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2. medicine
3. surgery

CASE REPORT
A CASE REPORT ON GRADE II PEDIATRIC LIMBAL DERMOID
SURGICALLY TREATED BY SUPERFICIAL KERATECTOMY WITH
BUCCAL MUCOSAL GRAFTING
H. R. Padmini1, Basavaraj Zalaki2
HOW TO CITE THIS ARTICLE:
H. R. Padmini, Basavaraj Zalaki. “A Case Report on Grade II Pediatric Limbal Dermoid Surgically Treated By
Superficial Keratectomy with Buccal Mucosal Grafting”. Journal of Evidence based Medicine and Healthcare;
Volume 1, Issue 15, December 15, 2014; Page: 1965-1968.
ABSTRACT: Limbal dermoids are benign congenital tumors that contain choristomatous tissue.
They appear most frequently at the inferior temporal quadrant of the corneal limbus. Standard
medical treatment for grade I pediatric limbaldermoids (ie, with superficial corneal involvement) is
initially conservative. In stages II (i.e, affecting the full thickness of the cornea with/without
endothelial involvement) and III (i.e, involvement of entire cornea and anterior chamber), a
combination of excision and lamellar keratoplasty, amniotic membrane, limbal stem cell
transplantation, buccal mucosa grafting is advocated. This case report describes a new surgical
technique for removal of pediatric corneal-limbaldermoid and ocular surface reconstruction using
buccal mucosal graft in achieving good anatomical integrity.
KEYWORDS: Limbaldermoid, Superficial keratectomy, Buccal mucosal graft.
INTRODUCTION: Limbaldermoids are the most common episcleralchoristomas, i.e, congenital
overgrowth of normal tissues by collagenous connective tissue covered by epidermoid epithelium
in an abnormal location, and involving the globe in children. These lesions may present
unilaterally or bilaterally.1
Histopathology, incidence, and pathogenesis limbal dermoids may present as a single
lesion or as multiple lesions. They are marginally vascularized, smooth, whitish lesions generally
located in the inferotemporal limbus.2 They may contain a variety of histologically aberrant
tissues, including epidermal appendages, connective tissue, skin, fat, sweat gland, lacrimal gland,
muscle, teeth, cartilage, bone, vascular structures, and neurologic tissue, including the brain.(3,4)
Malignant degeneration is extremely rare. Epibulbarchoristomas are thought to arise from an
early embryological anomaly (occurring at 5–10 weeks’ gestation) resulting in metaplastic
transformation of the mesoblast between the rim of the optic nerve and surface ectoderm.5
Anatomically, epibulbardermoids have been classified into three grades.6 This form of grading
allows clinicians to take a more stepwise approach to the clinical and surgical management of
such lesions. Grade I limbaldermoids am superficial lesions measuring less than 5 mm and are
localized to the limbus (Figure 1). Such lesions may lead to development of anisometropic
amblyopia, with slow growth resulting in oblique astigmatism and flattening of the cornea
adjacent to the lesion. Grade II limbaldermoids are larger lesions covering most of the cornea
and extending deep into the stroma down to Descemet’s membrane without involving it. Grade
III limbaldermoids, the least common of all the presenting dermoids, are large lesions covering
the whole cornea and extending through the histological structures between the anterior surface
of the eyeball and the pigmented epithelium of the iris. Visual morbidity may result from
J of Evidence Based Med & Hlthcare, pISSN- 2349-2562, eISSN- 2349-2570/ Vol. 1/Issue 15/Dec 15, 2014
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CASE REPORT
encroachment of the lesion into the visual axis, development of astigmatism, or formation of a
lipid infiltration of the cornea, which obstructs the visual axis. Large limbal dermoids can be
cosmetically disfiguring. Limbaldermoids are present at birth but may not be recognized until the
first or second decade of life. They may also appear to enlarge as the body matures.
CASE REPORT: A 7-year-old girl child presented with growth on her right eye that had been there since birth and led to mild visual disturbance, sensation of foreign body, and cosmetic disfigurement. There was no family history of similar lesions. Examination revealed a solid,
brownish-yellow, round mass measuring 6x6x3mm. Having rough surface with partly keratinized epithelium and hair involving the inferotemporal limbus and one third of the cornea. No
associated regional or systemic abnormalities were found. Visual acuity was 6/12 (20/40) in
the right eye and 6/6 in the left eye. The findings on slit-lamp examination, fundoscopy, and ocular ultrasonography were within normal limits, and intraocular pressure was normal.
