New Surgical Approach for Limbal Dermoids in Children: Simple Excision, Corneal Tattooing, and Sutureless Limboconjunctival Autograft

TECHNIQUES

Jinho Jeong, MD, PhD,* Young-Jin Song, MD,† Seung-Il Jung, MD,† and Ji-Won Kwon, MD, PhD†

is an elevated yellow or white round mass overlying both the Purpose: fi To evaluate the ef cacy of combined surgery of simple cornea and conjunctiva. The growth rate of these masses is excision, corneal tattooing, and a sutureless limboconjunctival very slow or even stationary.2,3 autograft in pediatric patients with grade 1 limbal dermoids. Anatomically, they have been classified into 3 grades. Methods: The grade 1 limbal dermoid is a superficial lesion ,5mmin We treated 4 eyes of 4 patients with combined surgery of 4–6 corneal tattooing and a sutureless limboconjunctival autograft with diameter and localized on the corneal limbus. For a small simple excision of limbal dermoids. Preoperative and postoperative asymptomatic grade 1 limbal dermoid, it is recommended that visual acuity, postoperative recovery time, cosmetic improvements, these children undergo close clinical observation with serial patient compliance, and complication rates were reviewed. examinations because even a simple excision may cause a wound scar and pseudopterygium.7 Parents may want Results: The mean follow-up period in this study was 27.3 6 2.8 surgical correction performed, whereas their children are (range, 24–30) months. Visual acuity was stable during the follow- growing and going to school. From a psychosocial standpoint, up period. Resolution of patient discomfort and wound epithelial- surgery could be considered because cosmetic improvement is ization was achieved in 1 week. Cosmetic outcomes improved in important for the whole family.8 Progressively increasing view of corneal opacity and conjunctival vascularization. All patients anisometric astigmatism is also another indication to prevent were satisfied with the cosmetic results with no complications. amblyopia secondary to severe astigmatism.7 Conclusions: The surgical combination of simple mass excision and Corneal tattooing and a sutureless limboconjunctival corneal tattooing proved to be a good alternative method, with autograft after simple excision of limbal dermoids can lead to good cosmetic results without significant complications, a satisfactory cosmetic outcome, better compliance, and early especially when a donor cornea is not available.9 However, postoperative recovery without complications for grade 1 limbal surgical complications, including epithelial defects and dermoids in pediatric patients. peripheral corneal vascularization, may occur with focal Key Words: limbal dermoid, corneal tattooing, limboconjunctival marginal limbal cell deficiency. Modified simple limbal autograft epithelial transplantation using a cryopreserved amniotic membrane proved effective for the treatment of limbal stem (Cornea 2015;34:720–723) cell deficiency.10 Lamellar keratoplasty is another surgical option, but the fibrin glue–assisted amniotic membrane transplantation method also showed several advantages over limbal dermoid is a congenital benign choristoma, which lamellar keratoplasty because it gives long-term ocular Acomprises tissues of ectodermal and mesodermal origins. surface cosmesis with better compliance and faster recovery, Choristomas make up approximately 3% of conjunctival and avoiding the risks associated with immune suppression.5,11 1 corneal tumors. It occurs most commonly in the inferotem- In this study, we report 4 cases of patients who poral quadrant as a solitary lesion but occasionally occurs as underwent combined surgeries, with simple excision, corneal multiple lesions. The typical morphology of a limbal dermoid tattooing, and a fibrin glue–assisted sutureless limboconjunctival autograft for limbal dermoids. We monitored the cosmetic outcomes of the corneal and conjunctival lesions, Received for publication December 23, 2014; revision received February 21, the rehabilitation period, and patient compliance. We also 2015; accepted February 24, 2015. Published online ahead of print April evaluated the efficacy and possible complications of this new 20, 2015. From the *Department of Ophthalmology, Jeju National University College of technique. Medicine, Jeju, Korea; and †Department of Ophthalmology, Seonam University College of Medicine, Myongji Hospital, Goyang, Korea. The authors have no funding or conflicts of interest to disclose. MATERIALS AND METHODS J. Jeong and Y.-J. Song contributed equally to this article. Reprints: Ji Won Kwon, MD, PhD, Department of Ophthalmology, Myongji Patients Hospital, Seonam University College of Medicine, 697-24, Hwajung- Dong, Deokyang-Gu, Goyang-Si, Gyeonggi-Do 412-270, Korea (e-mail: A retrospective chart review was conducted on 4 eyes [email protected]). of 4 pediatric patients who underwent corneal tattooing and Copyright © 2015 Wolters Kluwer Health, Inc. All rights reserved. sutureless limboconjunctival autografts after simple excision

