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Sequestration and Late Activation of Lenticular Candida Abscess in Premature Infants
Arch Ophthalmol. 2004;122:1393-1395.
Endogenous intraocular Candida infection typically presents as chorioretinitis with varying degrees of vitreous infiltration and inflammation. In patients with concurrent or recent candidemia, intralenticular fungal abscess is rare but has been reported previously in premature infants.1-3 We report a fourth and fifth case of this unusual syndrome, emphasizing its clinical signs, peculiar clinical course, and the possibility of a good visual outcome with appropriate therapy.
Report of Cases
Case 1. An infant girl was born at 24 weeks postconceptual age and weighed 750 g. The postnatal course was complicated by bronchopulmonary dysplasia, apnea, patent ductus arteriosis, anemia, hypertension, gastrointestinal reflux, and subclinical necrotizing enterocolitis. The patient also developed candidemia with blood and urine cultures positive for organisms at 3 weeks of age. She was found to have a large right atrial mass compatible with fungus that showed evidence of inferior vena cava obstruction. She was treated with amphotericin B and fluconazole for a total of 3 months. All cultures were negative for organisms for the final 2 weeks of her treatment prior to discharge from the neonatal intensive care unit. Serial ocular examinations revealed intralenticular opacities and a persistent tunica vasculosa lentis in the right eye by 3 weeks of age, which resolved itself by age 5 weeks. The left eye was not involved. At 3 months of age she was noted to have a chorioretinal scar in the right eye. At age 4 months, her mother observed a "white spot" at the edge of her pupil.
One month later, she was examined at our institution with a 3-day history of a red eye. It was noted that there was a mass in the pupil and beneath the iris inferotemporally, the cornea was cloudy, and there were dilated iris vessels as well as iris bombe. B-scan ultrasonography revealed no increase in vitreous opacities compared with the fellow eye. Examination results of the left eye were unremarkable except for 3 clock hours of stage 2, zone 3 retinopathy of prematurity. A fungal etiology was suspected and blood and urine cultures were obtained. The right eye was treated with prednisolone acetate 1% and atropine. After 1 week, the anterior segment inflammation was clearing with contraction of the anterior chamber mass. There was iris vessel dilation at the 9-o'clock position with a focus of white material at the pupillary border and underneath the iris. There were mildly increased vitreous opacities detected by ultrasonography.
An examination under anesthesia was performed (Figure 1). Intraoperatively, after performing an anterior chamber washout and cultures, the white mass was more clearly visualized. It was located at the temporal pupillary border, extending underneath the pupil and pushing the iris forward. The lens was opacified beneath this mass. An extracapsular cataract extraction was performed with aspiration of creamy-white material from beneath the iris and generous capsulectomy, anterior vitrectomy, and intravitreal injection of amphotericin B. Intraocular cultures grew Candida albicans (Figure 2). She received a 6-week course of amphotericin B because of previous cardiac problems. Her postoperative ocular course was uneventful. She was successfully treated with a Silsoft pediatric aphakic contact lens (Bausch & Lomb, Rochester, NY) and bifocal lenses, as well as part-time occlusion. The 2 small perimacular chorioretinal scars remained stable. Esotropia and right hypertropia were treated with eye muscle surgery. On the most recent follow-up at 31/2 years old, the patient's best-corrected vision in the right eye measured 5/200; her eyes were straight (Figure 3) and fundus unchanged.
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Figure 1. Case 1 had inflamed eye with a white mass beneath the pupil.
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Figure 2. The intraocular cultures in case 1 grew Candida albicans.
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Figure 3. After aggressive treatment, case 1 developed good visual function and cosmesis.
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Case 2. A boy, born at 25 weeks postconceptional age and weighing 855 g, had a postnatal course complicated by respiratory distress syndrome, pneumonia, anemia, patent ductus arteriosus, and hypotension. He developed candidemia and coagulase-negative staphylococcal sepsis that were treated with amphotericin and vancomycin, respectively, at an outside hospital.
