Delayed Onset and RecurrentAlcaligenes xylosoxidans Keratitis

Tim H. Pan, D.O., David G. Heidemann, M.D., Steven P. Dunn, M.D.,Christopher Y.C. Chow, M.D., and David Gossage, D.O.

Alcaligenes xylosoxidans (formerly Achromobacterxylosoxidans) is an opportunistic aerobic gram-negativerod that is oxidase positive and nonlactose fermenting. Itmay be confused with other gram-negative bacteria suchas Pseudomonas aeruginosa (1). The organism was firstdescribed and named in 1971 by Yabuuchi and Ohyama(2), who isolated the organism from purulent ear dis-charge of seven patients. It is frequently found in moistareas of hospitals and is resistant to many antimicrobialagents (3). Alcaligenes xylosoxidans is a transient colo-nizer of human gastrointestinal or respiratory tract inpatients with cystic fibrosis (4). Its mode of transmissionis often unknown, although it frequently involves expo-sure of debilitated patients to contaminated fluids ormedical solutions (1,5,6). It has caused bacteremia, uri-nary tract infections, meningitis, wound infections, pneu-monia, and peritonitis (6). To our knowledge, only fivecases of A. xylosoxidans keratitis have been reported(Table 1). We report a case of posttraumatic A. xylosoxi-dans keratitis that recurred after apparent resolution.

CASE REPORT

A 21-year-old tire technician was first seen on March25, 1998, with left-eye irritation after a tire inner tubeexploded. Visual acuity in the left eye was 20/100. Slit-lamp examination demonstrated a partial-thickness (90%depth) central corneal laceration. No infiltrate was pre-sent. The anterior chamber was quiet. No evidence ofintraocular foreign body was noted on gonioscopy, fun-duscopy, and computed tomography (CT) scan. The pa-tient was treated with ofloxacin drops every hour. Ex-

amination the next day revealed a mild anterior chamberreaction, and 1% prednisolone acetate drops and oralciprofloxacin were added. His vision improved to 20/40,and the anterior chamber reaction subsided. On April 3,1998, the patient had an intact epithelium, a mild anteriorchamber reaction, and a few fine keratic precipitates. Theprednisolone acetate was increased to every 2 h, andtobramycin (14 mg/ml) every 30 min was added to thetreatment regimen. Three days later, his condition wors-ened, and he was referred for corneal subspecialty evalu-ation.

Examination on April 6, 1998, revealed 20/200 vision,a 1.5-mm central epithelial defect, a 1.0-mm posteriorstromal infiltrate, diffuse corneal edema, and a 0.5-mmhypopyon (Fig. 1). B-scan ultrasonography revealed anormal posterior segment. The site of laceration was re-opened with a no. 11 blade to obtain corneal scrapingsfrom the posterior stromal infiltrate for cultures on thio-glycolate broth, blood, chocolate, and Sabouraud agar.The patient’s treatment regimen was modified to topicaltobramycin (14 mg/ml), vancomycin (50 mg/ml), andamphotericin B (1.5 mg/ml) every hour around the clock,and oral ciprofloxacin. The topical steroids were discon-tinued. On April 9, 1998, his vision worsened to countfingers. Cultures revealed moderate growth of A. xylos-oxidans. The patient was treated with hourly ciprofloxa-cin (3 mg/ml), 10% sodium sulfacetamide, and tobramy-cin (14 mg/ml) drops. Sensitivities eventually revealedthat the organism was resistant to tobramycin, interme-diately sensitive to ciprofloxacin, and sensitive to sulfa-methoxazole and piperacillin. On April 17, 1998, thepatient’s vision was 20/50, with an intact epithelium, anda quiet anterior chamber. On April 24, 1998, the visionwas 20/30, with complete resolution of the corneal infil-trate, and a 1.0-mm midstromal scar located in the visualaxis. Over the next 2-week period, the topical medica-tions were slowly tapered to twice-a-day dosage by May7. On May 11, 1998, the eye remained quiet, with a smallresidual central corneal stromal scar, and the antibioticdrops were discontinued.

