A rare case of Stenotrophomonas maltophilia endogenous endophthalmitis in pediatric age group

A 10-year-old male presented to the outpatient department of our hospital with complaints of loss of vision, redness, watering, and irritation in the right eye for 14 days. He was diagnosed elsewhere with a case of anterior uveitis for which he was started on hourly 1% prednisolone acetate and twice daily 2% homatropine eyedrops. Upon inquiry, he gave a history of fever 1 month ago along with a history of ear discharge in the left ear about 2 weeks before his eye symptoms started which resolved spontaneously. There was no history of ocular trauma or any previous ocular intervention (medical or surgical). On general physical examination, the child seemed under nourished, with thin and fragile hair. Upon further examination, the child’s weight (22 kg) and height (125 cm) fell below the 2nd standard deviation of normal according to the WHO (World Health Organization) weight for age and height for age charts [10].

The best corrected visual acuity was hand movements (HM) in the right and 20/20, N6 in the left eye. Intraocular pressure was recorded as 4 mm of mercury in the right eye and 12 mm hg in the left eye.

A slit lamp examination of the right eye revealed circumcorneal conjunctival congestion, a few keratic precipitates over the corneal endothelium with cells (grade 1), mild flare, and a streak hypopyon in the anterior chamber. The pupil was 5 mm dilated with posterior synechiae, a clear lens with dense vitreous exudates adherent to the posterior lenticular capsule was noted. There was no view of the fundus. The left eye examination was within normal limits.

B scan ultrasonography of the right eye revealed a large number of low-to-medium reflective dots, clumps, and membranous echoes in the vitreous with choroidal thickening and an attached retina (Fig. 1).

Fig. 1figure 1

B scan ultrasonography at presentation: Shows a large number of low-to-medium reflective dots, clumps, and membranous echoes in the vitreous with choroidal thickening and an attached retina

After taking a written, valid informed consent, and obtaining fitness for anesthesia, the patient underwent a right eye lensectomy with a core vitrectomy with intraocular antibiotics under general anesthesia on the same day on an emergency basis. Twenty-three-gauge pars plana vitrectomy was performed. Before the infusion fluid was turned on, an undiluted vitreous sample was sent for a microbiological exam which included gram stain, 10% KOH (potassium hydroxide) mount as well as plating on culture media (Blood agar, Chocolate agar, Sabouroud’s dextrose agar). Despite a clear lens, a lensectomy was performed due to the presence of dense exudates stuck to the posterior lens capsule and poor pupillary dilation which made it difficult to remove these exudates without causing lens injury. Moreover, lensectomy facilitated safe and thorough vitrectomy which was crucial for managing the fulminant endophthalmitis. Following lensectomy and clearing of the dense central vitreous exudates, a pale necrotic retina with extensive perivascular exudation was noted (Fig. 2).

Fig. 2figure 2

Intra-operative fundus photograph: Pale necrotic retina with extensive perivascular exudation seen after lensectomy and clearing of dense central exudates

Posterior vitreous detachment was not attempted, and core vitrectomy was completed. All ports were sutured with 7 − 0 vicryl, and intravitreal Vancomycin(1 mg/0.1 ml), ceftazidime(2.25 mg/0.1 ml), and dexamethasone(0.4 mg/0.1 ml) were injected at the end of the procedure. Postoperatively, the child was started on intravenous cefotaxime (500 mg 12 hourly), intravenous gentamicin (50 mg 8 hourly), oral prednisolone 20 mg once a day as well as topical prednisolone acetate hourly, atropine 4 hourly, and moxifloxacin hourly. On postoperative day 1, significant growth was observed on both blood and chocolate agar. Gram staining of the cultured bacteria revealed the presence of gram-negative bacilli (Figs. 3 and 4). Species identification and antibiotic susceptibility testing were pending.

Fig. 3figure 3

Gram’s stain of the teased growth from culture plate of vitreous exudate sample at 100x Magnification: Gram negative bacilli were noted

Fig. 4figure 4

Bacterial culture plates showing growth of Stenotrophomonas maltophilia on blood agar (right) and chocolate agar (left). Colonies on blood agar were non-hemolytic, with a faint lavender color and an ammonia odor. They formed small colonies after 24 h at 37 °C, and then formed smooth, convex colonies about 3 mm in diameter after 48 h

Urine and blood cultures showed no growth. Due to the fulminant clinical presentation, Pseudomonas sp. was suspected, and the child was administered intravitreal Imipenem (100µgm/ 0.1 ml) with dexamethasone(0.4 mg/0.1 ml) on postoperative day 1 due to the high risk of rapid clinical deterioration while awaiting identification and antibiotic sensitivity results. On postoperative day 3 bacterial isolate of Stenotrophomonas maltophilia was identified from culture using the Vitek microbiology analyzer followed by confirmatory biochemical tests which showed positive Catalase and negative Oxidase tests.

Antibiotic susceptibility testing performed using the Kirby Bauer disc diffusion method showed sensitivity to fluoroquinolones (ciprofloxacin, levofloxacin, ofloxacin, gatifloxacin), chloramphenicol, gentamicin, and resistance to both ceftazidime and imipenem. Intravitreal moxifloxacin (0.05 ml from topical moxifloxacin 0.5%) and dexamethasone(0.4 mg/0.1 ml) was injected on the same day and was repeated every alternate day till fundus examination showed resolution of perivascular exudation. A total of five doses of intravitreal moxifloxacin and dexamethasone were administered at 48-hour intervals. Intravenous antibiotics (cefotaxime 500 mg 12 hourly and gentamicin 50 mg 8 hourly) were continued till post-operative day 7 and subsequently, the child was given oral ciprofloxacin 250 mg twice a day for a week. Oral prednisolone was tapered weekly and stopped. At 1 month post operatively, the best corrected distance visual acuity was 20/80 with + 14.00 D of aphakic correction with a near visual acuity of N-10 with + 3.00 D addition. Fundus examination showed a resolving disc edema with fibrous bands causing traction over the macula and nasal retina with old sheathing on peripheral vessels. At the 6-month final follow-up, the best corrected visual acuity was maintained at 20/80(N-10) with stable fundus findings (Fig. 4). The child was using aphakic correction and was planned for a scleral fixated intraocular lens placement but was lost to follow-up (Figure 5).

Fig. 5figure 5

Colour Fundus photograph at 6 month follow up visit: Shows a resolving disc edema with fibrous bands causing traction over the macula and nasal retina with old sheathing on peripheral vessels

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