Bilateral pseudopterygia following pediatric cataract surgery: A case report

Hennaav K Dhillon; Sumita Agarkar; Rama Rajagopal; S Krishna Kumar

doi:10.4103/ijo.IJO_2114_22

CASE REPORT

Year : 2023 | Volume : 3 | Issue : 2 | Page : 263-265

Bilateral pseudopterygia following pediatric cataract surgery: A case report

Hennaav K Dhillon¹, Sumita Agarkar¹, Rama Rajagopal², S Krishna Kumar³
¹ Department of Pediatric Ophthalmology and Strabismus, Sankara Nethralaya, Chennai, Tamil Nadu, India
² Cornea and Refractive Surgery, Sankara Nethralaya, Chennai, Tamil Nadu, India
³ Larson and Toubro Department of Ocular Pathology, Vision Research Foundation, Sankara Nethralaya, Chennai, Tamil Nadu, India

Date of Submission	13-Sep-2022
Date of Acceptance	08-Feb-2023
Date of Web Publication	28-Apr-2023

Correspondence Address:
Sumita Agarkar
Sankara Nethralaya, Medical Research Foundation, 18, College Road, Chennai - 600 006, Tamil Nadu
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_2114_22

Abstract

Pseudopterygia are the advancement of the conjunctiva onto the corneal surface in the setting of active or old ocular surface inflammation. Pterygia and pseudopterygia are exceptional conditions in children.^[1] We report a case of a 3-year-old child with a visually significant bilateral double-headed pseudopterygium, which developed following pediatric cataract surgery. The child was managed with pseudopterygium excision with conjunctival autograft and amniotic membrane grafting followed by visual rehabilitation.

Keywords: 10-0 Vicryl, congenital cataract, pseudopterygium

How to cite this article:
Dhillon HK, Agarkar S, Rajagopal R, Kumar S K. Bilateral pseudopterygia following pediatric cataract surgery: A case report. Indian J Ophthalmol Case Rep 2023;3:263-5

How to cite this URL:
Dhillon HK, Agarkar S, Rajagopal R, Kumar S K. Bilateral pseudopterygia following pediatric cataract surgery: A case report. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 1];3:263-5. Available from: https://www.ijoreports.in/text.asp?2023/3/2/263/374935

Pterygia and pseudopterygia are exceptional clinical presentations in children and represent several forms of benign and malignant ocular surface disorders that may be potentially amblyogenic.^[1] The differential diagnosis of a pseudopterygium includes primary and secondary keloids, limbal dermoids, keratitis, focal limbal stem cell deficiency, neoplasm, and conditions with chronic epithelial damage such as chronic contact lens wear.^[2],[3],[4]

Case Report

A 4-month-old male infant presented with white reflex in both eyes since birth associated with shaky eyes. Antenatal and perinatal history was unremarkable. There was no family history of congenital cataract.

On examination, the child appeared visually inattentive but a blink response to light was present. There was uniplanar horizontal jerk nystagmus. Anterior segment examination showed bilateral total cataract. The rest of the anterior segment exam was unremarkable. There was no view of the posterior segment. Ultrasonography of both eyes was within normal limits. He was advised lens aspiration with additional procedures in both eyes. Preoperative evaluation under anesthesia (EUA) revealed smaller corneas at 9.5 mm, and intraocular pressure (IOP) was 6 mmHg by Perkins tonometer. The axial length was 17.59 mm and 17.84 mm in the right and left eye, respectively. Both eyes showed open angles with few visible iris processes on the gonioscopy. The child underwent uneventful lens aspiration with primary posterior capsulotomy with anterior vitrectomy via limbal approach in both eyes. The limbal wound was closed using a single 10-0 vicryl suture. Postoperative period was uneventful. He was prescribed aphakic spectacle correction (+24.50 DSph in both eyes). At 2-month follow-up his visual axis was clear, fixation was central, unsteady, and maintained, the eye was quiet and sutures had been absorbed. The child was under regular follow-up and continued to maintain good fixation with glasses in either eye. At 1-year follow-up post cataract surgery, early pseduopterygia was seen in both eyes nasally as well as temporally at the site of previous limbal incisions. It was located close to the sites of the limbal wound. The visual axis remained clear. There was no history of recent trauma or excessive ultraviolet rays exposure. An EUA was performed. In the right eye, the pseudopterygium measured 1.5 mm × 2 mm nasally and temporally, whereas in the left eye, it measured 2 mm × 3 mm both nasally and temporally. The IOP was 30 mmHg and 28 mmHg in the right and left eyes, respectively. Child was started on antiglaucoma medication. He again came a year later for a review and by this time the pseudopterygium had progressed in both eyes. The visual axis in the left eye was obscured while it was partially obscuring visual axis in the right eye. In the right eye, the nasal pseduopterygia measured 4.5 mm × 4 mm and temporally 4 mm × 4 mm, whereas, in the left eye, the nasal pseudopterygia measured 6.3 mm × 5 mm and temporally 6 mm × 3 mm [Figure 1]. Ultrasound biomicroscopy (UBM) was done and showed a thick hyper-reflective membrane at 3 and 9 'o' clock positions involving the peripheral cornea with no involvement of the anterior chamber in both eyes. Anterior segment optical coherence tomography (AS-OCT) confirmed the clinical findings. The child underwent pseudopterygium excision with conjunctival autograft and amniotic membrane grafting with fibrin glue in the left eye [Figure 2]a. The excised tissue was sent for histopathology. Hematoxylin and eosin (H&E) staining showed minimal goblet cells in the conjunctival epithelium and minimal elastotic degeneration in the conjunctival stroma suggestive of actinic damage associated with inflammatory cells. There was an invasion of the Bowman's membrane and the corneal stroma. There was no evidence of dysplasia. Histopathologically, it was suggestive of a pseudopterygium [Figure 3]. A bandage contact lens (BCL) was placed and the child was prescribed a course of topical steroids, lubricants, and antibiotics. The BCL was removed subsequently after the surface re-epithelialisation. The patient was seen 6 weeks later and his visual acuity had improved to 6/19 in the right eye and 6/30 in the left eye with Lea symbols using +18.50 DSph in both eyes. Cornea was clear except for scarring in the periphery, visual axis was clear in the left eye [Figure 2]b. Intraocular pressure was recorded as 12 mmHg in both eyes. Fundus evaluation showed a healthy disc with an attached retina. He was advised to continue antiglaucoma medication and glasses with part-time occlusion of the right eye to correct amblyopia. Right-eye pseudopterygium removal was also advised.

