Journal of Pediatric Ophthalmology and Strabismus

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Short Subjects 

Congenital Upper Eyelid Ectopic Cilia

Tanuj Nakra, MD; Sean M. Blaydon, MD; Vikram D. Durairaj, MD; Roman Shinder, MD

Abstract

Ectopic eyelid cilia is an uncommon congenital anomaly typically occuring in otherwise healthy children. It is asymptomatic, but parents may wish surgical excision for cosmetic benefits. The authors describe three cases of ectopic upper eyelid cilia, surgical intervention, and follow-up results.

Abstract

Ectopic eyelid cilia is an uncommon congenital anomaly typically occuring in otherwise healthy children. It is asymptomatic, but parents may wish surgical excision for cosmetic benefits. The authors describe three cases of ectopic upper eyelid cilia, surgical intervention, and follow-up results.

From Texas Oculoplastic Consultants (TN, SMB, RS), Austin, Texas; University of Colorado Denver School of Medicine (VDD), Aurora, Colorado; and SUNY Downstate Medical Center (RS), New York, New York.

The authors have no financial or proprietary interest in the materials presented herein.

Address correspondence to Roman Shinder, MD, SUNY Downstate Medical Center, 541 Clarkson Avenue, E Building, 8th Floor, Suite C, Brooklyn, NY 11203. E-mail: shinder.roman@gmail.com

Received: November 19, 2010
Accepted: January 13, 2011
Posted Online: February 15, 2011

Introduction

There is a spectrum of congenital cilial anomalies including agenesis, row duplication, and ectopic placement. Choristomas of ectopic cilia aberrantly protruding through the eyelid skin represent a rare congenital anomaly, with only 13 reported cases in the literature.1–11 Affected children typically present with an asymptomatic cluster of ectopic cilia involving the lateral upper eyelid. We report a case series of three patients with ectopic upper eyelid cilia, two of whom were treated by surgical excision and achieved good cosmetic outcomes without recurrence. The third is awaiting surgery.

Case Reports

Case 1

An otherwise healthy 4-year-old boy was referred for an asymptomatic, congenital area of ectopic cilia arising from a papule involving the lateral left upper eyelid (Fig. 1A). There was no history of complicated pregnancy or delivery, or systemic dermatologic condition. There was no family history of similar lesions. The parents denied a history of growth. On examination, there was a tuft of densely grouped cilia between the eyelid margin and eyelid crease that did not threaten the ocular surface. The ectopic cilia were firmly attached to the underlying tarsal plate. Eyelid eversion demonstrated an unremarkable palpebral conjunctiva. Comparison of cilia from the ectopic tuft to those at the eyelash line did not show any gross morphological differences. Complete surgical excision was recommended and performed using a vertical ellipse incision to include the dermal hair follicles, and reconstruction included simple wound closure. Histopathologic examination of the surgical specimen showed normal-appearing cilia. The patient obtained a satisfactory cosmetic result, and had no recurrence at 5 months of follow-up (Fig. 1B).

(A) Case 1 at Presentation Displaying a Localized Tuft of Ectopic Cilia Involving the Lateral Left Upper Eyelid. (B) Appearance 5 Months After Surgical Excision and Reconstruction.

Figure 1. (A) Case 1 at Presentation Displaying a Localized Tuft of Ectopic Cilia Involving the Lateral Left Upper Eyelid. (B) Appearance 5 Months After Surgical Excision and Reconstruction.

Case 2

An otherwise healthy 8-month-old boy was referred for evaluation and treatment of presumed ectopic cilia of the left upper eyelid. The birth, medical, surgical, and family histories were noncontributory. On examination, a 3 × 3 mm area of dense ectopic cilia was present 3 mm above the left lateral eyelid margin. Eversion of the eyelid showed normal tarsal conjunctiva. A pentagonal wedge resection of the left upper eyelid was performed and histopathologic examination confirmed normal-appearing cilia. The patient obtained a satisfactory cosmetic result and there was no evidence of recurrence at the 4-month follow-up examination.

Case 3

An otherwise healthy 5-year-old boy was referred for an asymptomatic, congenital tuft of ectopic cilia involving the lateral left upper eyelid (Fig. 2). The medical and family history were noncontributory. His parents reported that the cilia had become clinically more apparent since birth. On examination, a 3 × 4 mm tuft of dense ectopic cilia located between the eyelid margin and crease were abutting the normal upper cilia. Complete surgical excision is currently scheduled.

Case 3 at Presentation Displaying a Localized Tuft of Ectopic Cilia Involving the Lateral Left Upper Eyelid.

Figure 2. Case 3 at Presentation Displaying a Localized Tuft of Ectopic Cilia Involving the Lateral Left Upper Eyelid.

