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Recurrent endogenous candidal endophthalmitis in a premature infant
Recurrent Endogenous Candidal Endophthalmitis in a Premature Infant Jeffrey H. Stern, MD, PhD, Christopher Calvano, MD, PhD, and John W. Simon, MD
Endogenous Candida endophthalmitis resulting from candidemia in low-birth-weight infants usually occurs as a retinochoroiditis, which is effectively treated with systemic antifungal agents. We report a case of Candida endophthalmitis that recurred 4 months after completion of systemic antifungal therapy. The recurrent Candida infection affected primarily the iris and lens, rather than the retina and choroid. Vitrectomy was required for diagnosis and treatment.
CASE REPORT An 11-day-old girl was referred because of Candida albicans fungemia from a cutaneous source. The infant was premature (24 gestational weeks), with a birth weight of 800 g. Initial examination showed a perimacular white chorioretinal lesion and a small white iris nodule adjacent to the pupillary margin in the left eye. There was no evidence of lens involvement, and the right eye did not show signs of infection. The infant was treated with systemic amphotericin B and 5-fluorocytosine (5-FC) to maintain adequate serum levels for a total of 6 weeks. Her iris and retinal lesions remained stable, and she was discharged. At age 5 months, the child presented with an acutely inflamed left eye (Figure 1). There were prominent iris vessels, corneal edema, and an intraocular pressure of approximately 40 mm Hg. No hypopyon was observed. She was treated initially with topical β-blocker and steroid. The intraocular pressure normalized, but the eye became more inflamed during the next 24 hours. Results of an ultrasonographic examination were negative for signs of intravitreal infection. She was taken to surgery, and lensectomy, vitrectomy, and peripheral iridectomy were performed through a limbal incision. A peripheral segment of the lens was opaque From the Department of Ophthalmology, Lions Eye Institute, Albany Medical College, Albany, New York. Received November 16, 1999. Revision accepted July 18, 2000. Reprint requests: John W. Simon, MD, Dept of Ophthalmology, Albany Medical College, 35 Hackett Blvd, Albany, NY 12208. J AAPOS 2001;5:50-1. Copyright © 2001 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/2001/$35.00 + 0 75/1/111136 doi:10.1067/mpa.2001.111136
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February 2001
white. The previously observed retinal lesion had become an atrophic scar. The findings from samples of the aqueous were negative, but the lens/vitreous aspirate was positive for C albicans on smear and culture (Figures 2 and 3). Amphotericin B (5 µg) was injected intravitreally and the child was admitted to the hospital for repeat staging and for intravenous amphotericin. Other treatments included oral 5-FC and topical betaxolol hydrochloride (Betoptic) and prednisolone acetate. Culture and sensitivities later showed the organism to be susceptible to both amphotericin B and 5-FC. Eight days after surgery, the patient required repeat vitrectomy because of pupillary block with a fibrin clot. Again, the vitreous culture was positive for C albicans. Amphotericin B and miconazole were injected intravitreally, and fluconazole was given orally. The pupillary membrane gradually recurred. Seven years later, the eye was quiet, with hand motions visual acuity. The poor vision was attributed primarily to amblyopia and to retinal damage from the infection. There have been no systemic manifestations of candidal infection.
DISCUSSION Systemic candidal infection occurs in 1% to 4% of premature births, especially in children with very low birth
FIG 1. Clinical photograph shows acutely inflamed left eye at age 5 months. The cornea was edematous, there were prominent iris vessels, and the intraocular pressure was approximately 40 mm Hg. The reflection overlying the pupil is from the ring flash.
Journal of AAPOS
Journal of AAPOS Volume 5 Number 1 February 2001
Stern, Calvano, and Simon
51
FIG 2. Smears from the vitreous show hyphal forms of Candida albicans.
