Download Successful management of keratomycosis in a case of uncontrolled

Successful management of keratomycosis in a case
of uncontrolled Ocular Cicatricial Pemphigoid
Hatem Kobtan MD FRCS (Ed) (Glasg) Dina kobtan MD MSC
Cairo University
The authors have no financial interest to disclose
ASCRS
San Francisco 2009
Purpose
 To report the management of refractory keratitis in a case of
undiagnosed advanced ocular cicatricial pemphigoid (OCP).
Material and Methods
 60 year old farmer presents with pain and
diminution of vision of two months
duration in his right eye .
 Examination revealed Rt central corneal
infiltration
with
feathery
margins,
extensive stromal vascularization and
descmetocele formation.
 A chronic cicatrizing conjunctivitis with
fornix
shortening,
symblepheron,
ankyloblepheron and multiple rubbing
lashes was noted in both eyes.
 Visual acuity of light perception in the Rt
eye ,6/60 in Lt eye.
 Corneal scraping with culture and sensitivity was performed on
the Rt eye.
 Conjunctival biopsy with immunohistochemistry was performed
on the Lt eye.
 Topical Amphotericin B and systemic Itraconazole were initiated
on clinical suspicion of fungal keratitis.
 Systemic immunosuppressives were instituted after the resolution
of keratitis and the diagnosis of OCP by immunohistochemistry.
Results
 Culture plates grew Aspergillus. The patient’s keratomycosis resolved
with topical and systemic antifungal therapy.
 Vision improved from hand motion to ambulatory vision after
resolution of the keratitis .
 Clinical suspicion of OCP was confirmed with immunohistochemistry
of conjunctival biopsy from the other eye which showed IgG at the
epithelial basement membrane zone and low dose systemic steroids
combined with azathioprine were started.
5-2-2005
Rt central corneal infiltration with feathery
margins, extensive vascularization and
descmetocele formation. Chronic cicatrizing
conjunctivitis with fornix shortening,
symblepheron, ankyloblepheron and multiple
rubbing lashes.
24-7-2005
Five months later following resolution of
the keratitis with formation of a
paracentral leucoma and the initiation of
Imunosuppressive therapy (low dose steroid
combined with azathioprine) to control of
the OCP activity.
31-7-2005
Lt eye showing thickening of the lid margin with
loss of lashes, ankyloblepheron, conjunctival
hyperemia, fornix shortening and corneal
neovascularization
11-6-2006
Lt eye 10 months later showing the resolution of
the lid and conjunctival inflammation following
systemic immunosuppressives (low dose steroid
and azathioprine)
Conclusion
 The compromised ocular surface in patients with uncontrolled OCP
places them at an increased risk of microbial keratitis.
 Proper diagnosis and selection of the appropriate anti-microbial
can be sight saving in cases of refractory keratitis associated with
advanced OCP.
 Systemic immuosuppression is mandatory once the infection is
controlled to limit the progression of the ocular surface disease.
 Our reported case is unique due to the difficulty in laboratory
confirmation of clinically suspected keratomycosis and the
relatively rare occurrence of OCP.
References
 Barbara Iaccheri, Manolette Roque, Tito Fiore, Thekla Papadaki, Benjamin
Mathew, Stefanos Baltatzis, Barry Emara, A. C. Tokarewicz, and C. Stephen
Foster. Ocular Cicatricial Pemphigoid, Keratomycosis, and Intravenous
Immunoglobulin Therapy. Cornea 2004;23: 819– 822.
 Karla J. Johns, Denis M. O’Day. Pharmacologic management of keratomycoses
Survey of ophthalmology 1988; 33: 178- 188.
 Valerie P.J. Saw, John K.G. Dart, Saaeha Rauz, Andrew Ramsay, Catey Bunce,
Wen Xing, Patricia G. Maddison, Melanie Phillips. Immunosuppressive
Therapy for Ocular Mucous Membrane Pemphigoid: Strategies and Outcomes
Ophthalmology 2008; 115: 253-261.