Discussion

Cilia normally originate at the eyelid margin anterior to the gray line. The first reported case of ectopic cilia was described in 1936 by Weigmann in a patient who had aberrant cilia emanating from the palpebral conjunctiva.12 Since then, cases of ectopic eyelid cilia fall into two distinct categories: cilia originating in the anterior tarsal plate and protruding through the eyelid skin or cilia originating from the posterior tarsal plate and protruding either from the eyelid margin in the area of meibomian glands (distichiasis) or through the palpebral conjunctiva (Table).13–17

Ectopic cilia originating in the anterior tarsus is a rare clinical finding with only 13 prior reports in the literature.1–11 It is typically an isolated congenital finding in otherwise healthy children without a known family history of similar anomalies. However, cases have been reported in children with atopic dermatitis of the periocular skin7 and preseptal cellulitis.5 Additionally, Gordon et al. reported a complex choristoma containing ectopic cilia and functioning aberrant lacrimal gland tissue, intermittently producing tears.6 The ectopic cilia are uniformly found to be on the lateral quarter of the upper eyelid with a variable distance from the cilia of the eyelid margin. Histopathologic assessment demonstrates a close association between the ectopic cilia follicles with large apocrine sweat glands.10

The origin of ectopic cilia remains unknown. A prior theory that these cilia replaced and substituted meibomian glands15–17 has since been refuted by several reports observing normal meibomian glands in these cases.2,3,6 As proposed more recently by MacQuillen et al.8 and Chen et al.,9 the anomaly is likely due to inappropriate differentiation during embryogenesis, given its congenital presentation and its predilection for the lateral portion of the upper eyelid.

The preferred treatment of ectopic cilia protruding through the eyelid skin is surgical excision if it is cosmetically unacceptable. Recurrence in the form of partial cilial regrowth has been described, likely due to incomplete excision in two cases.5,10

We report three cases of ectopic cilia of the anterior tarsal plate, a rare congenital eyelid choristoma. They are most commonly isolated asymptomatic anomalies likely formed during embryogenesis. Complete surgical excision is the definitive treatment modality to achieve a good cosmetic outcome without recurrence.

References

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  2. Dalgleish R. Ectopic cilia. Br J Ophthalmol. 1966;50:592–594. doi:10.1136/bjo.50.10.592 [CrossRef]
  3. Owen RA. Ectopic cilia. Br J Ophthalmol. 1968;52:280. doi:10.1136/bjo.52.3.280 [CrossRef]
  4. Kant M, Dubey MK. Ectopic cilia: a rare anomaly. Med J Zambia. 1982;16:87.
  5. Riffle JE. Ectopic cilia and preseptal orbital cellulitis. Am J Ophthalmol. 1984;98:119–120. doi:10.1016/0002-9394(84)90204-6 [CrossRef]
  6. Gordon AJ, Patrinely JR, Knupp JA, Font RL. Complex choristoma of the eyelid containing ectopic cilia and lacrimal gland. Ophthalmology. 1991;98:1547–1550.
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  9. Chen TS, Mathes EF, Gilliam AE. ‘’Ectopic eyelashes’’ (ectopic cilia) in a 2-year-old girl: brief report and discussion of possible embryologic origin. Pediatr Dermatol. 2007;24:433–435. doi:10.1111/j.1525-1470.2007.00473.x [CrossRef]
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  11. Chappell MC, Spencer W, Day SH, Silkiss RZ. Congenital ectopic cilia of the upper eyelid. Ophthal Plast Reconstr Surg. 2010Aug23. [Epub ahead of print] doi:10.1097/IOP.0b013e3181e17501 [CrossRef]
  12. Weigmann E. Eine Zilie an einem ungewöhnlichen Platz, im Knorpel des Oberlides. Klin Monatsbl Augenheilkd. 1936;96:230–231.
  13. Jain SC, Saini VK. Bilateral ectopic cilia (a case report). Indian J Ophthalmol. 1985;3367–3368.
  14. Guler M, Yilmaz T, Guler O. A case of ectopic cilia. Int Ophthalmol. 2009;29:297–298. doi:10.1007/s10792-008-9228-0 [CrossRef]
  15. Bader A. Aplasia congenitalis glandularum Meibomi palpebrae inferioris. Graefes Arch Ophthalmol. 1950;150:411–413. doi:10.1007/BF00681437 [CrossRef]
  16. Tavolara L. Su un caso di ciglia insorte in sede anmomala in paziente affetta da distichiasi congenital. Boll Ocul. 1959;38:194–201.
  17. Fox SA. Distichiasis. Am J Ophthalmol. 1962;53:14–18.
Authors

From Texas Oculoplastic Consultants (TN, SMB, RS), Austin, Texas; University of Colorado Denver School of Medicine (VDD), Aurora, Colorado; and SUNY Downstate Medical Center (RS), New York, New York.

The authors have no financial or proprietary interest in the materials presented herein.

Address correspondence to Roman Shinder, MD, SUNY Downstate Medical Center, 541 Clarkson Avenue, E Building, 8th Floor, Suite C, Brooklyn, NY 11203. E-mail: shinder.roman@gmail.com

10.3928/01913913-20110208-08

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