FIG 3. Smears from the vitreous show yeast forms of Candida albicans.
weights.1-6 More recent studies have implicated C parapsilosis in addition to C albicans. Fortunately, relatively few of these patients develop ocular infection.2 Typically, these children develop a retinochoroiditis with overlying vitreous reaction. In most cases, systemic treatment with amphotericin B and 5-FC is effective. Intraocular surgery to acquire cultures, to remove infected tissue, and to inject antifungals is generally not necessary.3 Characteristic of candidal endophthalmitis, especially in adults, is a tendency for the infection to recur, perhaps because of sequestration in the lens. 4 Recurrence in children is rare, having been reported in only 2 cases, occuring 4 and 5 months after treatment had been completed.5,6 Diagnosis has proved difficult: Clinch et al6 initially diagnosed cataract, and we initially suspected glaucoma. In our case, diagnosis was especially difficult because of the negative results from the ultrasonographic examination. The lensectomy was performed, not only because of clinical suspicion that the lens was infected, but also to facilitate vitrectomy and intracameral injection of antifungal medications. Although we believe surgical intervention
to be rarely indicated, it may have helped to preserve the infected eye and avoid further systemic infection in this child. Recurrent candidal endophthalmitis should be suspected when premature infants with a history of candidemia have ocular inflammation, even if they were previously treated. References 1. Kossoff EH, Buescher ES, Karlowicz MG. Candidemia in a neonatal intensive care unit: trends during 15 years and clinical features of 111 cases. Pediatr Infect Dis 1998;17:504-8. 2. Baley JE, Annable WE, Kliegman RM. Candida endophthalmitis in the premature infant. Pediatrics 1998;98:458-61. 3. Annable WL, Dachmer ML, DiMarco M, DeSantis D. Long-term follow-up of Candida endophthalmitis in the premature infant. J Pediatr Ophthalmol Strabismus 1990;27:103-6. 4. Essman TF, Flynn HW, Smiddy WE, Brod RD, Murray TG, Davis JL, et al. Treatment outcomes in a 10 year study of endogenous fungal endophthalmitis. Ophthalmic Surg Lasers 1997;28:185-94. 5. Hill HR, Mitchell TG, Matsen JM, Quie PG. Recovery from disseminated candidiasis in a premature neonate. Pediatrics 1974;53:748-52. 6. Clinch TE, Duker JS, Eagle RC Jr, Calhoun JH, Augsberger JJ, Fischer DH. Infantile endogenous Candida endophthalmitis presenting as a cataract. Surv Ophthalmol 1989;34:107-12.
Endogenous Candida endophthalmitis resulting from candidemia in low-birth-weight infants usually occurs as a retinochoroiditis, which is effectively treated with systemic antifungal agents. We report a case of Candida endophthalmitis that recurred 4 months after completion of systemic antifungal therapy. The recurrent Candida infection affected primarily the iris and lens, rather than the retina and choroid. Vitrectomy was required for diagnosis and treatment.
CASE REPORT An 11-day-old girl was referred because of Candida albicans fungemia from a cutaneous source. The infant was premature (24 gestational weeks), with a birth weight of 800 g. Initial examination showed a perimacular white chorioretinal lesion and a small white iris nodule adjacent to the pupillary margin in the left eye. There was no evidence of lens involvement, and the right eye did not show signs of infection. The infant was treated with systemic amphotericin B and 5-fluorocytosine (5-FC) to maintain adequate serum levels for a total of 6 weeks. Her iris and retinal lesions remained stable, and she was discharged. At age 5 months, the child presented with an acutely inflamed left eye (Figure 1). There were prominent iris vessels, corneal edema, and an intraocular pressure of approximately 40 mm Hg. No hypopyon was observed. She was treated initially with topical β-blocker and steroid. The intraocular pressure normalized, but the eye became more inflamed during the next 24 hours. Results of an ultrasonographic examination were negative for signs of intravitreal infection. She was taken to surgery, and lensectomy, vitrectomy, and peripheral iridectomy were performed through a limbal incision. A peripheral segment of the lens was opaque From the Department of Ophthalmology, Lions Eye Institute, Albany Medical College, Albany, New York. Received November 16, 1999. Revision accepted July 18, 2000. Reprint requests: John W. Simon, MD, Dept of Ophthalmology, Albany Medical College, 35 Hackett Blvd, Albany, NY 12208. J AAPOS 2001;5:50-1. Copyright © 2001 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/2001/$35.00 + 0 75/1/111136 doi:10.1067/mpa.2001.111136
50
February 2001
white. The previously observed retinal lesion had become an atrophic scar. The findings from samples of the aqueous were negative, but the lens/vitreous aspirate was positive for C albicans on smear and culture (Figures 2 and 3). Amphotericin B (5 µg) was injected intravitreally and the child was admitted to the hospital for repeat staging and for intravenous amphotericin. Other treatments included oral 5-FC and topical betaxolol hydrochloride (Betoptic) and prednisolone acetate. Culture and sensitivities later showed the organism to be susceptible to both amphotericin B and 5-FC. Eight days after surgery, the patient required repeat vitrectomy because of pupillary block with a fibrin clot. Again, the vitreous culture was positive for C albicans. Amphotericin B and miconazole were injected intravitreally, and fluconazole was given orally. The pupillary membrane gradually recurred. Seven years later, the eye was quiet, with hand motions visual acuity. The poor vision was attributed primarily to amblyopia and to retinal damage from the infection. There have been no systemic manifestations of candidal infection.