Gonioscopy shows no angle structure involvement. After obtaining informed consent, the girl
underwent superficial keratectomy and ocular surface reconstruction using buccal mucosal graft
under GA.
The graft was obtained from the inner side of lower lip. A 8x8mm area of buccal mucosa
was harvested. The buccal wound was closed with interrupted 4-0 Catgut sutures. The graft is
sutured to the corneal wound with 10-0 vycryl and ocular surface reconstructed. After instillation
of antibiotic eye drops, the eye was patched with a light pressure pad.
On the first post-operative day, examination showed the graft tissue well apposed with
the host tissue. Thus the patient was discharged with instructions to use antibiotic eye drops.
When seen in the clinic a month later, the graft was well integrated with the ocular tissue. The
graft-host junction was healthy, with epithelialization of the surface of the graft. There was no
evidence of necrosis or infection.
DISCUSSION: A variety of surgical techniques has been described in the literature, ranging from
simple excision to lamellar and/or penetrating keratoplasty with relaxing corneal incision,
depending on the grade of the lesion. Depth, size, and site of such lesions are critical factors.
Other techniques include corneal-limbal scleral donor graft transplantation and surgical resection
followed by reconstructive sutureless multilayered amniotic membrane transplantation.7-9
The rationale for buccal mucosa transplantation is to augment complete volumetric filling
of the defective area. Extensive corneal defects appear to show improved healing following buccal
mucosa transplantation and augmentation. More significant corneal defects have also been filled
and reconstructed using buccal mucosa.
Complications are usually minor. They include infection, central graft ulceration,
granulomas. Most complications can be avoided by employing the careful surgical techniques.
CONCLUSION: This surgical approach offers an alternative surgical technique to a simple
excision with or without deep lamellar keratoplasty for removal of pediatric corneallimbaldermoids (grade I&II). In the management of pediatric limbaldermoids (grade I), surgical
excision combined with buccal mucosa transplantation eliminates painful postoperative recovery
and corneal neovascularization, and can achieve an improved long-term ocular surface strength.
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BIBLIOGRAPHY:
1. American Academy of Ophthalmology. Basic and Clinical Science Course. 2012. Series 6.
Amercian Academy of Opthalmology.
2. Mansour AM, Barber JC, Reinecke RD, Wang FM. Ocular choristomas. SurvOphthalmol.
1989; 33: 339–358.
3. Duke-Elder S. System of Ophthalmology: Congenital and Developmental Anomalies. Vol 3.
1963: 488-495.
4. Yanoff M, Fine B. Ocular Pathology. 1982: 316-317.
5. Mann I. Developmental Abnormalities of the Eye. Cambridge, UK: Cambridge University
Press; 1937.
6. Mann I. In: Developmental Abnormalities of the Eye. 2nd ed. Mann I, editor. Philadelphia,
PA: Lippincott; 1957.
7. Zaidman GW, Johnson B, Brown SI. Corneal transplantation in an infant with corneal
dermoid. Am J Ophthalmol. 1982; 93: 78–83.
8. Panton RW, Sugar J. Excision of limbaldermoids. Ophthalmic Surg. 1991; 22: 85–89.
9. Kaufman A, Medow N, Phillips R, Zaidman GJ. Treatment of epibulbarlimbaldermoids. J
PediatrOphthalmol Strabismus. 1999; 36: 136–140.
Fig. 1: Limbaldermoid right eye
Fig. 3: Lateral view
Fig. 2: Complete view
Fig. 4: Graft Implantated
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CASE REPORT
Fig. 5: buccal mucosal graft harvesting
from posterior part of lower lid
AUTHORS:
1. H. R. Padmini
2. Basavaraj Zalaki
PARTICULARS OF CONTRIBUTORS:
1. Professor & HOD, Department of
Ophthalmology, Adichunchanagiri Institute
of Medical Sciences.
2. Post Graduate, Department of
Ophthalmology, Adichunchanagiri Institute
of Medical Sciences.
Fig. 6: Post-operative view
NAME ADDRESS EMAIL ID OF THE
CORRESPONDING AUTHOR:
Dr. H. R. Padmini,
Professor & HOD,
Department of Ophthalmololgy,
Adichunchanagiri Institute of Medical Sciences,
B. G. Nagar.
E-mail: [email protected]
Date
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Submission: 24/11/2014.
Peer Review: 25/11/2014.
Acceptance: 02/12/2014.
Publishing: 15/12/2014.
J of Evidence Based Med & Hlthcare, pISSN- 2349-2562, eISSN- 2349-2570/ Vol. 1/Issue 15/Dec 15, 2014
Page 1968