass. Demographic details are enumerated in [Table 1].
Table 1: Description of cases

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There was a positive history of frequent pond baths in all the cases. Case 4 had an associated surgical history of excision biopsy with scleral patch graft for rhinosporidial conjunctival mass associated with ciliary staphyloma 14 years back (previous medical records). Currently, he has presented with complaints of similar mass and bluish scleral discoloration in the same eye for the past two years.

B. Examination:

On slit-lamp examination, a reddish pedunculated conjunctival granuloma with multiple yellow-white dots (spherules) and dilated conjunctival vessels was noted in all four eyes [Figure 1]a, [Figure 1]c, [Figure 1]e, [Figure 1]g. The investigative details are mentioned in [Table 1]. In all the cases, anterior segment examination of the unaffected eye was unremarkable. Intraocular pressure and retinal examination of all eyes were within normal limits. No abnormalities were detected in the otorhinolaryngological evaluation of these patients.
Figure 1: Slit-lamp examination: pre-operative (a, c, e, g) and post-operative images (b, d, f, h) showing the rhinosporidial mass, staphyloma, and well-placed scleral patch graft, respectively

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C. Ultrasound biomicroscopy (UBM):

UBM of the affected eye revealed areas of scleral thinning with ectasia correlating with clinically noted staphylomatous areas. Moderate reflective pedunculated mass lesions arising from the margin of the ectasia were noted in all the cases.

Diagnosis – All the cases were provisionally diagnosed as bulbar conjunctival oculosporidiosis associated with ciliary staphyloma.

Surgical technique – Excision biopsy with scleral patch graft was planned for all the cases.

Intraoperatively, the conjunctiva over the pedunculated growths was dissected and the overlying growths were removed from the base. The excised specimens were sent for HPE, and the bases were cauterized. Epithelium over the staphyloma was removed and minimal energy diathermy was applied to achieve adequate shrinkage of the staphyloma with the care of not causing any scleral perforation. Donor scleral patch graft was prepared according to staphyloma sizes measured intraoperatively [Table 1]. Fibrin glue was applied over the staphylomatous area and the donor full-thickness scleral graft was sutured over the area using 6-0 vicryl box sutures [Figure 2]. The dissected conjunctiva was reposited back. In cases 3 and 4, the conjunctival cover was inadequate as case 3 had an exposed inferotemporal location and case 4 being recurrent had scarred conjunctiva. In both patients, sclera was covered using an amniotic membrane graft. A similar surgical technique was followed by all three performing surgeries [Figure 1]b, [Figure 1]d, [Figure 1]f, [Figure 1]g.
Figure 2: Intraoperative images: (a) post mass excision diathermy-induced shrinkage of staphyloma, (b) scleral patch graft being placed before and (c) after suturing, (d) multilayered amniotic membrane placed over the scleral patch graft, (e) circular scleral patch graft secured with box sutures, and (f) schematic of [Figure 2]e.

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Postoperatively, all patients were treated with topical antibiotics and steroids. Oral tablet dapsone was started 100 mg/day for 6 months, after ruling out G-6-PD deficiency.

D. Follow-up –

The scleral patch graft was well taken up without any signs of oculosporidiosis recurrence on follow-up [Table 1].

E. Histopathology -

Excised specimens showed conjunctival tissue with irregular epithelium. Subepithelial tissue comprised multiple cysts containing spores suggestive of sporangia. The cysts were typical of rhinosporidium spores in different stages of maturity (multiple sporoblast and sporocyst). There was intense infiltration of lymphocytes, plasma cells, and foci of multinucleated giant cells [Figure 3].
Figure 3: Microphotograph of the conjunctival tissue showing sporangia (A) and sporoblast (B) of rhinosporidiosis with lymphocytic infiltration (Haematoxylin and eosin ×200).

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  Discussion Top


Rhinosporidiosis is a rare chronic infection of the mucous membrane; ocular structures are second most commonly involved after the nasal mucosa. The classical conjunctival lesion in oculosporidiosis presents as a red, fleshy, pedunculated polypoid lesion in the palpebral conjunctiva with multiple yellow dots representing the sporangia on the surface.[5]

Staphyloma formation is a rare manifestation of oculosporidiosis.[4] It has been postulated that enzymes produced by the organism corrode the sclera, resulting in thinning and staphyloma formation.[6] The staphyloma gradually increases in size and may ultimately perforate, resulting in the loss of the eyeball.

