CASE REPORTS

Rhodotorula glutinis endophthalmitis

Michael W. Dorey,*t MD; Seymour Brownstein,*t MD; Peter J. Kertes,* MD; Steven M. Gilberg,* MD; Baldwin Toye,:j: MD

F ungi would be uncommon ocular pathogens if it were not for the many medical advances of the

20th century, particularly the advent of immunosup­pressive agents. 1 Although rare in immunocompetent people, endogenous fungal endophthalmitis is increas­ingly common in patients who are immunocompro­mised, are intravenous drug users or have an indwelling venous catheter. 1 A greater diversity of organisms seem to be involved in endogenous fungal endoph­thalmitis than was previously thought. We present a case that, to our knowledge, is the first report of Rhodotorula glutinis endophthalmitis and, as such, rep­resents a unique form of fungal endophthalmitis in an immunocompromised patient without evidence of sys­temic illness.

CASE REPORT

A 26-year-old man presented with a 2-month histo­ry of floaters and deteriorating vision in his right eye associated with pain, redness and photophobia. His past medical history included intravenous drug use, hepatitis C and HIV seropositivity for 10 years, with no antiretroviral therapy during the preceding year and no previous diagnosis of AIDS. He had been a fre­quent cocaine and heroine user for several years but

From *the Department of Ophthalmology, University of Ottawa

Eye Institute, Ottawa Hospital, Ottawa, Ont., and the Departments

of tPathology and :j:Microbiology, University of Ottawa, Ottawa

Hospital, Ottawa, Ont.

Originally received Mar. 27, 2002 Accepted for publication Aug. 20, 2002

Correspondence to: Dr. Seymour Brownstein, Room 3818, University

of Ottawa Eye Institute, 501 Smyth Rd., Ottawa ON K1H 8L6; fax (613) 737-8826; sbrownstein@ohri.ca

This article has been peer-reviewed.

Can J Ophthalmol 2002;37:416-8

had stopped using these drugs 3 weeks earlier and was receiving methadone withdrawal therapy.

On examination, his best corrected visual acuity was hand motion in the right eye and 20/20 in the left. Slit­lamp examination showed the cornea to be normal, with 4+ cells in the anterior chamber and 2+ vitritis. Fundoscopic examination with pupil dilation disclosed multiple creamy white lesions in the vitreous and reti­na, most consistent with fungal endophthalmitis.

Apart from the patient's being somewhat thin and fragile, systemic examination was unremarkable. Cul­ture of blood and urine specimens failed to yield micro­organisms. The CD4 count was 224 cells/IJL, and the viral load was 66 531 copies/mL.

Antiretroviral therapy (stavudine [d4T], lamivu­dine [3TC] and efavirenz) was started immediately. A pars plana vitreous biopsy procedure was per­formed the same day. The patient was then treated empirically with amphotericin B (1 mg/kg per day administered intravenously) and vancomycin (1 mg), tobramycin (400 !Jg) and amphotericin B (5 J.Jg), all administered intravitreally. His vision did not im­prove. After 7 days of incubation, the vitreous biop­sy specimen grew R. glutinis that was sensitive to itraconazole and amphotericin but not fluconazole. Unfortunately, the patient left the hospital 5 days after admission, against medical advice, before the results of vitreous culture were available. He did not receive ongoing care, including vitrectomy or further systemic antifungal therapy.

Two months after initial presentation the patient sought care because of worsening vision in his right eye. B-scan ultrasonography showed a retinal detach­ment. A pars plana lensectomy and vitrectomy proce­dure was performed with opening of a dense trans­vitreal sheet, which revealed a markedly necrotic appearing retina that could not be reattached. Given the patient's immunocompromised status and the presence of viable fungi in the eye, which had no light perception vision, the right globe was enucleated.

Pathological examination of the enucleated globe

416 Rhodotorula glutinis endophthalmitis-Dorey et al

Fig. !-Horizontal anteroposterior section of right globe, showing white vitreous granulomas (G) just posterior to iris, with dense exudate filling most of vitreous cavity. Necrotic, totally detached retina is avulsed from optic nerve.

showed zonal granulomatous inflammatory infiltrates involving primarily the anterior vitreous (Fig. 1 and Fig. 2, top). The retina, which was totally detached, was avulsed from the optic papilla, and only fragments of degenerated retina were noted along the posterior aspect of the inflamed anterior vitreous. Budding yeast were identified predominantly in the vitreous granulo­mas (Fig. 2, bottom). These fungal elements were con­sistent with Rhodotorula species.

The patient received an orbital prosthesis after enu­cleation and did well. There was no evidence of sys­temic involvement with R. glutinis during 9 months of observation following the procedure.