720 | www.corneajrnl.com Cornea Volume 34, Number 6, June 2015

of limbal dermoids in our hospital. All patients were referred patient was satisfied with the cosmetic outcome, and there to our clinic for cosmetic improvement. was no postoperative complication (Fig. 1). This study was initiated after being approved by the appropriate institutional review board. All patients provided Case 2 written informed consent. This study adhered to the tenets of A 4-year-old female patient was referred because of the Declaration of Helsinki. a congenital limbal dermoid in the inferotemporal area. There was focal sectoral epithelial depigmentation of the ’ Surgical Methods iris at the 5 o clock position. Limbal dermoid excision, corneal tattooing, and a sutureless limboconjunctival auto- One surgeon (J.W.K.) performed all of the surgeries. graft from the superior conjunctiva, using fibrin glue, were All procedures were conducted under general anesthesia. The performed. The cosmetic outcome was satisfactory, and border of the limbal dermoid was marked at a shallow depth there was no postoperative complication, such as conjunc- using a #69 Beaver blade along the limbal margin. The tivalization of the limbus (Fig. 1). Recurrence did not occur corneal part of the dermoid was excised using a straight during the 30-month follow-up period. Beaver blade and Westcott scissors. A tissue-marking dye (Bradley Products Inc, Minneapolis, MN), which had been sterilized in an autoclave at 134°C for 6 minutes before surgery, was used for black coloring.12 With the bevel up, the RESULTS anterior stromal punctures were made using a 1-mL disposable The average age of the patients was 9.3 (range, 2–15) 6 syringe with a 30-gauge needle, creating a relatively long years. The mean follow-up period was 27.3 2.8 (range, – puncture canal. Because the corneal surface was obscured by 24 30) months. All patients had grade 1 limbal dermoids, and the dye after several punctures, the dye spread was confirmed the dermoid lesion did not occlude the visual axis. The mean by vigorous irrigation with balanced salt solution to visualize diameter of the limbal dermoids was 3.5 mm, and the location the staining. The conjunctiva was then dissected to expose the of the dermoid was mostly in the inferotemporal area (75%). dermoid. Shaving was performed on the conjunctival part of Mean preoperative corneal astigmatism was 1.3 (range, the dermoid using a straight Beaver blade. The superior 0.5–2.2) D. The preoperative BCVA was 20/20 in all cases. conjunctiva, including the limbus margin for the autograft, The postoperative BCVA remained unchanged during the was marked with gentian violet at the size of 4.0 (horizontal) · follow-up period. Resolution of patient discomfort and wound 3.0 mm (vertical). Lidocaine solution was injected into the epithelialization was achieved within 1 week. Cosmetic marked subconjunctival space. The limboconjunctiva was outcomes improved in view of corneal opacity and conjunc- carefully harvested, and the graft was immediately transferred tival vascularization. The sutureless limboconjunctival auto- onto the base sclera. Fibrin glue (Tisseel; Baxter Inc, graft increased compliance in these pediatric patients Deerfield, IL) was then applied under the conjunctival graft, postoperatively, and the wound showed no dehiscence. All fi giving firm adhesion between the sclera and the autografted patients and parents were satis ed with the cosmetic results. conjunctiva. The wounds were covered with therapeutic Neither dermoid recurrence nor limbal pannus formation contact lenses, and patients received levofloxacin (Cravit; occurred in any patient during the follow-up period. Santen Pharmaceutical Company, Osaka, Japan) and 1% prednisolone acetate (Pred Forte; Allergan, Irvine, CA) 4 times per day for 1 month. Applied therapeutic contact lenses were DISCUSSION removed at postoperative 1 week. Surgery for limbal dermoid removal has been variable The patients were followed up on postoperative day 7 from simple excision, lamellar graft, to simple excision with and at 1, 2, 6, 12, 18, and 24 months after surgery. corneal tattooing. The main sequelae of these operations were Ophthalmic examinations, including the best-corrected visual remaining corneal opacity and conjunctivalization as a result acuity (BCVA) tests, anterior segment photography, and slit- of limbal cell deficiency. To solve these problems, we lamp examinations, were performed. designed a combination of corneal tattooing and a limboconjunctival autograft using fibrin glue. Case 1 This method showed advantages in terms of shortening A 15-year-old female patient presented to the clinic for the operation time because conjunctival sutures could be cosmetic surgery regarding a congenital limbal dermoid in the skipped, with the aid of fibrin glue. The need for suture right eye. The size of the dermoid was 4.5 (horizontal) · 3.5 removal and the foreign body sensation from conjunctival (vertical) mm, which was located in the inferotemporal area. suture knots could be avoided by this surgery, which could be There were numerous conjunctival vessels over the limbal helpful as a surgical option for children with congenital dermoid, encroaching and obscuring the corneal margins, limbal dermoids. causing cosmetic concerns. Preoperative corneal astigmatism Harvesting of the limboconjunctival autograft was was 1.2 diopters (D). Limbal dermoid excision, corneal made including the limbus margin of the cornea, and we tattooing, and a sutureless limboconjunctival autograft from consider that this procedure has the benefit of preventing focal the superior conjunctiva, using fibrin glue, were performed. marginal limbal cell deficiency, by supplying limbal stem After 1 month, the BCVA was 1.0 in the right eye. Visual cells to the excised corneal limbal area. Thus, we could acuity was stable throughout the follow-up period. The prevent recurrence or conjunctivalization, which can result

