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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 28-29

Shield ulcer following corneal collagen cross-linking


1 All India Institute of Medical Sciences, Gorakhpur, Uttar Pradesh, India
2 Department of Ophthalmology, Guru Nanak Eye Centre, Maulana Azad Medical College, New Delhi, India

Date of Submission05-Mar-2021
Date of Acceptance24-Jun-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Richa Agarwal
27-M, Kilkari Child Care, Cantt Chauraha, Daudpur, Gorakhpur - - 273 001, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_520_21

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  Abstract 


A 10-year-old boy presented with right-eye (RE) hyperemia, tearing, photophobia, and diminution of vision. There was a history of right-eye corneal collagen cross-linking (CXL) done 1 month back for keratoconus. Slit-lamp examination showed a corneal ulcer in the right eye. The upper tarsal conjunctiva showed hyperemia and multiple papillae in both eyes. He was diagnosed to be a case of bilateral keratoconus with vernal keratoconjunctivitis (VKC) and shield ulcer in the right eye. He was treated with topical steroids and 2% cyclosporine but the shield ulcer remained unchanged. The patient was then undertaken for surgical removal of plaque, after which, the ulcer re-epithelialized completely in 2 weeks.

Keywords: Collagen cross-linking, debridement, keratoconus, shield ulcer, vernal keratoconjunctivitis


How to cite this article:
Agarwal R, Jain P. Shield ulcer following corneal collagen cross-linking. Indian J Ophthalmol Case Rep 2022;2:28-9

How to cite this URL:
Agarwal R, Jain P. Shield ulcer following corneal collagen cross-linking. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Aug 14];2:28-9. Available from: https://www.ijoreports.in/text.asp?2022/2/1/28/334955



Vernal keratoconjunctivitis (VKC) is a chronic allergic condition, causing bilateral recurrent inflammation of the conjunctiva and cornea. It typically occurs in males before the age of 10 and usually resolves during puberty.[1] The cornea is often involved during recurrences of inflammation ranging from superficial keratitis to epithelial defect known as 'Shield ulcer' or 'Togby's ulcer.' Corneal ulcer, reported in 3–20% of VKC patients,[1],[2],[3],[4] may take months to re-epithelialize, especially where inflammatory debris is deposited in the base of the ulcer forming a plaque of fibrin and mucus.[2] Complications of delayed epithelial healing have been reported including secondary infections, amblyopia, and corneal perforation.[2],[3] Besides, after healing, corneal scarring may result in a permanent decrease in visual acuity.[2],[3],[4] Cameron et al.[2] proposed a classification of shield ulcers based on clinical features and response to treatment: Grade 1, corneal ulcer with clear base and margins, responds to medical treatment; Grade 2, corneal ulcer with inflammatory debris in the base, has a poor response to medical therapy alone and responds to scraping the base of the ulcer; Grade 3: corneal ulcer with elevated plaque, responds best to surgical therapy.

We report a case of vernal shield ulcer post-corneal collagen cross-linking (CXL) for keratoconus with coexisting VKC, who was treated with surgical debridement along with topical steroids and cyclosporine A.


  Case Report Top


A 10-year-old boy presented with right-eye (RE) hyperemia, tearing, photophobia, and diminution of vision for 2 weeks and was on topical lubricants and low-strength steroids (loteprednol etabonate). There was a history of RE corneal CXL done 1 month back for keratoconus. His best-corrected visual acuity (BCVA) was 20/200 in RE and 20/20 in the left eye. Slit-lamp examination showed a paracentral corneal ulcer in RE covered by inflammatory debris and a plaque of fibrin (2.7 mm × 4.8 mm), which was fluorescein-stain positive [Figure 1]a. On eversion of the upper lid, the upper tarsal conjunctiva showed hyperemia and multiple papillae in both eyes [Figure 2].
Figure 1: (a) Right eye with a shield ulcer covered by a plaque of fibrin. (b) Right eye after re.epithelialization of ulcer with a macular corneal opacity

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Figure 2: Superior tarsal plate showing papillary reaction

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He was diagnosed with a case of bilateral keratoconus with VKC and shield ulcer in RE. He was treated with topical steroids (1% prednisolone acetate) and 2% cyclosporine. After 5 days, the symptoms improved slightly, but the ulcer remained unchanged. He was then undertaken for surgical removal of plaque. After topical anesthesia, the plaque was removed with the tip of a 26-gauge needle and the ulcer base was scrapped under the microscope. The eye was patched for 24 h. The scrapings were sent for microbiological examination which were negative for Gram and KOH stain, and culture. The topical treatment was continued and a bandage with contact lens was put. Five days after the removal of plaque, a remarkable decrease in the size of the ulcer was noted. The ulcer healed and re-epithelialized completely in 2 weeks, leaving behind a macular corneal opacity, which stained negatively with the fluorescein dye [Figure 1]b. His BCVA improved to 20/40 in the RE and the symptoms were resolved. However, there were persistent papillae on the upper tarsal conjunctiva. Hence, treatment with topical steroids and cyclosporine was continued for uncontrolled VKC. The patient was followed up for 2 months, and the ulcer did not recur.


