Fungal choroidal abscess in a case of aggressive retinopathy of prematurity

Taranpreet Kaur; Vipan Kumar Vig; Inderjit Kaur; Sunita Arora

doi:10.4103/jcor.jcor_213_20

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Year : 2021 | Volume : 9 | Issue : 3 | Page : 132-136

Fungal choroidal abscess in a case of aggressive retinopathy of prematurity

Taranpreet Kaur¹, Vipan Kumar Vig², Inderjit Kaur¹, Sunita Arora³
¹ Department of Ophthalmology, Sri Guru Ram Das Institute of Medical Sciences and Research, Amritsar, Punjab, India
² Sardar Bahadur Dr Sohan Singh Eye Hospital, Amritsar, Punjab, India
³ Department of Paediatrics, Sri Guru Ram Das Institute of Medical Sciences and Research, Amritsar, Punjab, India

Date of Submission	29-Sep-2020
Date of Decision	16-Feb-2021
Date of Acceptance	03-Mar-2021
Date of Web Publication	27-Sep-2021

Correspondence Address:
Taranpreet Kaur
Department of Ophthalmology, Sri Guru Ram Das Institute of Medical Sciences and Research, Vallah, Amritsar, Punjab
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jcor.jcor_213_20

Abstract

Fungal choroidal abscesses are rarely reported entities on routine retinopathy of prematurity screenings done on neonates. With the increasing use of broad-spectrum intravenous antibiotics on preterm neonates during their stay in neonatal intensive care unit who are already immunodeficient, we hereby bring this case into notice so as to keep fungal etiologies as a close differential in comparison to bacterial choroidal abscesses, the latter being more common in the older age group. The late diagnosis of fungal choroidal abscess and the lack of systemic treatment for the same could possibly lead to acquired hydrocephalus and other systemic complications.

Keywords: Aggressive posterior retinopathy of prematurity, candidemia, fungal choroidal abscess, hydrocephalus, neonatal intensive care unit, neonatal sepsis, retinopathy of prematurity, ventriculoperitoneal shunt

How to cite this article:
Kaur T, Vig VK, Kaur I, Arora S. Fungal choroidal abscess in a case of aggressive retinopathy of prematurity. J Clin Ophthalmol Res 2021;9:132-6

How to cite this URL:
Kaur T, Vig VK, Kaur I, Arora S. Fungal choroidal abscess in a case of aggressive retinopathy of prematurity. J Clin Ophthalmol Res [serial online] 2021 [cited 2023 Jun 8];9:132-6. Available from: https://www.jcor.in/text.asp?2021/9/3/132/326791

Fungi are a relatively uncommon cause of choroidal abscess in neonates and early infancy. They are usually associated with predisposing factors such as prematurity, low birth weight, use of broad-spectrum antibiotics, and a prolonged stay in the neonatal intensive care unit (NICU). The incidence of disseminated candidiasis has been variably reported as 1% of all NICU admissions, 4% of infants with birth weights <1500 g, and 10%–28% of infants weighing <1000 g at birth.^[1] Candida-related chorioretinal involvement was reported in 6% of total infants suffering from disseminated candidiasis.^[2] The chorioretinal lesions may be unilateral or bilateral.^[3] They most often appear as multiple, although occasionally single, yellow-white, elevated lesions with indistinct borders located in the posterior fundus. Vitreous lesions are occasionally seen.^[1],[3] Although most often found in the macular area, they may be found in the retinal periphery. They typically resemble fungal colonies on laboratory media but may also appear as hard exudates with distinct borders.^[1]

Their fortuitous detection as fungal seedlings in an eye of a preterm during routine retinopathy of prematurity (ROP) screening often times is a harbinger of a systemic fungal infection.^[4],[5] Neonatal candidal sepsis is associated with worse grades of ROP,^[6] and detection of fungal elements is difficult both on laboratory investigations as well as clinically because of their late presentations.^[7]

Case Report

A preterm neonate born to a secondary gravida by lower-segment cesarean section at 35-week gestational age with birth weight 1.46 kg was admitted to NICU with respiratory distress. On laboratory examination, his total leucocyte count was 19,000/μl which increased to 53,000/μl within 3 days of admission in NICU with neutrophils accounting for 84% pointing in favor of acute sepsis.

