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Contact Lens & Anterior Eye 35 (2012) 233– 235

Contents lists available at SciVerse ScienceDirect

Contact Lens & Anterior Eye

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ase report

terile keratitis after corneal collagen crosslinking in a child

itu Arora, Parul Jain ∗, Deepa Gupta, J.L. Goyaluru Nanak Eye Centre, Maulana Azad Medical College, New Delhi 110002, India

r t i c l e i n f o

rticle history:eceived 26 December 2011

a b s t r a c t

Corneal collagen crosslinking (CXL) is a non invasive technique used in halting the progression of kerato-conus. Complications with this modality are rare. We report a case of an 8-year-old child who developed

eceived in revised form 2 June 2012ccepted 18 June 2012

eywords:KCrosslinkingeratitis

sterile infiltrates in the immediate postoperative period after uneventful corneal collagen crosslinkingfor keratoconus. The infiltrates resolved with topical steroid therapy. There was also present coexistingvernal keratoconjunctivitis (VKC) which was controlled with topical 2% cyclosporine A eye drops prior tocrosslinking treatment. This case highlights importance of controlling VKC prior to CXL in keratoconusas it adds to the risk of developing post operative sterile keratitis.

© 2012 British Contact Lens Association. Published by Elsevier Ltd. All rights reserved.

. Introduction

Keratoconus is a slowly progressive, non-inflammatory cornealhinning disorder characterized by changes in the structure andrganization of corneal collagen [1]. The ectasia progresses at aariable rate. The progression may be more rapid in the pread-lescent population with VKC so much so that many times theyresent with acute hydrops [2]. Hard contact lenses have been theajor treatment modalities for keratoconus in age group >16 years.

ome patients though, may benefit from intracorneal ring segmentmplantation [1,3], while others may need surgical intervention.hese modalities, however, have not been tried in paediatric ageroup patients with keratoconus. None of these techniques, how-ver, treat the underlying cause of ectasia and thus cannot stop therogression of the disease [4].

Corneal collagen crosslinking (CXL), introduced by Wollensakt al. [4], is a non invasive technique that increases the biomechan-cal strength of the human cornea by about 300% by the combinedction of a photosensitizing substance (riboflavin) and ultravio-et light from a solid state UVA (ultraviolet A) source. It has beenhown to stabilize the progressive keratoconus with only isolatedeports of side effects like diffuse lamellar keratitis, herpetic ker-titis with iritis, development of corneal haze and corneal melting5–10]. To the best of our knowledge there is no report of severeterile keratitis, post CXL in a child.

We herein report a case of severe sterile keratitis after CXL with

iboflavin and UVA for the treatment of grade III keratoconus in an-year-old male child.

∗ Corresponding author. Tel.: +91 9971927902.E-mail address: pjain811@gmail.com (P. Jain).

367-0484/$ – see front matter © 2012 British Contact Lens Association. Published by Elsttp://dx.doi.org/10.1016/j.clae.2012.06.002

2. Case report

An 8-year-old healthy male child presented with rapidly pro-gressive reduction of vision in both eyes of six months duration. Theleft eye vision was poorer than the right eye. There was associatedsevere itching in both the eyes. This was his first ophthalmologicalevaluation. He was not on any oral/topical medication. There wasno significant birth history.

His uncorrected visual acuity was 4/60 OD which improved to6/24 with −8.0 DS/−6.0 Dcyl @ 180◦ and 3/60 OS with no furtherimprovement in refraction. He was not using any visual aid at pre-sentation and there was no history of any ocular examination priorto presentation. There were present giant cobblestone papillae inthe upper palpebral conjunctiva in both eyes suggestive of ver-nal keratoconjunctivitis. He also had central corneal thinning inboth eyes with additional stromal scarring in the left eye. Intraoc-ular pressure was 14 mm of Hg in both the eyes. An Orbscan (OD)revealed a pattern consistent with keratoconus with maximum ker-atometry (Kmax) of 54.5 D and thinnest corneal thickness of 413 �.The thinnest corneal thickness was 417 � on ASOCT (OPTOVUECAM 2.7). Orbscan of the left eye could not be done because ofpoor imaging. The child was diagnosed as Krumeich [11] stageIII keratoconus in the right eye and Krumeich stage IV in the lefteye. He was planned for crosslinking with riboflavin for the righteye and penetrating keratoplasty in the left eye. Prior to undergo-ing crosslinking the child was treated with topical 2% cyclosporineeye drops four times a day for six weeks to control the coexistingsevere vernal conjunctivitis. The ocular examination repeated at 6

weeks after initial presentation did not reveal any further changesin topography and there was reduction in upper palpebral papillae.

