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Management of Inflammatory Corneal Melt Leading to Central Perforation In Eye (2016) 30, 593–601 © 2016 Macmillan Publishers Limited All rights reserved 0950-222X/16 www.nature.com/eye 1 2 2 Management of A Medsinge , E Gajdosova , W Moore CLINICAL STUDY and KK Nischal1,2,3 inflammatory corneal melt leading to central perforation in children: a retrospective study and review of literature Abstract Purpose To assess the outcome of early Waiting may sometimes allow the marked therapeutic penetrating keratoplasty (PKP) inflammatory response seen in children to for corneal melt leading to perforation in cause irreversible structural and/or functional children. damage. Methods Case notes of all the consecutive Eye (2016) 30, 593–601; doi:10.1038/eye.2015.278; patients presenting with acute corneal published online 29 January 2016 perforation that underwent urgent therapeutic PKP between 2000 and 2010 to the practice Introduction of one of the authors, both NHS at Great Ormond Street Hospital for Children and Non-traumatic corneal perforation in children is private, were retrospectively reviewed. Onset rare, but may be seen in neurotrophic ulcers,1 of perforation, underlying cause, medical and infectious keratitis,2 peripheral ulcerative surgical treatment, pre- and post-operative keratitis,3–5 ocular surface diseases such as visual acuity, graft clarity, length of follow- blepharokeratoconjunctivitis (BKC) with or 1 up, and post-operative complications were without rosacea,6,7 and chemical burns.8 Department of ’ recorded. Ophthalmology, Children s Systemic conditions including juvenile Hospital of Pittsburgh of Results 9,10 Four eyes of four consecutive rheumatoid arthritis and Stevens Johnson UPMC, Pittsburgh, PA, USA patients (mean age of 9.5 years and median syndrome11,12 can rarely cause corneal – 2 8.5 years, range 4 17 years) were treated for perforation, while in developing countries, Clinical and Academic acquired acute onset corneal perforations. vitamin A deficiency is a common cause of Department of Ophthalmology, Great There were three females and one male. non-infectious corneal melt in children.13 Ormond Street Hospital for Etiologies included herpes simplex keratitis The diagnosis and treatment of corneal Children, London, UK secondary to immune recovery disease post perforation in children is difficult, because bone marrow transplantation, acanthamoeba they are often unable to communicate their 3University of Pittsburgh keratitis, recessive dystrophic epidermolysis symptoms clearly, have limited cooperation School of Medicine, bullosa, and blepharokeratoconjunctivitis and many cases present late. Pittsburgh, PA, USA with acne rosacea. Pre-operative visual acuity To complicate the issue further, children Correspondence: ranged from hand movements to 6/150. can lose vision not only from subsequent KK Nischal, Department of All the patients had severe anterior chamber scarring but also from amblyopia. Ophthalmology, Children’s inflammation. All eyes improved in visual Although the literature has reports about Hospital of Pittsburgh of acuity ranging from 6/9 to 6/18 with clear management of bacterial and fungal keratitis UPMC, 4401 Penn Avenue, – FP 5106, Pittsburgh, grafts at last follow-up. There was no in mixed cohorts of adults and children,2,14 18 PA 15224, USA. recurrence of melt or perforation. especially in developing countries, there is little Tel: +1 412 692 8940; Mean follow-up was 67 months (median about the management of protozoal or viral Fax: +1 412 692 3203. 44 months). keratitis and even less about non-infectious E-mail: [email protected] Conclusion PKP during the acute phase keratitis leading to perforation in children. together with aggressive medical therapy and We present four cases of central corneal melt Received: 11 April 2015 Accepted in revised form: close follow-up may achieve good visual leading to perforation, all secondary to an 17 November 2015 fl outcomes in children with corneal melt with in ammatory etiology (two infectious and two Published online: perforation and should be considered. non-infectious) and all treated successfully with 29 January 2016 Management of central inflammatory corneal melt in children A Medsinge et al 594 Table 1 Demographic profile of the patients Case no. Age (years)/ Predisposing condition BCVA of the BCVA of Time FU (M) sex affected the between eye at affected eye perforation presentation at last FU and PKP 1 4/F Acanthamoeba keratitis HM 6/9 24 h 90 M 2 17/F Epidermolysis bullosa, MGD HM 6/9 7 days 26 M 3 12/F MGD, Blepharokeratoconjunctivitis secondary 6/150 6/9 Less than 2 weeks 62 M to acne rosacea 4 5/M HSK secondary to immune recovery disease s/p HM 6/18 Less than 2 weeks 90 M BMT for severe combined immunodeficiency (SCID) Abbreviations: M, male; F, female; MGD, Mebomian Gland