Figure 1: (a and b) The clinical image of the right and the left eye showing a double-headed pterygium

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Figure 2: (a) Preoperative clinical picture of the left eye after pterygium excision and amniotic membrane grafting. (b) Six-week postoperative clinical picture of the left eye showing a clear visual axis and minimal scarring in the periphery

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Figure 3: (a) Histopathological microphotograph showing metaplastic and hyperplastic stratified squamous epithelium with goblet cell loss. (b) Histopathological microphotograph showing elastotic degeneration with infiltration into the corneal stroma and disruption of the Bowman's membrane. (c) Histopathological microphotograph showing elastotic degeneration, fibroblastic proliferation, and mild lymphomononuclear cell infiltrate

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Discussion

Pterygium is a benign, wedge-shaped fibrovascular growth of the degenerated bulbar conjunctiva that progressively encroaches on the cornea.^[1],[4],[5] It has usually been associated with chronic ultraviolet light exposure and frequently presents on the nasal side.^[1] In contrast, a pseudopterygium occurs as a result of an inflammatory response (e.g., post chemical injury, trauma, ulcerations, etc.) and can arise from anywhere on the bulbar conjunctiva.^[1]

Pterygia and pseudopterygia are usually observed and managed symptomatically with topical lubricants.^[4],[5] Surgical excision is indicated if it induces visually significant amounts of astigmatism, encroaches upon the visual axis, or occasionally for cosmetic reasons.^[5],[6],[7] In younger patients, surgery for pseudopterygia is preferred due to the likely progressive nature and increased inflammatory response in children.

Our case is uniquely associated with a bilateral double-headed pseudopterygia in a young infant post bilateral cataract surgery. The location of pterygium was at the sites of limbal corneal incisions made during cataract surgery. Limbal entries were sutured using a 10-0 vicryl. Although the safety and efficacy of 10-0 vicryl suture for the closure of wounds in cases of cataract surgery in children have been well established and it is routinely used, we believe that suture was contributory to pterygium in this case perhaps due to an exaggerated inflammatory response to the suture material used.^[8],[9],[10]

Other possible differentials in the particular case would include congenital pterygium, corneal keloids, or malignancy such as ocular squamous cell neoplasia (OSSN). Since these growths were not present since birth at the time of initial presentation, the diagnosis of a congenital pterygium was ruled out.

The diagnosis of a corneal keloid and OSSN was ruled out on the basis of histopathology. The HPE of a corneal keloid typically shows hyperplastic epithelium with irregular acanthosis with mild hyperkeratosis and edematous disorganization of the basal cell layer. Corneal keloids usually stain positive with Vimetin on immunohistochemistry.^[11] The histopathological diagnosis of an OSSN was ruled out by the absence of dysplastic cells.^[12]

On a literature review, we found one case of a bilateral double-headed recurrent pterygium in an 11-year-old child who had undergone pterygium excision with a conjunctival limbal autograft for primary pterygium.^[6] Authors have postulated that the double-headed pterygium originated from sites where there was limbal stem cell deficiency as evident from areas of absent palisades of Vogt.^[6] However, our patient had no overt limbal stem cell deficiency.

A few systemic associations of pseudopterygium such as chronic autoimmune conditions, especially affecting the mucous membranes such as pemphigoid. A few other systemic factors predisposing to pseudopteyrgium are actinic prurigo (hypersensitivity reaction as a result of prolonged exposure to direct sunlight), acne rosacea (chronic inflammatory condition), and certain atopic dermatitis.^[13] However, no distinct clinical features of either condition were seen in the present case.

We believe that the surgical trauma along with an exaggerated inflammatory response from the vicryl sutures could be a possible mechanism in this case.

Conclusion

While pseudopterygium is a benign entity, it may cause visual impairment by either directly encroaching on the visual axis or by inducing a significant amount of astigmatisms. Both these mechanisms are potentially amblyogenic hence pseudopterygium leading to any one of these must be surgically excised.

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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