The child subsequently underwent uneventful cornealcrosslinking of the right eye under general anaesthesia. The

evier Ltd. All rights reserved.

234 R. Arora et al. / Contact Lens & Anterior Eye 35 (2012) 233– 235

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margin, due to tear pooling in the gutter formed by the flap edge.Our patient presented with corneal infiltrates of severe intensity

from the first post operative day. They were present at the marginsof debrided epithelium. Absence of pain, worsening of infiltrates

Fig. 1. First post op day.

entral 8.0 mm epithelium was mechanically debrided withinhe circle marked with corneal trephine with hockey-stick blade.nstillation of 4 drops of a solution containing riboflavin 0.1% andextran 20%(Ricrolin SOOFT, Italia Inc.) every 2 min for approx-

mately 30 min was done. After the instillation of the riboflavinolution, the UV lamp was turned on to irradiate 8.0 mm diameterf central cornea. Riboflavin 0.1% solution was applied every 5 minuring the irradiation. The UV (wavelength 370 nm) source was

solid-state device consisting of 1 UV diode Vega CBM X linkerCSO, Florence, Italy). The power of the UVA 370-nm light sourceas checked before treatment using a UV-meter included with theV-XTM cross-linking device. The source was focused on the apexf the cornea at a distance of 50 mm to obtain a radiant energyf 3 mW/cm2. The cornea was irradiated with the UVA diode for0 min. After treatment, the surface of the eye was washed with0 ml balanced salt solution. A bandage contact lens (Purevisionausch & Lomb) was placed at the end of surgery. Postoperatively,opical chloramphenicol drops 0.3% 4 times daily (preservativeree), lubricants (Tears naturale, Alcon) 2 hourly, cyclopentolate% drops twice daily and ibuprofen 200 mg three times a day wererescribed.

The next day (post operative day one) the child complainedf severe photophobia and blurring of vision in right eye. Thereas present unusual stromal infiltration in the mid peripheral

ornea from 1 o’clock to 4 o’clock with associated keratic precipi-ates and an anterior chamber reaction. The bandage contact lensas removed and an hourly regimen of topical fortified cefazolin

50 mg/ml) and fortified tobramycin (15 mg/ml) was institutedlong with cyclopentolate 1% drops (3 times a day) and tearubstitutes (Tears natural, Alcon). There was also present severeongestion of the upper tarsal conjunctiva. Smears from cornealcraping did not reveal any organism. The intensive antibiotic reg-men was, however, continued. Cultures at the end of 24 h did nothow any growth. With the worsening of corneal infiltrates extend-ng further circumferentially till 9 o’clock over next 24 h (Fig. 1)nd associated corneal oedema and absence of pain the infiltratesere presumed to be of sterile origin. Low dose topical steroid (0.5%

oteprednol) drops was added to the treatment regimen. The childas further evaluated six hourly for any improvement or worsening

f signs and symptoms. There was no further progression of infil-rates. Loteprednol was replaced with two hourly 1% prednisolonecetate on the 3rd day. Resolution of the marginal infiltrates startedt 1 week leaving a central 5 mm of corneal oedema with infiltra-ion. Frequency of topical steroids was further increased to hourlyegimen and topical antibiotics were reduced to 3 times daily. With

rogressive improvement of corneal infiltrates the topical steroidsere tapered to four times a day at 4 weeks. At 6 weeks after pre-

entation there was complete resolution of infiltrates with totalpithelial healing (Fig. 2) and all topical medications except tear

Fig. 2. Three weeks post treatment.

substitutes were tapered and stopped. Stromal scarring was seenat 8 weeks which reduced in density over next 4 weeks. The uncor-rected visual acuity improved to 6/36 from 4/60 at 3 months aftercrosslinking (Fig. 3).