Dysfunction; BCVA, best-corrected visual acuity; HM, hand movements; FU, follow-up; M, months; PKP, penetrating keratoplasty; S/p BMT, status post bone marrow transplantation; ft, feet. prompt and early therapeutic penetrating keratoplasty oral steroids postoperatively. All the post-operative (PKP), while the eyes were still inflamed. The degree of medications are shown in Table 2. All the cases were inflammation prompted intervention as it was felt that followed up for a mean period of 67 months and median despite anti-inflammatory therapy, damage sustained by of 44 months (range 26–90 months). Cases are described inflammation would be too detrimental. in detail below. The results of the literature review are summarized in Table 3. Materials and methods Case 1 Case notes of all consecutive patients between 2000 and 2010 with acquired corneal melt and perforation who A 4-year-old girl was reoffered to GOSH with a diagnosis presented to one surgeon (KKN) in a single tertiary of left eye Herpes Simplex Virus (HSV) keratitis. care pediatric hospital or private office (KKN) were Corneal scrapings at the outside hospital did not grow retrospectively reviewed. At Great Ormond Street any organisms, and she was treated empirically with Hospital for Children, this study was considered to topical as well as oral acyclovir and topical steroids. be a part of an audit and as such not considered to On presentation, her BCVA was hand movements (HM) require ethics committee approval. Onset of perforation, in the left eye. Right eye was unremarkable. She underlying cause, medical and surgical treatment, pre- and responded well to the topical treatment with remarkable post-operative visual acuity, length of follow-up, and post- improvement in vision to 6/12 in 2 weeks. At this stage, operative complications were recorded. A literature review the steroids were tapered. Soon after tapering the steroids, was performed using PubMed using the MeSH terms the keratitis recurred with a decrease in the visual acuity corneal melt, perforation, keratoplasty, and children. to counting fingers. She was again started on intensive steroids and her improved to 6/30. Three months after her initial presentation, she developed corneal epithelial Results defect for which a temporal tarsorrhaphy was performed Four eyes of four patients (mean age of 9.5 years and in addition to the treatment with topical and systemic median 8.5 years, range 4–17 years) were treated for acyclovir, topical steroids, chloramphenicol, and atropine acquired corneal perforations. There were three eye drops. A month later, she developed corneal ulcer females and one male (see Table 1). with hypopyon with a decrease in visual acuity to HM. The onset of all corneal perforations was acute after Corneal scrapings performed at this stage were positive progressive corneal melting. PKP was performed in for acanthamoeba. She was started on polyhexamethylene all four eyes while the eyes were still inflamed. All the biguanide (PHMB) and hexamidine eye drops. Two perforations were central. Two of four eyes showed an weeks later, the ulcer perforated needing an emergent aggressive fibrinous reaction at the time of the surgery, PKP (Figure 1a). Postoperatively, topical steroid and involving the lens and necessitating lensectomy. antibiotic combination drops along with PHMB drops Conservative measures such as bandage contact lens were started. She was evaluated twice a week in the in the (BCL) and tissue adhesive were used before PKP in the immediate post-operative period; and the drops were two non-infective cases. The two patients with infective tapered gradually to once a day over a period of 4 weeks keratitis were treated medically with appropriate and PHMB was continued for another 4 weeks. She was antimicrobial agents after microbiological workup. also continued on topical and systemic acyclovir until the The two patients with non-infectious etiology received confirmation of acanthamoeba in the corneal button, and Eye Table 2 Summary of clinical data and outcome of patients Case no. Initial presentation Initial management Surgery Management after PKP Complications Final outcome Medical Surgical Medical Surgical 1 Herpes keratitis, corneal epithelial defect Oral+Topical ACV, Tarsorrhaphy PKP Syt+topical steroids, ATR, PHMB, CE + IOL YAG 1. Graft rejection Clear graft hypopyon, ulcer, later diagnosed as PDF, CHL, ATR, Hexamidine, antiglaucoma tear capsulotomy 2. PSC Controlled IOP acanthamoeba keratitis, perforated ulcer PHMB, Cefuroxime supplements 3. Glaucoma 2a Corneal infiltrate with perforation, MGD, Topical EC, AMK, BCL, Histoacrylate glue PKP+CE LVF, ATP, ECZ, CIPRO, IV AMPH B, YAG PCO Clear graft corneal vascularization CF Syst+Topical steroids, FLCZ capsulotomy 3 Spontaneous corneal perforation 3 mm IV CF, CIPRO, Double
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