Polymicrobial and microsporidial keratitis in a patient using Boston scleral contact lens for Sjogren's syndrome and ocular cicatricial pemphigoid

doi:10.1016/j.clae.2012.10.082

Contact Lens and Anterior Eye

Volume 36, Issue 2, April 2013, Pages 95-97

https://doi.org/10.1016/j.clae.2012.10.082 Get rights and content

Abstract

Aim

To report a rare case of microsporidial and polymicrobial keratitis in a patient with Sjogren's syndrome and ocular cicatricial pemphigoid.

Method

This is a descriptive case report. A 66-year-old lady diagnosed with Sjogren's syndrome (SS) and ocular cicatricial pemphigoid (OCP) presented to us with microbial keratitis after using a Boston sclera contact lens for a painful epithelial defect. After 9 days of medical treatment, she underwent therapeutic penetrating keratoplasty.

Results

10% potassium hydroxide and calcofluor white wet mount revealed microsporidial spores. Gram positive cocci and Gram variable bacilli on Gram stain were identified as Staphylococcus epidermidis and Corynebacterium accolens in culture. Histopathological examination of the corneal tissue confirmed the presence of microsporidial spores.

Conclusion

Microsporidal keratitis can occur in patients with severe ocular surface disease due to SS and OCP. Predisposing factors include dry eye, local and systemic immunosuppression and Boston scleral contact lens. Early surgical intervention may be needed to eradicate the infection.

Introduction

Sjogrens syndrome is a systemic autoimmune disease occurring more commonly in females and affecting the lacrimal and salivary glands causing dry eye and dry mouth [1]. Ocular cicatricial pemphigoid is a chronic cicatrizing autoimmune disease of the mucous membrane and skin occurring predominantly in females, due to auto antigens directed against α4β6 integrin in the epithelial basement membrane [2]. Each of these conditions has severe ocular surface disease requiring preservative free artificial tears, topical cyclosporine A, punctal plugs [3] and scleral contact lenses [4]. It is believed that these patients are at a higher risk of developing microbial keratitis [5].

Microsporidia are ubiquitous organisms causing systemic and ocular infections in humans [6]. Ocular features include superficial keratoconjunctivitis in immuno-suppressed persons and stromal keratitis in immuno-competent persons [6].

We describe the rare occurrence of microsporidial stromal keratitis with polymicrobial keratitis in a patient with secondary Sjogren's syndrome and ocular cicatricial pemphigoid who was using a Boston scleral contact lens.

Section snippets

Case report

A 66-year-old lady was diagnosed with ocular cicatricial pemphigoid (OCP) and secondary Sjogrens’ syndrome (SS) in 2004 and presented to the Cornea services at L V Prasad Eye Institute, Hyderabad, India with complaints of dryness in both eyes and loss of vision in the left eye. She had positive immunohistochemistry for OCP and the triad of dry eye, dry mouth and sero-negative rheumatoid arthritis with positive anti Ro and anti La titers and a positive lip biopsy. Her best-corrected visual

Discussion

Our patient was an HIV negative lady with Sjogren's syndrome and OCP, who developed a rapidly progressive polymicrobial keratitis with S. epidermidis, C. accolens and Microsporidia following use of a Boston scleral contact lens.

Patients with ocular surface disease are at an increased risk of developing microbial keratitis [5]. S. epidermidis and Corynebacterium spp. are commensals present in the conjunctival sac [9] and hence this could be a potential source of infection in patients with severe

Conflict of interest

The authors have no financial or proprietary interest in any of the methods or materials used in this study.

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Case reportPolymicrobial and microsporidial keratitis in a patient using Boston scleral contact lens for Sjogren's syndrome and ocular cicatricial pemphigoid

Abstract

Aim

Method

Results

Conclusion

Introduction

Section snippets

Case report

Discussion

Conflict of interest

American Journal of Ophthalmology

Ophthalmology

Ophthalmology

Survey of Ophthalmology

American Journal of Ophthalmology

Classification criteria for Sjogren's syndrome: a revised version of the European criteria proposed by the American-European Consensus Group

Annals of the Rheumatic Diseases

Cicatricial pemphigoid

Advances in understanding and managing dry eye disease

American Journal of Ophthalmology

Ocular cicatricial pemphigoid review

Current Opinion in Allergy and Clinical Immunology

Case report
Polymicrobial and microsporidial keratitis in a patient using Boston scleral contact lens for Sjogren's syndrome and ocular cicatricial pemphigoid