COMMENTS

In recent years fungal infections have become an important cause of infectious disease. 1- 3 Fungi ac­count for more than one-half of cases of endogenous endophthalmitis. 4 Risk factors include intravenous drug use, hyperalimentation, recent surgery or injury, indwelling bladder catheters, malignant disorders and immunosuppression.4 Most cases occur in the absence of systemic infection.4·5

Endogenous fungal endophthalmitis develops slow­ly as areas of chorioretinitis. Granulomatous or non-

Rhodotorula glutinis endophthalmitis-Dorey et al

Fig. 2-Photomicrographs of right globe. Top: Granuloma (G) within organized vitreous adjacent to iris and ciliary body and remnants of lens capsule (L). Numerous fungi (arrows) are pres­ent paracentrally in necrotic centre of granuloma (periodic acid-Schiff; original magnification X 60). Bottom: Budding yeast within necrotic centre of same granuloma (methenamine silver; original magnification X 2000).

granulomatous inflammation is typically observed, with the presence of keratic precipitates, hypopyon and vitritis. Chorioretinal lesions tend to appear as "snowball" lesions ranging in size from small cotton­wool spots to several disc diameters.5

Endophthalmitis may be caused by many different fungi, including Candida, Aspergillus, Histoplasma, Coccidioides, Cryptococcus, Blastomyces, Turolopsis, Mucor, Fusarium and Sporotrichum. 1•4•5 Although ex­ceedingly rare, Rhodotorula species have been reported as a source of ocular infection in chronic dacryocysti­tis,6 keratitis7 and corneal lamellar graft infections.8

One case of R. minuta endophthalmitis in a patient who had undergone surgery9 and another case in an injecting drug userS have also been reported. We are not aware of any reported cases of R. glutinis endophthalmitis.

Rhodotorula species are yeast-like fungi from the Cryptococcaceae family. They are ubiquitous air­borne organisms that may be found in food, the air,

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Rhodotorula glutinis endophthalmitis-Dorey et al

soil, water and the gastrointestinal tract and on the skin.5·6·10 When isolated from healthy people, they are often considered to represent contamination or harmless colonization and usually do not have any pathological significance.10·11 Macroscopically they are asporogenic, nonfermenting yeast cells with carot­enoid pigment.6•12 Microscopically they are round, oval or elongate cells that reproduce by budding.6

Although several species have been described, R. rubra and R. glutinis are the most common.l 1·13 .14

The role of Rhodotorula species in systemic infec­tion is becoming better understood. Recently they have emerged as serious pathogens when there is disruption or deficiency of host defence mecha­nisms.10·15 Risk factors include granulocytopenia, damage to the skin or the mucosa, cellular immune dysfunction and the presence of indwelling cathe­ters. 10 At risk are immunocompromised patients with neoplastic disease, AIDS, chronic renal failure or diabetes mellitus. 10 A range of infections, from uncomplicated transient fungemia to potentially life­threatening invasive infections such as endocarditis, meningitis, ventriculitis and peritonitis, have recent­ly been described. 10 The most common scenario is septicemia in immunocompromised patients receiv­ing intravenous therapy. 13 However, a case of Rhodotorula-related meningitis has been reported in an immunocompetent patient. 11

Rhodotorula species are rarely encountered in clini­cal specimens and may be dismissed as culture con­taminants.11 However, they may be associated with substantial infection in immunocompromised pa­tients.2·3·10·13·14 Ophthalmologists should notify the microbiology laboratory of such clinical possibilities so that appropriate media and lengthy incubation are used, which will allow uncommon, slowly replicating organisms, in particular fungi, to grow.5·9

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11. Lanzafame M, De Checchi G, Parinello A, Trevenzoli M, Cattelan AM. Rhodotorula glutinis-related meningitis. J Clin Microbiol2001;39:410.

12. Perrier V, Dubreucq E, Galzy P. Fatty acid and carotenoid composition of Rhodotorula strains. Arch Microbiol1995; 164:173-9.

13. Pien FD, Thompson RL, Deye D, Roberts GD. Rhoda­torula septicemia: two cases and a review of the literature. Mayo Clin Proc 1980;55:258-60.

14. Colombo AL, Dantas LS, Abramczyk ML, Cypriano M, Fischman 0, Iazzetti A V, et al. Rhodotorula glutinis fungemia: a case report and literature review. Braz J Irifect Dis 1997;1:204-7.

15. Hazen KC. New and emerging yeast pathogens. Clin Microbial Rev 1995;8:462-78.

Key words: Rhodotorula glutinis, fungal endophthalmitis, immunocompromised, human immunodeficiency virus (HIV), Cryptococcaceae