Abstract
Purpose
The aim of this report was to describe a case of cataract surgery and Descemet stripping automated endothelial keratoplasty (DSAEK) after cytomegalovirus (CMV) corneal endotheliitis and bullous keratopathy (BK) following immunosuppressive treatment for Mooren’s ulcer.
Observations
A 64-year-old man was referred to our hospital because of peripheral ulcerative keratitis in his left eye. He had a history of trabeculectomy for open angle glaucoma in his left eye. He was diagnosed with Mooren’s ulcer and treated with topical betamethasone and tacrolimus with systemic cyclosporine. The corneal ulcer improved, but the peripheral cornea thinned from 6 to 12 and 0–2 o’clock. Five months later, cells were observed in the left anterior chamber, and real-time polymerase chain reaction examination of the aqueous humor showed CMV-DNA-positive results. The patient was diagnosed with CMV corneal endotheliitis, and oral ganciclovir was administered. Fifteen months after the initial presentation, BK appeared with decreased vision to 20 cm/n. d. After confirmation of negative CMV-DNA in the aqueous humor, DSAEK was performed following cataract surgery. The postoperative visual acuity recovered to 0.3. Mooren’s ulcer exacerbation and CMV corneal endotheliitis did not recur postoperatively.
Conclusions and Importance
This is the first report of a case in which a patient with Mooren’s ulcer developed BK due to CMV corneal endotheliitis and required DSAEK. Cataract surgery and DSAEK could be performed without issue by creating the main wound and side ports in a manner that avoids the thinned parts of the cornea.
1
Introduction
Mooren’s ulcer is a progressive and painful idiopathic peripheral corneal ulcer. The treatment for Mooren’s ulcer includes steroids and immunosuppressive agents, and corneal transplantation is required in case of corneal perforation. , In contrast, adverse effects of long-term administration of steroids and immunosuppressive agents have also been reported, such as cytomegalovirus (CMV) corneal endotheliitis, , resulting in bullous keratopathy (BK) after anterior uveitis. Furthermore, it is difficult to perform corneal transplantation in patients with Mooren’s ulcer because the peripheral cornea of these patients is often very thinned and easy to perforate. Therefore, careful observation and ingenuity are required for the clinical decision of corneal transplantation for patients with Mooren’s ulcer. However, our research showed that no study has reported Descemet stripping automated endothelial keratoplasty (DSAEK) performed for patients with Mooren’s ulcer with BK due to CMV corneal endotheliitis. Herein, we report our experience of a case in which cataract surgery and DSAEK were successfully performed for BK owing to CMV corneal endotheliitis after immunosuppressive treatment for Mooren’s ulcer.
1.1
Case report
A 64-year-old man with a peripheral corneal ulcer in his left eye was referred to our hospital. He had bilateral primary open angle glaucoma and had undergone trabeculectomy of the left eye 5 years earlier. He visited a local ophthalmologist with the chief complaint of conjunctival hyperemia and pain and afterward, our hospital for consultation. At the initial presentation, hyperemia and cells inside the anterior chamber were observed in his left eye ( Fig. 1 a and b). A corneal ulcer with cell infiltration in the superior cornea was also found. The best-corrected visual acuity (BCVA) of his left eye was 0.4. As a result of systemic medical examination, including for collagen disease, he was diagnosed with Mooren’s ulcer, and treatment was initiated with topical 0.1% betamethasone 8 times daily, 0.1% tacrolimus 4 times daily, 0.3% gatifloxacin 4 times daily, and systemic cyclosporine 400 mg/day. The corneal ulcer resolved after 3 months, although thinning of the cornea was observed at 0 to 2 and 6 to 12 o’clock.
Two months later, he complained of reduced visual acuity in the left eye. Cells and massive broad keratic precipitates (KPs) in the anterior chamber were observed in his left eye with conjunctival hyperemia ( Fig. 1 c). The BCVA in the left eye was 0.3. Because real-time polymerase chain reaction (PCR) examination of the aqueous humor of the left eye revealed 1.5 × 10 7 copies/ml of CMV-DNA, he was diagnosed with CMV corneal endotheliitis. Therefore, topical tacrolimus and systemic cyclosporine were discontinued, and oral valganciclovir (900 mg/day) was administered for 6 weeks with topical 0.1% betamethasone eye drops 6 times daily. Three months later, real-time PCR revealed remaining 4.0 × 10 4 copies/mL of CMV-DNA in the aqueous humor, and oral valganciclovir was administered for another 6 weeks. Four months later, CMV-DNA was not detected by real-time PCR in the aqueous humor, and no relapse of the anterior uveitis was observed with use of betamethasone 6 times daily.
However, 15 months after the first presentation, corneal edema occurred in the left eye ( Fig. 2 a and b). Central corneal thickness was increased to 815 μm and visual acuity decreased to 20 cm/n. d. in the left eye ( Fig. 2 c). Because CMV-DNA was negative in the aqueous humor, the main reason of deteriorated visual acuity was considered to be BK with cataract. Therefore, cataract surgery was first performed through a 2.75-mm sclerocorneal incision at 4 o’clock with local anesthesia. Corneal epithelial removal, 0.1% trypan blue injection into the anterior chamber, and a hands-free chandelier endo-illumination system were used to help visualize the intraocular procedures. With a 27-gauge needle, two small holes were created at the 8 and 10 o’ clock positions 3.5 mm from the limbus, through which the chandelier fiber optic probes (BrightStar endoilluminator, DORC International, Zuidland, The Netherlands) were inserted. After phacoemulsification and aspiration, the intraocular lens was inserted in the capsule. After cataract surgery, the BCVA increased to 0.03, and no recurrence of CMV corneal endotheliitis or Mooren’s ulcer was observed ( Fig. 2 d).