Submitted March 12, 1999. Revision received May 24, 1999. Ac-cepted June 1, 1999.

From the Department of Ophthalmology, William Beaumont Hos-pital, Royal Oak (D.G.H., S.P.D., C.Y.C.C.), and Pontiac OsteopathicHospital, Pontiac (T.H.P., D.G.), Michigan, U.S.A.

Address correspondence and reprint requests to Dr. D.G.Heidemann, Michigan Cornea Consultants, Suite 201, FarmbrookMedical Building, 29829 Telegraph, Southfield, MI 48034, U.S.A.

Cornea 19(2): 243–245, 2000. © 2000 Lippincott Williams & Wilkins, Inc., Philadelphia

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On May 13, 1998, the patient had an acute worseningof symptoms. He had vision of count fingers, a 2.0-mmepithelial defect, a 1.5-mm stromal infiltrate with sur-rounding stromal edema, and a 0.5-mm hypopyon. Re-peated corneal cultures were taken. The patient wastreated with tobramycin (14 mg/ml), vancomycin (33mg/ml), and 10% sodium sulfacetamide drops. Over thenext 2-week period, the keratitis resolved. Cultures againyielded a moderate growth of A. xylosoxidans on bloodand chocolate agar, with the same sensitivity pattern asthe previous cultures. On June 16, 1998, the patient’svision was 20/30, and there was no evidence of infection.Treatment with tobramycin and sodium sulfacetamidedrops was maintained for 2 months. Six months later, hiseye remained quiet, with 20/40 visual acuity.

DISCUSSION

The first reported ocular infection by A. xylosoxidanswas in 1977, when it was isolated from the infected orbitof a patient who had lost the globe after a perforatinginjury from shrapnel (3). It was implicated in a case ofchronic bacterial endophthalmitis in a 63-year-oldwomen after cataract surgery (4). To our knowledge,there have been five reported cases of A. xylosoxidans

corneal infection (Table 1). Our case was similar in someaspects to these reported cases. All patients had a com-promised cornea and did not respond to conventionalantimicrobial therapy. Our patient sustained a partial-thickness corneal laceration from a tire explosion andwas treated with topical steroids and antibiotics. It isunclear if the previous reported cases of A. xylosoxidanskeratitis were late onset. Our patient did not develop aninfiltrate until 10 days after injury.

As in the previously reported cases, our patient did notrespond to conventional broad-spectrum antibiotics. Hewas taking ofloxacin and fortified tobramycin dropswhen the ulcer initially developed. The previously re-ported ineffective regimens included topical gentamicin,amikacin, cefazolin, and tobramycin. Alcaligenes xylos-oxidans is usually resistant to aminoglycosides and first-generation cephalosporins. In the previously reportedcases, resolution of the keratitis was achieved only afterthe initiation of carbenicillin, ticarcillin, piperacillin,and/or azlocillin drops. The �-carboxypenicillins (e.g.,carbenicillin, ticarcillin) and the acylaminopenicillins(e.g., azlocillin, mezlocillin) are effective both in vitroand in vivo against A. xylosoxidans (9,12). Our patientresponded well to topical ciprofloxacin and sodium sul-facetamide drops after the discontinuation of topical cor-ticosteroids. Alcaligenes xylosoxidans has many similari-ties to Pseudomonas. As with Pseudomonas keratitis,steroids may have been a risk factor in exacerbation of A.xylosoxidans keratitis. In our patient’s treatment regi-men, only sulfamethaxozole, a bacteriostatic agent, re-ceived a “sensitive” rating. The organism was only in-termediately sensitive to ciprofloxacin and was resistantto aminoglycosides. It appears that the discontinuation ofsteroids and addition of sodium sulfacetamide was suf-ficient to cure the infection.

