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 Table of Contents  
CASE REPORT
Year : 2023  |  Volume : 3  |  Issue : 1  |  Page : 3-5

An unusual presentation of vernal keratoconjunctivitis


1 Department of Ophthalmology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Pathology, Apollo Hospital, Bhubaneswar, Odisha, India
3 Department of Ophthalmology, PGI, Chandigarh, India

Date of Submission21-Aug-2022
Date of Acceptance12-Oct-2022
Date of Web Publication20-Jan-2023

Correspondence Address:
Bijnya B Panda
B-3,202, Shreekhetra Residency, Aiginia, Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2082_22

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  Abstract 


Vernal keratoconjunctivitis is diagnosed mainly on the basis of typical signs and symptoms of allergic conjunctivitis such as conjunctival papillae, limbal gelatinous thickening, peri-limbal Horner–Trantas dots, and shield ulcers. Limbal hyperplasia can be a rare finding in such patients, which mimics an ocular surface neoplasm and is difficult to diagnose clinically unless a histopathological examination is done. We report a case of a young boy with a longstanding history of allergic conjunctivitis refractory to topical corticosteroids who presented with a giant isolated limbal conjunctival nodule and was successfully managed without any recurrence.

Keywords: Amniotic membrane graft, limbal hyperplasia, vernal keratoconjunctivitis


How to cite this article:
Panda BB, Mohapatra N, Vishwanath S. An unusual presentation of vernal keratoconjunctivitis. Indian J Ophthalmol Case Rep 2023;3:3-5

How to cite this URL:
Panda BB, Mohapatra N, Vishwanath S. An unusual presentation of vernal keratoconjunctivitis. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Feb 6];3:3-5. Available from: https://www.ijoreports.in/text.asp?2023/3/1/3/368207



The incidence of vernal keratoconjunctivitis is higher in hot and humid climates of the Middle East, Japan, India, South America, West Africa, and the Mediterranean basin, probably due to the higher level of pollens and other allergens.[1],[2],[3],[4] Males are more commonly affected than females and the age group of presentation varies from 5 to 25 years. Medical management with topical antihistamines, mast cell stabilizers, and topical corticosteroids is the first line of management. Refractory cases may need topical immunomodulators and systemic immunotherapy. The presence of an isolated limbal conjunctival nodule in such patients can lead to a diagnostic and surgical dilemma.


  Case Report Top


A 14-year-old boy presented with chief complaints of a gradually increasing swelling over the lateral part of the left eye for 2 years duration. He was diagnosed elsewhere as a limbal dermoid and sent to us for further management. The mass was sessile, extending from a 2–5 O' clock area, immobile, pink in color with intrinsic vascularity along with a feeder vessel [Figure 1]a. His best corrected visual acuity was 6/6 in both eyes and intraocular pressures were within normal limits. He had a history of allergic eye disease with frequent redness and itching for quite a long time. He belonged to a medium socio-economic background without any family history of allergy or atopy. He had been previously treated with topical Pred Forte 1%, Loteprednol 0.5%, topical cyclosporine 0.1%, and tacrolimus 0.03% for the last 6 months elsewhere, without any significant improvement in the size of the limbal mass. The differential diagnoses were ocular surface squamous neoplasia, pseudo epitheliomatous hyperplasia, limbal dermoid, pyogenic granuloma, and ocular adnexal lymphoma. Because he had not shown any regression with previous corticosteroid and immunomodulator treatment, a wide excision biopsy of the mass was planned. Anterior segment optical coherence tomography was done to rule out any deep scleral invasion. The picture showed hyper-reflective conjunctival epithelium of superficial conjunctival mass at the temporal limbus without any scleral involvement [Figure 1]b. Peripheral blood smear and complete blood counts showed normal leucocyte morphology and number, thus ruling out leukemia/lymphoma. Routine blood investigations were carried out before the surgery. Wide excision of the mass was done followed by cryotherapy to the margins, and ocular surface reconstruction was performed with amniotic membrane grafting with glue. The mass was 8 × 6 × 4 mm in size, pink in color, and soft in consistency. Histopathology revealed lymphoid hyperplasia in the sub-epithelium comprising multiple follicles with germinal centers containing tangible body macrophages with apoptotic debris. Hyperplastic small caliber blood vessels were seen without any granulomatous lesion [Figure 1]d. The patient recovered well with a short course of topical corticosteroids postoperatively. He was seen at regular follow-ups every 3 months. At 1 year follow-up, there was no evidence of any recurrence [Figure 1]c.
Figure 1: (a) Clinical photo of the left eye showing a sessile conjunctival mass extending from 2-5 O'clock, pink in color with intrinsic vascularity along with a feeder vessel. (b). AS-OCT showing a thickened hyperreflective epithelium conjunctival mass at the temporal limbus without any scleral involvement. (c) Clinical photo of the left eye after 1 year showing healthy conjunctiva without any recurrence. (d) Histopathology of the lesion showing lymphoid hyperplasia in the subepithelium comprising multiple follicles with germinal centers containing tangible body macrophages and apoptotic debris

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  Discussion Top


Vernal keratoconjunctivitis is a chronic, recurrent, bilateral, seasonally exacerbated ocular surface inflammation affecting children and young adults. It is a chronic allergic inflammation of the ocular surface mediated mainly by Th2 lymphocytes, overexpression of mast cells, eosinophils, neutrophils, Th2-derived cytokines, chemokines, adhesion molecules, growth factors, fibroblasts, and lymphocytes.[1],[2],[3],[4] Family history of atopy and hereditary associations may play a role in etiology.