Chest X-ray showed hyaline membrane disease. Blood hemoglobin was 17.7 mg/dl which dropped down to 11.4 mg/dl for the next 1 week. Intravenous (IV) antibiotic injection cefotaxime sodium 50 mg I/V 8 h for 3 days and injection amikacin 10 mg I/V 12 hourly for 5 days were started with inotropic support and surfactant therapy from the 1^st day of life (DOL) of the neonate.

In view of clinical worsening with rising C-reactive protein (CRP) from 1.4 to 11.4, the first blood culture was sent which was found to be negative for both bacteria and fungi. Injection piperacillin 100 mg I/V 8 hourly for 8 days was added on 4^th DOL.

Injection meropenem 30 mg I/V 12 hourly for 8 days was added on 7^th DOL in view of increased CRP from 11.4 to 34.3. The second blood culture was sent and was again negative.

As CRP continued to rise from 34.4 to 101, injection vancomycin 25 mg I/V 8 hourly for 12 days was added on 10^th DOL after sending third blood culture which was again negative both for bacteria and fungi. Peripheral blood film examination on 15^th DOL revealed toxic metamyelocyte granules. Injection linezolid 15 mg I/V 8 hourly and injection colistimethate 40,000 IU I/V 8 hourly for 14 days were added finally leading to improvement both systemically and on laboratory investigations.

A routine indirect ophthalmoscopy examination for ROP at 3 weeks, i.e., 21^st DOL, revealed dilated and tortuous veins over the posterior pole in both eyes and an incidental finding of a choroidal abscess in the avascular retinal periphery of the right eye[Figure 1]a and [Figure 1]b. Diagnosis of aggressive posterior retinopathy of prematurity (APROP) zone 2 posterior both eyes with choroidal abscess in the right eye was made.

Figure 1: (a) Fundus images captured through an insufficiently dilating pupil by Smartphone-based fundus camera device (MII Ret Cam) and +28 D showing dilated and tortuous vessels over optic disc. (b) Fundus images captured through Miitcam and 28 D showing dilated and tortuous vessels as seen in aggressive posterior retinopathy of prematurity although abscess could not be captured due to its peripheral location

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Abscess was around 2 disc diameter in size located in the avascular retinal periphery in the inferotemporal quadrant (7'o' clock position) with one edge of abscess touching ora serrata. The eye was quiet with media being clear and no overlying vitritis near the abscess area in the right eye [Figure 2].

Figure 2: Schematic retinal diagrams with above two showing active choroidal abscess in the right eye with dilated and tortuous veins all quadrants of both eyes with below two schematic diagrams showing regressing choroidal abscess in the right eye after intravitreal amphotericin and ranibizumab with subsidence of aggressive posterior retinopathy of prematurity in both eyes

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On external examination, the eye was quiet. Since the neonate had both entities APROP and choroidal abscess in the right eye, it was decided to manage APROP by lasering both eyes with frequency-doubled neodymium-doped yttrium aluminum garnet green laser (532 nm) and giving intravitreal antivascular endothelial growth factor in the left eye first.

Blood sample was again sent for bacterial and fungal cultures, which did not show any growth.

Fundus examination was repeated every 3 days to observe the rate of increase in the size of abscess and to observe any other external signs of congestion in that eye. The neonate remained afebrile during all these days.

Since the abscess hardly increased in size with noncongested eye externally, with the neonate being afebrile, along with aggressive antibiotic course in NICU, a provisional diagnosis of fungal choroidal abscess was made and an empirical trial of intravitreal amphotericin 25 μg (one half adult dose) was given in right eye along with intravitreal ranibizumab 0.016 mg (one third adult dose) and 0.025 mg (half adult dose) intravitreal ranibizumab in the left eye too.

The neonate started responding to empirical intravitreal trial of antifungal, as abscess started decreasing in size. APROP also started regressing as the vessels decreased in dilatation and tortuosity. The skip area underneath the abscess was also covered with laser spots. At 1 month of follow-up, abscess changed its color from bright white to dirty gray, but still, some residual part could be seen.

The neonate was also advised a course of systemic antifungal in view of abscess but could not be given due to noncompliance of the parents for further treatment.

One-month post-ROP laser treatment, the neonate presented in the eye outpatient department (OPD) with sunset signs in eyes and bulging anterior fontanelle, which was further referred in the pediatric OPD to rule out hydrocephalus.

On ultrasound of the head, hydrocephalus was confirmed. On magnetic resonance imaging (MRI) of the brain, grossly enlarged third and fourth ventricles could be seen [Figure 3].