Our patient differs from the previous reported cases inthat the keratitis recurred after an apparent resolution.Viable organisms were most likely still present at theoriginal ulcer site. In each instance, corneal culturesyielded a moderate growth of A. xylosoxidans on bloodand chocolate agar. The infection resolved permanentlyafter prolonged treatment with topical sodium sulfaceta-mide. In retrospect, treatment with carbenicillin or piper-

TABLE 1. Reported cases of Alcaligenes xylosoxidans keratitis

Author, date ImmunocompromiseSteroid

use

“Conventional”treatment (topical)

failedTreatment required

(topical)

Majekodunmi, 1975 Concurrent Clostridium welchii infection ? ? ?Boisjoly et al., 1973 Chronic herpes simplex keratitis Yes ? ?Newman et al., 1984 Neovascular glaucoma Yes Gentamicin, amikacin CarbenicillinFiscella et al., 1989 Bullous keratopathy with bandage

contact lensNo Cefazolin, gentamicin Carbenicillin

Siganos et al., 1993 Penetrating keratoplasty Yes Tobramycin, cefazolin (and i.v.trimethoprim/sulfa, gentamicin)

Piperacillin (andi.v. azlocillin)

Current case, 1999 Corneal laceration Yes Ofloxacin (and oral ciprofloxacin) Sulfacetamide

FIG. 1. Posterior stromal corneal infiltrate at lacerationsite, with diffuse stromal edema and small hypopyon.

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acillin may have been more effective at eradicating theinfection.

In conclusion, A. xylosoxidans should be considered apotential pathogen in compromised or traumatized cor-neas. The infection may be late onset and exacerbated bysteroids. It does not respond to conventional broad-spectrum antimicrobial therapy and may be difficult toeradicate.

REFERENCES

1. Forbes, Weissfeld. Bailey and Scott’s diagnostic microbiology. St.Louis: Mosby, 1998:476–85.

2. Yabuuchi E, Ohyama A. Achromobacter xylosoxidans from humanear discharge. Jpn J Microbiol 1971;15:477–81.

3. Holmes B, Snell JJ, Lapage SP. Strains of Achromobacter xylos-oxidans from clinical material. J Clin Pathol 1977;30:595–601.

4. Ficker L, Meridith TA, Wilson LA, Kaplan JK, Kozarsky AM.Chronic bacterial endophthalmitis. Am J Ophthalmol 1987;103:745–8.

5. McGuckin MB, Thorpe RJ, Koch KM, Alavi A, Staum M, AbrutynE. An outbreak of Achromobacter xylosoxidans related to diagnos-tic tracer procedures. Am J Epidemiol Infect Dis 1982;1:242–56.

6. Reverdy ME, Freney J, Fleurette J, et al. Nosocomial colonizationand infection by Achromobacter xylosoxidans. J Clin Microbiol1984;19:140–3.

7. Majekodunmi S, Odugbemi T. Clostridium welchii corneal ulcer: acase report. Can J Ophthalmol 1975;10:290–2.

8. Boisjoly HM, Paven-Langston D, Kenyon KR, Sullivan Baker A.Superinfections in herpes simplex keratitis. Am J Ophthalmol1983;96:354–61.

9. Newman PE, Hider P, Waring GO III, Wilson LA, Harbin TS.Corneal ulcer due to Achromobacter xylosoxidans. Br J Ophthal-mol 1984;68:472–4.

10. Fiscella R, Noth J. Achromobacter xylosoxidans corneal ulcer in atherapeutic soft contact lens wearer. Cornea 1989;8:267–9.

11. Siganos DS, Tselentis IG, Papatzanaki ME, Tsilimbaris MK, Pal-likaris IG. Achromobacter xylosoxidans keratitis following pen-etrating keratoplasty. Refract Corneal Surg 1993;9:71–3.

12. Igra-Siegman Y, Chmel H, Cobbs C. Clinical and laboratory char-acteristics of Achromobacter xylosoxidans infection. J Clin Micro-biol 1980;11:141–5.

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