Diagnosis is easy and mainly based on the typical signs and symptoms of allergic conjunctivitis, yet the treatment is difficult in terms of controlling inflammation for a prolonged period as they tend to be recurrent and are cumbersome. Long-term sequelae of vernal keratoconjunctivitis may be sight-threatening when the cornea gets involved as in shield ulcers, keratoconus (due to constant rubbing of the ocular surface), and corneal scarring (due to vascularization). Sometimes, the limbal lesions may be confused with pseudoepitheliomatous hyperplasia (PEH), nodular episcleritis, and ocular surface squamous neoplasia (OSSN).[5],[6],[7] In a recent case series by Kaliki et al.,[7] out of nine patients with histologically proven pseudoepitheliomatous hyperplasia, 44% of cases had a previous history of vernal keratoconjunctivitis apart from previous surgical intervention (22%), idiopathic severe blepharitis (22%), and without any cause (11%). Anterior segment optical coherence tomography helps in differentiating limbal hyperplasia from ocular surface squamous neoplasia by the absence of a typical abrupt transition zone between normal and abnormal epithelium.[8] However, the confirmatory diagnosis depends on the histopathology examination findings.

Treatment options include topical corticosteroids, antihistamines, mast cell stabilizers, and oral corticosteroids. In refractory cases, topical immunomodulators such as cyclosporine and tacrolimus may be an effective treatment along with a supra-tarsal injection of triamcinolone or dexamethasone to the giant papillae. Very rarely, the limbal gelatinous thickening may progress to a giant nodule, which is refractory to both topical corticosteroids as well as immunomodulatory drugs. There are only four case reports described in the literature that had clinical presentations similar to our patient.[9],[10],[11],[12] All the cited cases were surgically excised, and ocular surface reconstruction was performed by autologous conjunctival grafting. But in our case, we tried a novel technique of additional cryotherapy to the margins of the residual conjunctiva and reconstruction by amniotic membrane grafting. We do not favor autologous conjunctival grafting as the conjunctiva is already prone to inflammation and can continue to get inflamed further. We found this technique useful as it did not produce any adverse effects and helped to prevent a recurrence. Therefore, we propose that the addition of cryotherapy (wherever a facility is available) to the margins can help in preventing recurrence in the long run.


  Conclusion Top


Vernal keratoconjunctivitis can rarely manifest as a conjunctival mass mimicking an ocular surface tumor. Wide surgical excision of the conjunctival mass along with cryotherapy to the margins and ocular surface reconstruction is successful in preventing recurrence. Avoidance of allergens and careful follow-up are crucial to avoid recurrence.

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.
Buckley RJ. Vernal keratoconjunctivitis. Int Ophthalmol Clin 1988;28:303-8.  Back to cited text no. 1
    
2.
Kumar S. Vernal keratoconjunctivitis: A major review. Acta Ophthalmologica 2009;87:133-47.  Back to cited text no. 2
    
3.
De Smedt S, Wildner G, Kestelyn P. Vernal keratoconjunctivitis: An update. Br J Ophthalmol 2013;97:9-14.  Back to cited text no. 3
    
4.
La Rosa M, Lionetti E, Reibaldi M, Russo A, Longo A, Leonardi S, et al. Allergic conjunctivitis: A comprehensive review of the literature. Ital J Pediatr 2013;39:18. doi: 10.1186/1824-7288-39-18.  Back to cited text no. 4
    
5.
Malhotra C, Jain AK, Thapa B. Limbal pseudoepitheliomatous hyperplasia mimicking ocular surface squamous neoplasia in palpebral vernal keratoconjunctivitis. Case Rep Ophthalmol Med 2013;2013:527230. doi: 10.1155/2013/527230.  Back to cited text no. 5
    
6.
Mohebbi M, Ameli K, Mafi M, Bashiri A, Mahbod M. Pseudoepitheliomatous hyperplasia as a limbal mass mimicking nodular episcleritis. Korean J Ophthalmol 2016;30:148-9.  Back to cited text no. 6
    
7.
Kaliki S, Maniar A, Jakati S, Mishra DK. Anterior segment optical coherence tomography features of pseudoepitheliomatous hyperplasia of the ocular surface: A study of 9 lesions. Int Ophthalmol 2021;41:113-9.  Back to cited text no. 7
    
8.
Ong SS, Vora GK, Gupta PK. Anterior segment imaging in ocular surface squamous neoplasia. J Ophthalmol 2016;2016:5435092. doi: 10.1155/2016/5435092.  Back to cited text no. 8
    
9.
Schwab IR, Schwab L, Cavender JC. Limbal vernal keratoconjunctivitis with a hypertrophic limbal mass lesion. Ann Ophthalmol 1987;19:79-80.  Back to cited text no. 9
    
10.
Sugandhi P, Arora R. Isolated large limbal nodule as the presenting feature of vernal keratoconjunctivitis. J Pediatr Ophthalmol Strabismus 2007;44:49-50.  Back to cited text no. 10
    
11.
Khurana D, Chandrasekharan A, Angula M, Mulay K. Solitary limbal nodule in a child: An unusual presentation in pediatric vernal keratoconjunctivitis. Indian J Ophthalmol Case Rep 2021;1:32-3.  Back to cited text no. 11
  [Full text]  
12.
Kobayashi A, Nagata A, Shirao Y, Kawasaki K, Ohta T, Amaya-Ohkura Y, et al. Surgical treatment of limbal vernal keratoconjunctivitis by resection of a limbal lesion. Jpn J Ophthalmol 2002;46:679-81.  Back to cited text no. 12
    


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