DISCUSSION Systemic candidal infection occurs in 1% to 4% of premature births, especially in children with very low birth
FIG 1. Clinical photograph shows acutely inflamed left eye at age 5 months. The cornea was edematous, there were prominent iris vessels, and the intraocular pressure was approximately 40 mm Hg. The reflection overlying the pupil is from the ring flash.
Journal of AAPOS
Journal of AAPOS Volume 5 Number 1 February 2001
Stern, Calvano, and Simon
51
FIG 2. Smears from the vitreous show hyphal forms of Candida albicans.
FIG 3. Smears from the vitreous show yeast forms of Candida albicans.
weights.1-6 More recent studies have implicated C parapsilosis in addition to C albicans. Fortunately, relatively few of these patients develop ocular infection.2 Typically, these children develop a retinochoroiditis with overlying vitreous reaction. In most cases, systemic treatment with amphotericin B and 5-FC is effective. Intraocular surgery to acquire cultures, to remove infected tissue, and to inject antifungals is generally not necessary.3 Characteristic of candidal endophthalmitis, especially in adults, is a tendency for the infection to recur, perhaps because of sequestration in the lens. 4 Recurrence in children is rare, having been reported in only 2 cases, occuring 4 and 5 months after treatment had been completed.5,6 Diagnosis has proved difficult: Clinch et al6 initially diagnosed cataract, and we initially suspected glaucoma. In our case, diagnosis was especially difficult because of the negative results from the ultrasonographic examination. The lensectomy was performed, not only because of clinical suspicion that the lens was infected, but also to facilitate vitrectomy and intracameral injection of antifungal medications. Although we believe surgical intervention
to be rarely indicated, it may have helped to preserve the infected eye and avoid further systemic infection in this child. Recurrent candidal endophthalmitis should be suspected when premature infants with a history of candidemia have ocular inflammation, even if they were previously treated. References 1. Kossoff EH, Buescher ES, Karlowicz MG. Candidemia in a neonatal intensive care unit: trends during 15 years and clinical features of 111 cases. Pediatr Infect Dis 1998;17:504-8. 2. Baley JE, Annable WE, Kliegman RM. Candida endophthalmitis in the premature infant. Pediatrics 1998;98:458-61. 3. Annable WL, Dachmer ML, DiMarco M, DeSantis D. Long-term follow-up of Candida endophthalmitis in the premature infant. J Pediatr Ophthalmol Strabismus 1990;27:103-6. 4. Essman TF, Flynn HW, Smiddy WE, Brod RD, Murray TG, Davis JL, et al. Treatment outcomes in a 10 year study of endogenous fungal endophthalmitis. Ophthalmic Surg Lasers 1997;28:185-94. 5. Hill HR, Mitchell TG, Matsen JM, Quie PG. Recovery from disseminated candidiasis in a premature neonate. Pediatrics 1974;53:748-52. 6. Clinch TE, Duker JS, Eagle RC Jr, Calhoun JH, Augsberger JJ, Fischer DH. Infantile endogenous Candida endophthalmitis presenting as a cataract. Surv Ophthalmol 1989;34:107-12.