Interactive Case Reports

Candida albicans keratitis complicated with endophthalmitis after penetrating keratoplasty in Cogan’s syndrome

Case Report Details

First Author: B.Romero-Nunez SPAIN

Co Author(s):    C. Rocha-de-Lossada   N. Sabater-Cruz   J. Peraza-Nieves   J. Torras-Sanvicens           

Abstract Details

Purpose:

To report a case of Candida albicans endophthalmitis following keratitis after penetrating keratoplasty (PK) in a patient diagnosed of Cogan's syndrome (CS) interstitial keratitis (IK). Anterior and posterior segment examination, visual acuity, intraocular pressure (IOP) measurements and anterior segment optical coherence tomography (AS-OCT) and microbiological tests were performed and analyzed.

Setting:

Cogan’s syndrome is a rare chronic inflammatory disorder of un unknown origin characterized by ocular and audio-vestibular symptoms. Fungal keratitis and endophthalmitis are uncommon but devastating complications after penetrating keratoplasty. In the past 10 years, an increase in the incidence of exogenous fungal endophthalmitis has been noticed.

Report of Case:

A 30-year-old woman diagnosed of CS presented to our clinic with photophobia and blurred vision in both eyes for the past year. She was treated with 10mg/ml rimexolone eye drops and systemic methotrexate. The ophthalmological examination revealed a best corrected visual acuity (BCVA) of 20/25 in the right eye (RE), 20/20 in the left eye (LE) and an IOP of 16 and 18 mmHg in the RE and LE, respectively. Slit lamp examination showed corneal ghost vessels and lipidic stromal degeneration poorly preserving the corneal central area. All those features were compatible with inactive IK. During the follow up, a progressive visual acuity deterioration was noticed, BCVA sloped to 20/40 in the RE and 20/50 in the LE. Also, progression of the IK and development of cortical cataract in the left eye were found. A PK of the left cornea was performed. Oral and topical corticosteroids were prescribed and tapered gradually. The corneal graft maintained its transparency throughout the initial postoperative follow-up, although later, corneal graft melting and persistent cellularity in the anterior chamber led us the suspicion of a possible IK recurrence. So that, 40 mg subtenonian triamcinolone was prescribed. At the end of the first postoperative month, slit lamp examination revealed perikeratic hyperemia, keratic precipitates and round infiltrates suggesting infective keratitis. In order to assess anterior chamber status, an AS-OCT was performed. Endothelial graft hyperreflective irregularities and hyperreflective material in the anterior capsule of the crystalline lens were found out. Corneal microbial culture revealed the presence of Candida albicans in the corneal graft. The corneoscleral rim was found out to be contaminated by it as well. A new surgical procedure was performed and a PK, lensectomy and intravitreal amphotericin B and voriconazole were carried out due to medical treatment failure.

Conclusion/Take Home Message:

CS frequently manifests with IK and may need corneal transplantation. Although infective keratitis is a rare complication after PK and endophthalmitis is even more infrequent, patient’s immunosuppression and graft contamination may play an important role. In the context of fungal keratitis or endophthalmitis after PK further surgical intervention is usually required to achieve cure. Positive fungal donor corneoscleral rim cultures are uncommon but can carry devastating complications. Systematic culture of corneoscleral rim may provide diagnostic orientation and may have prognostic implications, as prophylactic treatment could be initiated. We consider that frequent follow up should be conducted specially in immunosuppressed patients as the case reported.

Financial Disclosure:

None