  Discussion Top


Many pediatric keratoconus patients suffer from ocular comorbidities such as allergy especially VKC.[5] VKC compounds the problems with keratoconus as continued surface inflammation and eye rubbing further accelerates keratoconus progression and may lead to advanced disease at a young age.[5]

CXL is a safe and effective intervention in children with and without VKC. Alrobaian et al.[6] evaluated the effect of VKC on clinical outcomes of CXL in 27 eyes of keratoconus. They found that the clinical outcomes of CXL in eyes with keratoconus and VKC are comparable to that in eyes with keratoconus alone. Both groups also had similar rates of corneal haze, which resolved 6 months after CXL. Only two patients with VKC exhibited exacerbations after CXL, whereas no patient had any VKC signs following CXL in the non-VKC group, alleviating concerns that CXL may stimulate ocular surface inflammation. This suggested that the final outcome does not differ after CXL in cases of keratoconus with or without ocular allergy.[6] However, a close follow-up is required in the postoperative period to detect epithelial breakdown and steroid-induced complications.

As per the information given by the parents, corroborated with records, this patient with VKC and keratoconus underwent CXL without adequately controlling the inflammation, resulting in the development of a non-healing corneal ulcer with plaque. This could be a complication of uncontrolled inflammation in VKC or could be delayed epithelial healing, a complication of CXL. Many eyes with VKC demonstrate partial limbal cell deficiency which may result in delayed epithelial healing after CXL.[7] Therefore, it is recommended that VKC should be controlled in these eyes before CXL and patients should be counseled to avoid eye rubbing. Besides, care should be taken to protect limbal stem cells during irradiation by maintaining centration during the procedure.[7]

The rationale for surgical debridement is to remove the inflammatory material in the base of the ulcer that is composed of proteins secreted from eosinophils and possesses cytotoxic properties hindering wound healing.[4] Similarly in our patient, the ulcer did not show any improvement despite topical steroids and cyclosporine A; however, it healed rapidly after debridement. In conclusion, shield ulcers are serious sight-threatening complications, with the risk of secondary infection and amblyopia considering the young age of patients with VKC. Aggressive management should be considered when there is corneal involvement, and surgical intervention should not be delayed in cases of non-healing ulcers, because simple scraping may yield dramatic improvement.


  Conclusion Top


There are reports of shield ulcer and its surgical treatment but no case of shield ulcer after CXL in a patient of keratoconus with coexisting VKC has been reported in the literature.

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 initials 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.



 
  References Top

1.
Bonini S, Bonini S, Lambiase A, Marchi S, Pasqualetti P, Zuccaro O, et al. Vernal keratoconjunctivitis revisited: A case series of 195 patients with long-term follow up. Ophthalmology 2000;107:1157-63.  Back to cited text no. 1
    
2.
Cameron JA. Shield ulcers and plaques of cornea in vernal keratoconjunctivitis. Ophthalmology 1995;102:985Y993.  Back to cited text no. 2
    
3.
Reddy JC, Basu S, Saboo US, Murthy SI, Vaddavalli PK, Sangwan VS. Management, clinical outcomes, and complications of shield ulcers in vernal keratoconjunctivitis. Am J Ophthalmol 2013;155:550-9.  Back to cited text no. 3
    
4.
Solomon A, Zamir E, Levartovsky S, Frucht-Pery J. Surgical management of corneal plaques in vernal keratoconjunctivitis: A clinicopathologic study. Cornea 2004;23:608-12.  Back to cited text no. 4
    
5.
Arora R, Gupta D, Goyal JL, Jain P. Results of corneal collagen cross-linking in pediatric patients. J Refract Surg 2012;28:759-62.  Back to cited text no. 5
    
6.
Alrobaian M, Elsayed M, Alotaibi AK, AlHarbi M, May W, Stone DU. Safety and efficacy of corneal cross-linking in pediatric patients with keratoconus and vernal keratoconjunctivitis. Middle East Afr J Ophthalmol 2019;26:95.  Back to cited text no. 6
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7.
Sangwan VS, Jain V, Vemuganti GK, Murthy SI. Vernal keratoconjunctivitis with limbal stem cell deficiency. Cornea 2011;30:491-6.  Back to cited text no. 7
    


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  [Figure 1], [Figure 2]



 

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