3. Discussion

The advent of corneal collagen crosslinking is one of the morepromising developments of this decade in the management of ker-atoconus. It appears to have the potential to reduce the morbidityof progressive forms of this disease and may ultimately reduce theneed for corneal transplantation. Isolated case reports of infectiveand sterile infiltrates post CXL have been reported in the literature.There also exists a series of four cases with severe sterile kerati-tis and superficial stromal scarring post CXL [12]. These reportsdescribe sterile keratitis occurring in individuals with age rang-ing from 17 to 40 years. Sterile infiltrates and iritis were seen 2–5days after the CXL procedure. Hypoxia induced from bandage con-tact lens, allergies, use of topical NSAIDs, alcohol used for epithelialdebridement, modified response to UV rays, reaction to UV rays,overdose of UV rays, over irradiation of corneal surface have beenhypothesized as factors for severe corneal inflammation and sterilecorneal infiltrates.

Sterile corneal stromal infiltrates have been described as a resultof enhanced cell-mediated immunity to staphylococcal antigensdeposited at high concentrations in areas of static tear pooling [5].They are usually located in peripheral cornea, probably due to tearpooling at the lid margins in rosaceae keratitis, meibomian glanddysfunction conditions. Post LASIK they may be localized to flap

Fig. 3. Resolution of infiltrates at six weeks follow up visit.

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n intensive topical antibiotic regimen and non isolation of anyrganism from corneal scrapings on smear examination and cultureere strong pointers towards sterile nature of infiltrates. Hypoxia

nduced from bandage contact lens cannot be attributed to be aausative factor as the contact lens was silicone hydrogel high oxy-en permeability contact lens. Overdosage of UV rays from singleED source was an unlikely contributing factor as care for preciseocusing from UV source was taken. Here, probably young age ofatient with coexistent severe VKC contributed towards a stateith enhanced cell mediated immunity to either riboflavin or UV

ays. VKC is present in nearly 40–50% of patients with keratoconusnd is also thought to contribute to this aetiology by inducing severeubbing of eyes [13]. Prognosis of penetrating Keratoplasty/deepnterior lamellar keratoplasty is also guarded in these patientsecause of associated dry eye, limbal stem cell deficiency and vac-larization. CXL in children with VKC and keratoconus should beerformed with extreme caution for the fear of sterile keratitis postXL. Severe corneal inflammation in the form of sterile infiltratesay be anticipated and managed accordingly. There may occur con-

equent stromal scarring and reduction in BCVA. However stromalcarring and reduction in density contributed to improved UCVAue to flattening of the cone.

onflict of interest

None of the authors have any financial interests to disclose orny conflict of interest.

ontributors

All authors have (1) substantial contributions to conception andesign, acquisition of data, analysis and interpretation of data; (2)

[

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rior Eye 35 (2012) 233– 235 235

drafting the article and revising it critically for important intellec-tual content; and (3) final approval of the version to be published.

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[2] Rehaney U, Reumelt S. Corneal hydrops associated with vernal conjunc-tivitis as a presenting sign of keratoconus in children. Ophthalmology1995;102(12):2046–9.

[3] Colin J, Velou S. Current surgical options for keratoconus. Journal of Cataractand Refractive Surgery 2003;29:379–86.

[4] WollensakG, SpoerlE, Seiler T. Riboflavin/ultraviolet-A-induced collagencrosslinking for the treatment of keratoconus. American Journal of Ophthal-mology 2003;135:620–7.

[5] Koppen C, Vryghem JC, Gobin L, Tassignon MJ. Keratitis and corneal scar-ring after UVA/riboflavin cross-linking for keratoconus. Journal of RefractiveSurgery 2009;25:S819–23.

[6] Zamora KV, Males JJ. Polymicrobial keratitis after a collagen cross-linkingprocedure with postoperative use of a contact lens: a case report. Cornea2009;28:474–6.

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[8] Perez-Santonja JJ, Artola A, Javaloy J, Alio JL, Abad JL. Microbial keratitisafter corneal collagen crosslinking. Journal of Cataract and Refractive Surgery2009;35:1138–40.

[9] Rama P, Di Matteo F, Matuska S, Paganoni G, Spinelli A. Acanthamoeba kerati-tis with perforation after corneal crosslinking and bandage contact lens use.Journal of Cataract and Refractive Surgery 2009;35:788–91.

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11] Krumeich JH, Daniel J, Knlle A. Live-epikeratophakia for keratoconus. Journalof Cataract and Refractive Surgery 1998;24:456–63.

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13] Harrison RJ, Klouda PT, Easty DL, Manku M, Charles J, Stewart CM. Asso-ciation between keratoconus and atopy. British Journal of Ophthalmology1989;73:816–22.