A rare case of infectious crystalline keratopathy caused by Burkholderia cepacia

Abstract

We report a unique case of infectious crystalline keratopathy (ICK) caused by Burkholderia cepacia in a 47-year-old patient with history of multiple penetrating keratoplasties (PKPs). Slit-lamp examination revealed a white nonsuppurative needle-like branching anterior stromal infiltrate. Microscopic examination of the Gram-stained smear showed gram-negative bacilli. Cultures revealed Burkholderia cepacia, which was multidrug resistant and sensitive to piperacillin/tazobactam. Based on antibiotic sensitivity report, patient was started fortified piperacillin-tazobactam (0.32%) eye drop on an hourly basis. After 8 weeks of aggressive therapy, the infiltrate resolved with subsequent corneal scarring and vascularization. To our knowledge, this is the first reported case of Burkholderia-related ICK. ICK, first described by Gorovoy et al.[1] in 1983, is an indolent keratitis with characteristic needle-like branching opacities in the absence of corneal or anterior segment inflammation.[2] The presence of an immunocompromised corneal state, such as chronic topical steroid use post PKP, is a known risk factor for the development of ICK. Other risk factors include surgical procedures such as lamellar keratoplasty, corneal cross-linking, laser-assisted in situ keratomileusis, pterygium excision, and trabeculectomy with 5- FU. Topical anaesthetic abuse as a risk factor has also been described.[3] Burkholderia is a group of Gram-negative bacteria, composed of at least 20 different species. We describe a case of Burkholderia keratitis presenting as ICK in a patient who underwent multiple PKPs and a history of long-term topical steroid therapy.