CLINICOPATHOLOGIC REPORT

SECTION EDITOR: W. RICHARD GREEN, MD szulgai Keratitis

Beatrice E. Frueh, MD; Olivier Dubuis, MD; Pascal Imesch, MD; Matthias Bo¨hnke, MD; Thomas Bodmer, MD

ontuberculous mycobacteria rarely cause keratitis. Most reported cases have been described in compromised corneas (ie, after penetrating keratoplasty, refractive sur- gery, and contact lens wear). The most frequently recovered nontuberculous myco- are the rapid growers, and Mycobacterium chelo- Nnae.1,2 Occasional cases of keratitis caused by the slower-growing Mycobacterium gordonae have also been described.3 To our knowledge, this is the first reported case of Mycobacterium szulgai keratitis.

REPORT OF A CASE tanoprost (Xalatan), and acyclovir oint- ment (Zovirax). Systemic valacyclovir hy- A 40-year-old patient developed a non- drochloride (Valtrex) and acetazolamide healing peripheral corneal infiltrate in (Diamox) were also given. his right eye without a history of Because of worsening of the peri- trauma. An initial diagnosis of herpetic pheral ulcer (Figure 1), conjunctival keratouveitis was made, and he was and corneal biopsies were performed 1 treated with topical and systemic antivi- month later. The result of the Gram ral medication, topical corticosteroids, stain was negative, and bacterial cul- and ciprofloxacin. The result of a poly- tures remained sterile. However, the merase chain reaction for herpes sim- auramine O fluorescent and Ziehl- plex was negative. Neelsen stains revealed a few acid-fast Five months later, the patient was bacilli (Figure 2). The histological fea- referred to our service. His visual acuity tures showed a granuloma with early was 20/50 OD and 20/20 OS. A nasal central necrosis (Figure 3). Myco- stromal corneal infiltrate with epithelial bacteria were recovered from cultures ulceration and vascular pannus at its (BACTEC 12B; Becton Dickinson, edge was seen in the right eye. Micro- Sparks, Mass) after 14 days of incuba- cystic epithelial edema and an intra- tion at 37°C. These were subsequently ocular pressure of 37 mm Hg were identified as M szulgai by polymerase noted. Grade 1 anterior chamber in- chain reaction–restriction fragment flammation was recorded. A diagnosis length polymorphism analysis4 and by of corticosteroid-induced glaucoma and conventional methods.5 Minimum possible herpetic keratouveitis was inhibitory concentrations were deter- made, and corneal scrapings were per- mined using a modification of the formed. microbroth dilution method described Coagulase-negative staphylococcus by Yajko et al.6 The in vitro susceptibil- grew in the cultures. The topical treat- ity of M szulgai is as follows: ment included ciprofloxacin hydrochlo- Minimum ride (Ciloxan), scopolamine, hydrocorti- Inhibitory sone with oxytetracycline hydrochloride Drug Concentration, mg/L and polymyxin B sulfate (Terra-Cortril), Amikacin 2.000 dorzolamide hydrochloride (Trusopt), la- Clarithromycin 0.250 Ciprofloxacin 0.250 Rifabutin 0.500 From the Departments of Ophthalmology, University of Berne (Drs Frueh, Imesch, and Ethambutol hydrochloride 2.000 Bo¨hnke), and the Institute for Medical Microbiology, University Hospital (Drs Dubuis and Bodmer), Berne, Switzerland. None of the authors has a financial interest in the Topical corticosteroids were discon- products described in this article. tinued, and the treatment was changed to

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©2000 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/27/2021 Figure 1. Slitlamp picture of the left cornea showing a peripheral corneal Figure 2. High-power illustration of acid-fast bacilli (Ziehl-Neelsen, original ulcer and a heavily vascularized nodule. magnification ϫ1000).

szulgai. The keratitis healed with Accepted for publication February 11, topical and systemic treatment, 2000. and a final visual acuity of 20/25 Reprints: Beatrice E. Frueh, MD, OD was attained. The patient was Department of Ophthalmology, Insel- immunocompetent and did not spital, 3010 Bern, Switzerland (e- wear contact lenses, but had been mail: [email protected]). treated with topical corticosteroids for several months before he was REFERENCES first seen by us. Mycobacterium Figure 3. High-power photomicrograph of the szulgai is an unusual pathogen that 1. Ford JG, Huang JW, Pflugfelder SC, Alfonso EC, excised conjunctiva shows chronic accounts for less than 1% of all Forster RK, Miller D. Nontuberculous mycobac- granulomatous inflammation cases of nontuberculous mycobac- terial keratitis in south Florida. Ophthalmology. (hematoxylin-eosin, original magnification 1998;105:1652-1658. ϫ200). teria infections. Diseases caused by 2. Bullington RH Jr, Lanier JD, Font RL. Nontuber- this slow-growing mycobacterium culous mycobacterial keratitis: report of two cases ciprofloxacin hydrochloride, 5 times usually involve the lung but may and review of the literature. Arch Ophthalmol. daily, and amikacin, 20 mg/mL, also lead to infections of soft tissue 1992;110:519-524. 4 times daily. Clarithromycin and bone.8 3. Sossi N, Feldman RM, Feldman ST, Frueh BE, McGuire G, Davis C. Mycobacterium gordonae drops, 20 mg/mL, were prepared In our case, in vitro suscepti- keratitis after penetrating keratoplasty. Arch Oph- according to the method of Helm bility testing showed rather low thalmol. 1991;109:1064-1065. et al,7 but were not tolerated and minimum inhibitory concentra- 4. Telenti A, Marchesi F, Balz M, Bally F, Bottger EC, had to be discontinued after a few tions to clarithromycin and cipro- Bodmer T. Rapid identification of mycobacteria to the species level by polymerase chain reaction and days. Clarithromycin (Klacid), floxacin. Because topical clarithro- restriction enzyme analysis. J Clin Microbiol. 1993; 500 mg twice daily, was given mycin was not well tolerated, 31:175-178. orally for 3 months. Clinically, the amikacin was also used. The in vitro 5. Kent PT, Kubica GP. Public Health Mycobac- inflammation subsided during the susceptibility of mycobacteria to an- teriology: A Guide for the Level II Laboratory. following 3 weeks, the epithelial tibiotics is different from that in vivo Atlanta, Ga: US Dept of Health and Human Services, Public Health Service; 1985. erosion healed, and a vascularized and may not predict the effective- 6. Yajko DM, Nassos PS, Hadley WK. Broth micro- scar formed. Eleven months after ness of topical antibiotics. This may dilution testing for susceptibilities to 30 antimi- the biopsy and 3 months after dis- explain why the keratitis healed de- crobial agents of Mycobacterium avium strains continuation of topical treatment, spite the fact that we did not use the from patients with acquired immune deficiency the best-corrected visual acuity is optimal drug combination as indi- syndrome. Antimicrob Agents Chemother. 1987; 31:1579-1584. 20/25 OD and the eye is not cated by the minimum inhibitory 7. Helm CJ, Holland GN, Lin R, Berlin OGW, Bruck- inflamed. concentrations. ner DA. Comparison of topical antibiotics for treat- This case underscores the im- ing Mycobacterium fortuitum keratitis in an animal portance of performing corneal bi- model. Am J Ophthalmol. 1993;116:700-707. COMMENT 8. Benator DA, Kann V, Gordin FM. Mycobacterium opsies to detect unusual pathogens szulgai infection of the lung: case report and re- To our knowledge, this is the first in patients with therapy-resistant view of an unusual pathogen. Am J Med Sci. 1997; reported case of keratitis due to M keratitis. 313:346-351.

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