CASE REPORT

Nocardia exalbida Blebitis: A Case Report Cristos Ifantides, MD, MBA,* Oscar R. Batlle, MD,* David Mushatt, MD,w and Ramesh S. Ayyala, MD, FRCS, FRCOphth*

Purpose: To report the first documented case of Nocardia exalbida blebitis. Methods: A 57-year-old immunocompetent African American man with a long-standing history of open-angle glaucoma in both eyes treated with trabeculectomy presented with a diffusely hyperemic, thin, cystic, leaky bleb with no discharge in his left eye. The patient underwent bleb revision using an amniotic membrane patch graft followed by 1 month of antibiotics. He presented second time with an inflamed eye and brisk leakage and underwent a second bleb revision. His cultures remained negative. Two months after this second surgery, an anterior staphyloma had formed within the bleb area, and visible leakage of purulent material and a dense hypopyon was noted. Gram stain of the material showed rare longbranching rods. The material was sent to an outside laboratory for culture and identification. Results: All 6 cultures were positive for N. abscessus complex and N. exalbida. The patient underwent 6 months of Bactrim therapy with topical sulfonamide and amikacin, leading to the disappearance of the hypopyon and an inflammation-free eye. Conclusions: N. exalbida is a newly identified Nocardia species that must be considered as a possible infectious agent in immunocompetent patients with blebitis refractive to initial topical antibiotic therapy. Delay in diagnosis and initiation of appropriate antibiotic regimen can result in an aggressive inflammatory process and vision loss. Key Words: Nocardia infection, glaucoma valve implant, blebitis, Nocardia exalbida, Nocardia abscessus

(J Glaucoma 2015;24:e19–e21)

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n recent years, the common use of antiproliferative agents such as mitomycin C and 5-fluorouracil in trabeculectomy has led to increased reports of bleb-related infections because of the generation of thin avascular filtering blebs.1 A retrospective chart review of 421 surgeries by Sharan et al2 reported a 0.96% bleb infection rate with a mean follow-up of 5.3 years. The average time until infection was 31.3 months after surgery. Risk factors for infection included black race, bleb leak, bleb manipulation, and inferior bleb placement. Blebitis can be either early onset (1 mo). Early onset blebitis has a similar bacteria profile Received for publication December 2, 2012; accepted April 14, 2013. From the *Department of Ophthalmology; and wInfectious Diseases Section, Tulane School of Medicine, Tulane University, New Orleans, LA. Supported in part by the Tulane Glaucoma Research Fund. Disclosure: The authors declare no conflict of interest. Reprints: Ramesh S. Ayyala, MD, FRCS, FRCOphth, Glaucoma Service, Department of Ophthalmology, Tulane University Medical Center, 1430 Tulane Avenue SL-69, New Orleans, LA 70112 (e-mail: [email protected]). Copyright r 2013 Wolters Kluwer Health, Inc. All rights reserved. DOI: 10.1097/IJG.0b013e3182a07574

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to that seen in acute endophthalmitis after cataract surgery, with Staphylococcus epidermidis being the most common causative agent. Conversely, late onset blebitis is mostly dominated by Streptococcus pneumoniae and gram-negative bacteria such as Haemophilus influenzae.3,4 Nocardia are aerobic, gram-positive, partially acidfast, nonmotile bacteria belonging to the class actinomycetaceae that form extensive branched filaments that fragment into rod-shaped elements.5 These organisms have been shown to be responsible for cutaneous, ocular, pulmonary, and disseminated disease, with many different species of Nocardia involved. Although immunocompetent and immunocompromised patients are at risk of Nocardia infection, immunocompromised patients are at a higher risk of infection and disease.6 N. exalbida is a particular strain of Nocardia discovered in 2006. Only 2 other cases of a N. exalbida ocular infection, that is, keratitis7 and endogenous endophthalmitis,8 have been reported in the literature. We report the first ever case of N. exalbida blebitis.

CASE PRESENTATION A 57-year-old immunocompetent African American man presented with complaints of redness, tearing, and decreased vision in his left eye for 3 days. Past ocular history was significant for long-standing open-angle glaucoma in both eyes treated with trabeculectomy. The patient worked as a streetcar repairman and grass cutter and reported frequent exposure to dirt and dust. He denied any other complaints and was in otherwise good health. Before this presentation, the patient had uncomplicated trabeculectomy with mitomycin C (0.4 mg/mL 45 s) in his left eye and right eye, in 2000 and 2004, respectively. Since then, he was followed routinely, with good control of his intraocular pressures (IOPs) while on a timolol 0.5% and dorozolamide 1% combination OU bid. His follow-up visits remained uneventful, with a baseline best corrected visual acuity of 20/25 in the right eye and 20/40 in the left eye ( 0.75 + 0.5090 OD and 100 + 0.750 90 OS). He had repeated Humphrey visual field analysis showing dense arcuate scotomas bilaterally with loss of central fixation only in the left eye. The patient had thin corneas, with pachymetry measurements of 487 and 500 mm in the right and left cornea, respectively. He had advanced cupping bilaterally with a cup-to-disc ratio of 0.99 OU. This was further confirmed by a thin RNFL on optical coherence tomography, which measured 39.5 mm in the right eye and 25.04 mm in the left eye. He presented in 2011 with redness and decreased vision in his left eye. On presentation, the patient was found to have a hypotonous left eye with IOP of 3 mm Hg and a BCVA of 20/100. Slit-lamp examination revealed a diffusely hyperemic, thin, cystic, and translucent bleb with no discharge. Fluorescein exam showed the bleb to be Seidel positive, with positive staining of the bleb roof indicating a localized area of conjunctival epithelial erosion. He had deep anterior chambers with 1 + cells and flare but no evidence of vitritis or posterior segment involvement. The patient was immediately started on fortified vancomycin (25 mg/mL q1h), gatifloxacin (q1h), and tobramycin and dexamethasone ointment (qhs). After a 1-week course of antibiotics, the conjunctival injection resolved but bleb leakage persisted. The patient underwent bleb revision using an amniotic membrane graft and conjunctival advancement and was continued on the fortified antibiotics. The

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FIGURE 3. Visible leakage of the purulent material from the superior limbal area with hypopyon demonstrating persistent infection. Samples taken from the cornea and scleral junction at the site of the sclerotomy grew Nocardia.

FIGURE 1. Recurrent blebitis with hypopyon with an inflamed eye, conjunctival retraction and necrosis, and an exposed scleral flap. patient initially responded well, with an IOP of 10 mm Hg, a deep and quiet anterior chamber, and a Seidel-negative bleb. Antibiotics were slowly discontinued over a period of 1 month. However, 3 weeks after discontinuing the antibiotics, he presented with an inflamed eye, conjunctival retraction and necrosis, an exposed scleral flap, brisk leakage, and hypotony with an IOP of

Nocardia exalbida blebitis: a case report.

To report the first documented case of Nocardia exalbida blebitis...
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