Figure 3: Magnetic resonance imaging brain showing moderate supratentorial ventriculomegaly with small hemorrhagic foci in bilateral periventricular and subcortical regions

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On systemic examination, there was no fever or suckling inability.

Acquired hydrocephalus was diagnosed as it was gradual in onset and slowly progressive with no other neurological signs in the neonate. The neonate was immediately operated for ventriculoperitoneal (VP) shunt operation. Lumbar and shunt cerebrospinal fluid (CSF) cultures and India ink preparations were negative for organisms.

As the choroidal abscess responded formerly to empirical trial of intravitreal amphotericin (25 μg), I/V fluconazole (6 mg/kg/day) for 4 weeks was added in the treatment along with I/V antibiotics in the postoperative period in view of fungal meningitis as the probable cause of acquired hydrocephalus.

The neonate was followed up for 1 year, and no recurrence was seen for hydrocephalus, except for delayed motor milestones.

At 1 year follow-up, on systemic examination, there were delayed motor milestones. VP shunt was functional with normal MRI [Figure 4]. On visual acuity examination, the infant followed objects with fundus examination revealing pallor of optic discs with regressed ROP [Figure 5]a and [Figure 5]b.

Figure 4: Magnetic resonance imaging postventriculoperitoneal shunt showing nondilated ventricle system. Hyperintensities seen on T2-weighted images in the right frontal, occipital, and posterior parietal lobes appearing hypointense on T1-weighted images with no restriction on diffusion-weighted images with adjacent areas of hemorrhagic foci

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Figure 5: (a) Right eye fundus image captured by Miitcam and +28 D showing pale optic disc and regressed retinopathy of prematurity. (b) Left eye fundus image captured by Miitcam and +28 D showing pale optic disc and regressed retinopathy of prematurity

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Discussion

Preterm neonates are often immunocompromised. Septicemia with aggressive IV antibiotics makes them prone to fungal seedlings in the eye,^[4],[5] brain, and other systemic locations.^[8] Most commonly, the infective organism reaches the eye through the posterior vasculature. In our case, involvement of the right eye resulted from endogenous spread to the right eye, due to direct route through the right carotid artery.^[7],[9]

Fungal choroidal abscess in this case probably resulted due to fungal septicemia during initial few weeks after birth, further corroborated by negative blood cultures for bacteria. In fungal meningitis with negative csf fungal cultures, acquired hydrocephalus may be the only presenting feature.

Frequently initial clinical features of candidal meningitis are indistinguishable from those of other causes of systemic infection in premature neonates, and CSF parameters within normal range do not exclude meningitis.^[6] In the retrospective study by Fernandez et al., one neonate had hydrocephalus with multiple microabscesses on computed tomography scan, who was started on intravenous amphotericin and responded well, and did not require VP shunt operation.^[6]

Candidemia is itself implicated in progression of ROP to Stage 3 or beyond, possibly due to induction of cytokines and angiogenic factors.^[1]

Mittal et al.^[10] reported a higher incidence of ROP in neonates with Candida sepsis as compared to those who did not have Candida sepsis. This association was further proven in Tadesse et al.^[11] in their study.

Since the choroidal abscess in the right eye was seen in the avascular zone of the retina, it further supports its sequestration at the choroidal level through hematogenous route.

The diagnosis of fungal meningitis is notoriously difficult, even when considered. Because of the dynamics of CSF flow and the propensity for organisms to infect the basilar meninges, fluid sampled from the lumbar subarachnoid space is often devoid of organisms. Lumbar and shunt CSF cultures and India ink preparations were negative for organisms in our case which is the same as reported by Mangham et al.^[12]

Fungal choroidal abscesses are rarely reported entities. Choroidal abscess could be the only sign of systemic fungal infection in preterm neonates who can even present with fungal meningitis and its complications like hydrocephalus.

In such cases, intravitreal trial of antifungal agent can pinpoint the etiological agent as in our case where CSF culture did not reveal any pathological agent. Acquired hydrocephalus with delayed milestones could have been prevented if the neonate had received early IV Antifungal (Fluconazole ^[13],[14]6mg/kg/day,Amphotericin B ^[3] 15mg/kg/day for 4-6 weeks) with 5 Fluorocytocine (150mg/kg/day) by mouth,as the choroidal abscess responded well to intravitreal antifungal.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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