All cultures were negative for organisms after transfer to our facility at 38 days of age. Serial dilated fundus examinations were performed from 7 weeks to 3 months of age, when threshold retinopathy of prematurity was diagnosed. Indirect laser photocoagulation of avascular peripheral retina using approximately 1300 green diode laser spots was performed bilaterally. Regression of the retinopathy was noted at the first postoperative examination 1 week later. During this examination a 1-mm, discrete, round, anterior lens opacity with an associated iridolenticular adhesion was noted superiorly in the left eye and felt to be consistent with a laser burn. Follow-up examinations through 6 months of age confirmed resolution of the retinopathy with no change in the lenticular opacity.
At 7 months of age the patient developed conjunctival injection in his left eye. Examination showed moderate corneal haze, iris bombe, miosis, and the sectoral cataract superiorly. The intraocular pressure was 46 mm Hg and was unresponsive to medical management. A trabeculotomy with iridectomy and lysis of posterior synechiae with removal of an anterior chamber membrane was performed. A red reflex was noted inferiorly through the miotic pupil. During the procedure, a thin, milky-white fluid flowed from the iridectomy and out through the limbal incision. A Candida abscess was suspected and the white material was cultured.
The intraocular pressure normalized postoperatively but the cultures grew C albicans on the second postoperative day. The patient was taken back to the operating room and examination after placement of iris hooks revealed a creamy-white lenticular opacity underlying the previous focal cataract. A cataract extraction with anterior vitrectomy and intravitreal injection of amphotericin B was performed. An infectious workup revealed a nidus in the patient's right kidney, consistent with a Candida abscess, that resolved itself during a 28-day course of systemic fluconazole. The postoperative course was uneventful with treatment consisting of a Silsoft aphakic contact lens and part-time occlusion. He had a strong right fixation preference but was able to fix and follow with his left eye on his most recent follow-up examination at 13 months of age.
Comment
To our knowledge these are the fourth and fifth reported cases of a Candida intralenticular abscess with a white plaque in the pupil after treated candidemia in a premature, low-birth-weight infant. Similar to a previous case,2 our patients responded well to aggressive treatment that included cataract extraction, anterior vitrectomy, and intravitreal injection of amphotericin B. Case 1 is unique in that intralenticular opacities were noted in the affected eye at the time of initial candidemia. The opacities resolved with antifungal treatment. Of interest is the fact that all reported cases of endogenous intralenticular fungal abscess have occurred in premature infants. We believe that in such eyes, the persistent tunica vasculosa lentis serves as the route for lenticular seeding by fungal organisms at the time of Candida septicemia. As the tunica regresses, Candida organisms become sequestered adjacent to and within the lens, resulting in inadequate treatment by systemic antifungal therapy. The lenticular abscess then slowly activates, appearing months later as a focal lens opacity accompanied at first by relatively little intraocular inflammation. Case 1 represents the first infant treated aggressively who developed good visual function and cosmesis. Case 2 is the first case to present as glaucoma.
Our cases and review of the literature suggest that premature neonates who have been successfully treated for systemic candidiasis remain at risk for developing sequestration of fungal organisms within the lens, which later appears as a focal lens opacity that evolves into a white papillary plaque up to 6 months following treatment. Close ophthalmologic follow-up and parental education regarding this presentation is recommended for this group of patients. As demonstrated by these cases, a high index of suspicion and aggressive therapy can result in an excellent clinical outcome.
The authors have no relevant financial interest in this article.
AUTHOR INFORMATION
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Rita Singh-Parikshak, MD;
Erick D. Bothun, MD;
Roseanne Superstein, MD;
Monte Del Monte, MD;
Steven Archer, MD;
Mark W. Johnson, MD
Correspondence: Dr Singh-Parikshak, Simpson Eye Associates, 650 Springhill Ring Rd, West Dundee, IL 60118 (ritasing10{at}hotmail.com).
REFERENCES
1. Hill H, Mitchell TG, Matsen JM, Quie PG. Recovery from disseminated candidiasis in a premature neonate. Pediatrics. 1974;53:748-752.
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2. Clinch T, Duker JS, Eagle RC Jr, Calhoun JH, Augsburger JJ, Fischer DH. Infantile endogenous Candida endophthalmitis presenting as a cataract. Surv Ophthalmol. 1989;34:107-112.
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3. Shah G, Vander J, Eagle RC. Intralenticular Candida species abscess in a premature infant. Am J Ophthalmol. 2000;129:390-391.
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