The thinned and ectatic scleral area predisposes the eye to injury and infection. Thus, there is a need for a patch graft over the area. Donor scleral grafts have the advantages of being readily available, strong, and easily preservable.[7] Amniotic membrane transplantation has been used in conjunction with scleral patch grafts when there is associated conjunctival deficiency as it promotes re-epithelialization and reduces the chance of scleral melt.[8]

We advocate the use of box sutures instead of commonly employed interrupted sutures for securing scleral patch grafts. Box sutures can be tightened in a titrated manner whereas the interrupted sutures may give way or cut through the graft. Box sutures provide more tensile strength as the vector force of pull of the suture is divided along both arms of the box. In addition, there is a broader intragraft course that helps in providing better tamponade of the graft to the thinned-out area. Suture weave technique with the placement of mattress sutures carries similar advantages.[9]

Recurrences are common in ocular adnexal rhinosporidiosis; however, conjunctival rhinosporidiosis recurrence with staphyloma is a rare entity.[10] Conjunctival recurrences are seen in case of inadequate excision or intraoperative inadvertent seeding of spores to a new site.


  Conclusion Top


Conjunctival rhinosporidiosis can cause scleral ectasia in young and healthy individuals. HPE of excised sample is definitive. Box sutures with greater tensile strength and tamponade result in better post-operative stability. The long-term efficacy and reproducibility of this surgical maneuver are demonstrated by the fact that the aforementioned cases were operated by different surgeons for the same indication, and all of them achieved desired tectonic stability and ensured no recurrences across the follow-up period.

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.

Financial support and sponsorship

Nil

Conflicts of interest

There are no conflicts of interest



 
  References Top

1.
Lamba PA, Shukla KN, Ganapathy M. Rhinosporidium granuloma of the conjunctiva with scleral ectasia. Br J Ophthalmol 1970;54:565-8.  Back to cited text no. 1
    
2.
Reidy JJ, Sudesh S, Klafter AB, Olivia C. Infection of the conjunctiva by Rhinosporidium seeberi. SurvOphthalmol 1997;41:409-13.  Back to cited text no. 2
    
3.
Castelino AM, Rao SK, Biswas J, Gopal L, Madhavan HN, Kumar SK. Conjunctival rhinosporidiosis associated with scleral melting and staphyloma formation: Diagnosis and management. Cornea 2000;19:30-3.  Back to cited text no. 3
    
4.
De Doncker RM, de Keizer RJ, Oosterhuis JA, Maes A. Scleral melting in a patient with conjunctival rhinosporidiosis. Br J Ophthalmol 1990;74:635-7.  Back to cited text no. 4
    
5.
Chowdhury RK, Behera S, Bhuyan D, Das G. Oculosporidiosis in a tertiary care hospital of western Orissa, India: A case series. Indian J Ophthalmol 2007;55:299-301.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Kuriakose ET. Scleral transplantation in scleral staphylomas caused by Oculosporidiosis. Indian J Ophthalmol1979;27:227-8.  Back to cited text no. 6
    
7.
Sangwan VS, Jain V, Gupta P. Structural and functional outcomes of scleral patch grafts. Eye 2007;21:930-5.  Back to cited text no. 7
    
8.
Ma DH, Wang SF, Su WY, Tsai RJ. Amniotic membrane graft for the management of scleral melting and corneal perforation in recalcitrant infectious scleral and corneoscleral ulcers. Cornea 2002;21:275-83.  Back to cited text no. 8
    
9.
Wen JC, Lam J, Banitt MR. Scleral patch graft with a suture reinforcement technique in surgical management of necrotizing scleritis with ectasia. Cornea 2018;37:933-5.  Back to cited text no. 9
    
10.
Kuriakose ET. Oculosporidiosis: Rhinosporidiosis of the eye. Br J Ophthalmol 1963;47:346-9.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
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