FIGURE 1. Preoperative and postoperative anterior segment photo- graphs of patients with limbal dermoids. A, A 4.5- [horizontal (H)] · 3.5-mm [vertical (V)], slightly elevated round dermoid lesion with numerous conjunctival vessels encroaching the inferotemporal corneal limbal margin in the right eye (case 1). B, At postoperative 12 months, the limboconjunctival autografted area is free of abnormal conjunctival vessels, and the tattooing area is well stained and demarcated. C, A 3.5- (H) · 2.5-mm (V), ﬂat, round, opaque dermoid lesion at the 5 o’clock limbus in the left eye shows invasion at the corneal limbus (case 2). D, At postoperative 10 months, vision remains unchanged and the cornea remains well stained and demarcated. E, A 2.5- (H) · 3.5-mm (V), ﬂat, round, opaque dermoid lesion at the 2 o’clock limbus in the right eye shows invasion at the corneal limbus (case 3). F, At postoperative 1 month, the sharp and clear wound margin was well maintained. G, A 4.0- (H) · 4.0-mm (V), elevated, ovoid, well-vascularized colored dermoid lesion at the 9 o’clock limbus in the right eye shows invasion at the corneal limbus (case 4). H, At postoperative 12 months, vision remains unchanged and the cornea remains well stained and demarcated.

from limbal cell deficiency without any additional surgical psychosocial reasons, not just as a cosmetic issue.8,13 In our procedures, such as amniotic membrane transplantation. cases, not only the patients but also their families showed To our knowledge, this is the first report to apply these improved psychosocial behavior, reducing stress due to appear- methods to limbal dermoids. However, our study has some ances, especially in school-age children. Because all the patients limitations, including its retrospective design, the limited were Asians, corneal tattooing might blend in better with the number of patients, and the lack of a control group. Long- natural iris color. Even in non-Asians, the prevention of limbal term follow-up to assess recurrence or limbal cell deficiency deficiency and early recovery could be expected from the should be monitored especially in young patients. technique of a sutureless limboconjunctival graft using fibrin We mainly treated grade 1 limbal dermoids, which were glue. Larger dermoids, invading the cornea by .100 mmin relatively small and did not cause amblyopia. Traditionally, depth, should raise concerns about astigmatism control and small grade 1 limbal dermoids, not related to severe corneal amblyopia prevention, and additional surgical treatments astigmatism or amblyopia, were observed. However, it may be including lamellar keratoplasty and amniotic membrane trans- necessary to consider surgical reconstruction as an option for plantation may be considered.

In conclusion, combined corneal tattooing and a suture- 6. Stergiopoulos P, Link B, Naumann GOH, et al. Solid corneal dermoids less limboconjunctival autograft with simple excision in and subconjunctival lipodermoids: impact of differentiated surgical therapy on the functional long-term outcome. Cornea. 2009;28:644–651. patients with grade 1 limbal dermoids resulted in good 7. Pirouzian A. Management of pediatric corneal limbal dermoids. Clin cosmetic and functional outcomes. It can be a good choice Ophthalmol. 2013;7:607–614. for treatment if patients are selected appropriately. 8. Kaufman A, Medow N, Phillips R, et al. Treatment of epibulbar limbal dermoids. J Pediatr Ophthalmol Strabismus. 1999;36:136–140. 9. Cha DM, Shin KH, Kim KH, et al. Simple keratectomy and corneal tattooing for limbal dermoids: results of a 3-year study. Int J Ophthalmol. REFERENCES 2013;6:463–466. 1. Shields CL, Shields JA. Tumors of the conjunctiva and cornea. Surv 10. Amescua G, Atallah M, Nikpoor N, et al. Modified simple limbal Ophthalmol. 2004;49:3–24. epithelial transplantation using cryopreserved amniotic membrane for 2. Mansour AM, Barber JC, Reinecke RD, et al. Ocular choristomas. Surv unilateral limbal stem cell deficiency. Am J Ophthalmol. 2014;158: Ophthalmol. 1989;33:339–358. 469–475. 3. Mohan M, Mukherjee G, Panda A. Clinical evaluation and surgical 11. Choi SK, Lee D, Kim JH, et al. A novel technique: eccentric lamellar intervention of limbal dermoid. Indian J Ophthalmol. 1981;29:69–73. keratolimbal allografting using a femtosecond laser. Cornea. 2010;29: 4. Mader TH, Stulting D. Technique for the removal of limbal dermoids. 1062–1065. Cornea. 1998;17:66–67. 12. Kim C, Kim KH, Han YK, et al. Five-year results of corneal tattooing for 5. Pirouzian A, Holz H, Merrill K, et al. Surgical management of pediatric cosmetic repair in disfigured eyes. Cornea. 2011;30:1135–1139. limbal dermoids with sutureless amniotic membrane transplantation and 13. Watts P, Michaeli-Cohen A, Abdolell M, et al. Outcome of lamellar augmentation. J Pediatr Ophthalmol Strabismus. 2011;49:114–119. keratoplasty for limbal dermoids in children. J AAPOS. 2002;6:209–215.