Double trouble: Patient has fungal corneal ulcer, ocular surface squamous neoplasia
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Ocular surface squamous neoplasia is used to describe neoplastic epithelial abnormalities of the conjunctiva and cornea, ranging from squamous dysplasia to invasive squamous cell carcinoma.
The etiology of ocular surface squamous neoplasia (OSSN) is mainly due to exposure to ultraviolet radiation, immune suppression, HIV, HPV mutation or deletions of tumor suppressor gene p53.
Source: Dhivya Ashok Kumar, MD, FRCS, FICO, FAICO, Preethi Naveen, MS, Swetha Ravichandran, MBBS, DO, and Amar Agarwal, MS, FRCS, FRCOphth
OSSN coexisting with fungal corneal ulcer is a rare case scenario that has been sparsely reviewed in literature. Immune predisposition may be the common factor to cause both conditions. Here, we present a patient with the rare combination of fungal corneal ulcer with OSSN. We highlight the comprehensive evaluation and staged treatment plan to achieve a good clinical outcome.
Case summary
A 60-year-old man, a known case with uncontrolled diabetes for the past 8 years, presented to us with complaints of redness and pain in the left eye for 1 month duration. It was preceded by a history of dust particles falling into his left eye 4 weeks prior. The patient was diagnosed with fungal corneal ulcer by scraping and microbiological analysis and had been using a topical antifungal for almost 3 weeks before presentation to us. The patient had undergone cataract surgery in the same eye 25 years ago.
On examination, the patient’s best corrected vision was counting fingers in the left eye. On slit lamp biomicroscopy examination, the conjunctiva showed generalized congestion. There was a whitish-pink mass nasally extending from the 7 to 9 o’clock position. It measured about 7 mm × 5 mm, encroaching 2 mm to 2.5 mm of peripheral cornea with surrounding vessels, suggestive of OSSN (Figure 1). Central cornea revealed dense stromal infiltrate, 4 mm × 4 mm in dimension with grayish-white color and a dry surface, an immune ring suggestive of fungal corneal ulcer. Anterior chamber revealed flare and a 2-mm immobile hypopyon. There was an IOL in situ in the posterior chamber. Anterior segment OCT of the left eye revealed an epithelial hypertrophy corresponding to the limbal mass lesion with a sharp delineation between normal and abnormal epithelium. The thickened epithelium was observed as hyperreflective on OCT at the limbus and on the corneal surface (Figure 2).
OSSN excision
With the probable clinical diagnosis of OSSN with fungal corneal ulcer, the patient was planned for a staged procedure of initial left eye OSSN excision with a marginal clearance of 3 mm with amniotic membrane graft and histopathological examination. This was performed under sedation with local anesthesia. Subsequently, histopathology revealed hyperplastic squamous epithelium with mild dysplasia and confirmed the clinical diagnosis. The surgical margins were clear of tumor. Intraoperatively, repeat corneal ulcer scraping and debridement were done and sent for microbiological examination. The patient was kept on topical and oral antifungal medications.
Penetrating keratoplasty
The postoperative period was stable after OSSN excision, and the patient continued the topical antifungal. As there was no clinical improvement in corneal ulcer with medical management, the patient underwent therapeutic penetrating keratoplasty 6 weeks after OSSN removal. The patient responded well after the procedure with improved vision, and there were no signs of recurrence (Figure 3) at 3 months follow-up.
Management of rare combination
Concurrent OSSN and fungal corneal ulcer is rare. With few previous case reports on the topic, it has to be remembered that immunological decompensation can induce both. Patients with chronic ocular infections should be examined and followed closely for abnormally thickened limbal lesions. OSSN is a disease affecting the limbal stem cells and can have conjunctival, corneal or conjunctival-corneal involvement. The patient usually presents to the outpatient department with a slow-growing ocular lesion, and it requires a high index of clinical suspicion to diagnose it. However, timely and adequate medical and surgical treatment usually salvages the eye in the majority of cases. Other ocular predispositions include chronic exposure, lagophthalmos in seventh nerve palsy, periocular ichthyosis as part of systemic congenital ichthyosis, and corneal scar or degenerations.
In the concomitant presence of OSSN and fungal corneal ulcer, few studies have showed treating the ulcer component first followed by OSSN excision. We did the OSSN excision first to prevent the spread of the tumor followed by treatment of the fungal ulcer, and the patient responded well and had a good postoperative period.
Immunological change locally in the cornea may be a factor as patients with such combinations are usually immunosuppressed. In our case report, the patient also had uncontrolled diabetes. Therefore, it is essential to rule out immunosuppressive status in long-standing or nonresponding infections. After the occurrence of fungal keratitis, the treating ophthalmologist directed the treatment toward the ulcer and failed to recognize OSSN. The patient presented to us in a stage of nonhealing ulcer. The challenges we faced included the clear margin excision, which may be difficult on the corneal side due to ulcer infiltrate. Moreover, there is always a risk for extension of infiltrate into the corneal postoperative wound. A good preoperative anterior segment OCT will aid in delineation of the narrow margin between the two lesions. Additionally, necessary measures are to be taken to give proper preoperative antifungal to reduce the load of organisms, and surgical debridement with microbiological analysis is needed to confirm diagnosis. One should also make sure not to implant tumor cells in ulcerative cornea and follow a “no-touch” technique of tumor excision. Although necessary precautions are taken preoperatively and intraoperatively, there still can be recurrence. Hence, good postoperative follow-up is mandatory in such scenarios.
Conclusion
Comorbidity of fungal corneal ulcer with OSSN is a rare entity. Chronic ocular infections should be examined and followed closely for abnormally thickened limbal areas. Early detection and proper planning can give the patient good functional results.
- References:
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- For more information:
- Amar Agarwal, MS, FRCS, FRCOphth, director of Dr. Agarwal’s Eye Hospital and Eye Research Centre, is the author of several books published by SLACK Books, sister company of Healio publisher Ocular Surgery News, including Phaco Nightmares: Conquering Cataract Catastrophes, Bimanual Phaco: Mastering the Phakonit/MICS Technique, Dry Eye: A Practical Guide to Ocular Surface Disorders and Stem Cell Surgery and Presbyopia: A Surgical Textbook. He can be reached at aehl19c@gmail.com; website: www.dragarwal.com.
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