Peripheral Ulcerative Keratitis With Concurrent Herpetic Zoster And Burkholderia Cenocepacia Co-Infection

The UK Health Security Agency reported a cluster of Burkholderia cenocepacia (B. cenocepacia) cases isolated on eye swabs and thought to be related to use of contaminated carbomer gel.  Peripheral ulcerative keratitis (PUK) is a rare inflammatory condition of the peripheral cornea. Rheumatoid arthritis constitutes the majority of autoimmune systemic PUK cases but infections, particularly herpetic disease make up another important localised cause. B. cenocepacia is an opportunistic Gram negative bacillus that has rarely been reported as a cause of keratitis and endophthalmitis, particularly in patients with concurrent herpetic keratitis. We report a single case of PUK with Herpes zoster and B. cenocepacia with no prior ocular lubricant use.

We describe a single case report in the Norfolk and Norwich University Hospital, a tertiary centre corneal service based in the United Kingdom.

A 83-year-old pseudophakic gentleman presented to the eye emergency clinic with a 3-week history of a painful red left eye. He presented with best corrected visual acuity of HM and intraocular pressure of 7 mmHg. His right eye was unremarkable.

His left eye was markedly injected with inferior crescenteric perilimbal thinning consistent with peripheral ulcerative keratitis (PUK) with 90% stromal loss, pigmented mutton fat keratic precipitates, grade 3+ anterior chamber cells, no flare but with a fine hypopyon. Six months prior he had a left facial rash attributed to atopic dermatitis and managed with emollients only. He denied previous topical lubricant use, and had a past medical history of diabetes, angina and previous radiotherapy for bladder cancer. B-scan was performed due to limited fundal view and at this point carbomer gel may have been used as a coupling agent.

Laboratory investigations raised white cell count, neutrophilia, and raised C-reactive peptide.  Conjunctival swabs of the left eye returned positive for varicella zoster virus (VZV), negative for herpes simplex virus (HSV) and bacterial culture returned B. cenocepacia, and serology was negative for syphilis and hepatitis C.

He was commenced on preservative free topical levofloxacin, lubricants, dexamethasone and oral valaciclovir and prednisolone. At 3 months, his vision improved to 6/12 with a quiescent, stable cornea.

This case represents a case of zoster sine herpete with potential concurrent infection with B. cenocepacia leading to first presentation of PUK. It remains possible that the carbomer coupling agent may have been the source of B. cenocepacia, it nonetheless reinforces the importance of considering infectious differentials including microbial co-infection the identification and management of PUK.