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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 3  |  Page : 747-748

Ectopic cilia of the eyelid with ectopic lacrimal gland


Department of Ophthalmology, UCMS and GTB Hospital, Delhi, India

Date of Submission30-Jan-2022
Date of Acceptance09-Mar-2022
Date of Web Publication16-Jul-2022

Correspondence Address:
Dr. Janani Rajagopal
Department of Ophthalmology, UCMS and GTB Hospital, Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_306_22

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  Abstract 


Ectopic cilia of the eyelid is a rare congenital anomaly. Here we report a case of a 22-year-old patient with an extra tuft of eyelashes with watering since birth. Excision of the tuft and histopathology revealed a “pilosebaceous unit with ectopic lacrimal gland.” Ectopic cilia are caused by the replacement of meibomian glands with skin glands. Associated lacrimal gland tissue is a choristoma. The typical site of occurrence of ectopic cilia is mostly in the junction of the medial two-third and lateral one-third of the eyelid. Proper preoperative planning with complete surgical excision provides gratifying cosmetic results with no recurrence.

Keywords: Complex choristoma, ectopic cilia, ectopic lacrimal


How to cite this article:
Rajagopal J, Das GK. Ectopic cilia of the eyelid with ectopic lacrimal gland. Indian J Ophthalmol Case Rep 2022;2:747-8

How to cite this URL:
Rajagopal J, Das GK. Ectopic cilia of the eyelid with ectopic lacrimal gland. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Aug 11];2:747-8. Available from: https://www.ijoreports.in/text.asp?2022/2/3/747/351170



Congenital ectopic cilia of the eyelid have been reported in animals but are a rare occurrence in humans.[1] It can occur either as anterior type from the surface of eyelid skin or posterior type from the tarsal plate projecting into the conjunctival side.[1] Ectopic cilia have been reported as a separate entity and in association with certain other abnormalities such as nail patella syndrome,[2] orbital dermoid cyst,[3] and hypochromic naevus in the literature. The association of ectopic cilia along with ectopic lacrimal gland tissue at the same location has not been reported so far. We report the first such case in a 22-year-old man from India with ectopic cilia and lacrimal gland tissue together causing reflex tearing, cosmetic disfigurement, and discomfort.


  Case Report Top


A 22-year-old young male presented to the OPD with complaints of watering and an extra tuft of hair in his right upper lid since birth. On examination, a tuft of hair 8–10 in number and similar in appearance and direction as that of the upper lid lashes was observed 2 mm above the lash line at the junction of medial two-third and lateral one-third of the upper lid. Intermittent watery discharge from the base of the hair tuft was noticed in the absence of any surrounding orifices. The adjacent lid margin and underlying conjunctiva were found to be normal with no evidence of congestion, trichiasis, or distichiasis. General physical examination and systemic examination were within normal limits with no evidence of any syndromic features. Anterior and posterior segment ocular examination was normal with emmetropia. Both lacrimal drainage system was patent without any evidence of fistulous tract on syringing [Figure 1].
Figure 1: Showing ectopic tuft of hairs with watering at the base of the tuft in the right upper lid few millimeters away from the lid margin

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A clinical diagnosis of “ectopic cilia with ectopic lacrimal tissue” was made. Surgical excision of the eyelashes site was carried out by making a pentagon-shaped excision. No fistulous tract was observed intraoperatively. Lid repair was done in layers. Histopathological study revealed a “pilosebaceous unit with ectopic lacrimal gland” [Figure 2].
Figure 2: Histopathology- Black arrow points to the ectopic lacrimal gland in the excised specimen. A pilosebaceous unit can be seen – H and E stain; 40× and 400× magnification

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The patient is being regularly followed up. With good cosmetic result, the patient remains asymptomatic. No recurrence of watering or regrowth of eyelashes has been observed so far [Figure 3].
Figure 3: Postoperative photograph on day 20 showing good cosmetic result with no evidence of watering or recurrence at the surgical site

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  Discussion Top


Congenital ectopic eyelid cilia in humans is a rare congenital abnormality with only a few cases reported in the literature. Cordon et al.[4] reported a case of ectopic cilia with lacrimal gland tissue, managed with elective excision with no recurrence in follow-up. McCulley et al.[5] reported a case of ectopic cilia of the lid with abnormal tear secretion near the lateral canthus and hypothesized that watering is through a misdirected lacrimal ductule from the lacrimal gland into the eyelid. The secretion from the ectopic cilia in a case reported by Godar et al.[1] was found to be the byproduct of sebaceous and apocrine glands owing to the presence of glandular tissue and the absence of lacrimal glands or ducts.

The excised cilia site subjected to histopathology in our case revealed both hair follicles and the ectopic lacrimal gland that caused watering at that site. The embryologic origin of the ectopic lashes has been thought to be a developmental deformity of the upper lid  Meibomian gland More Detailss due to replacement with skin glands.[6] The hair bulbs of these ectopic cilia are at the level of dermis of the eyelid skin above the orbicularis muscle and tarsus and is due to the absence of mesodermal tissue but not due to the metaplastic meibomian glands.[7]

Ectopic cilia have been found to commonly occur at the same typical site in the upper lid. In a study by Nakra et al.[8] in three patients with ectopic cilia, all three patients developed ectopic cilia at the same site at the junction of medial two third and lateral one-third of the upper lid. This site is embryologically a watershed area of two angisomes: the superficial temporal artery and the termination of facial artery.[8] The anatomical location of Tessier Type 9 facial cleft at the same site suggests that ectopic cilia is also a congenital anomaly due to abnormal differentiation during embryogenesis.[9] The ectopic site of lacrimal gland tissue away from the usual anatomical area is an unusual occurrence of choristoma.[10] With proper preoperative planning, complete removal of the ectopic cilia and lacrimal gland tissue will provide good cosmetic result and prevent recurrence.

To our knowledge, this is the first reported case of ectopic cilia of the eyelid with ectopic lacrimal gland occurring together at the same location.


  Conclusion Top


Anomalous site of ectopic lashes should be examined for associated choristoma tissues for proper preoperative planning and management. This will help patients ameliorate the symptoms without future 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.
Godar MS, Shrestha R, Godar KC. Ectopic eyelid in a 15 years old girl: A rare presentation. J Lumbini Med Coll 2018;6:122-3.  Back to cited text no. 1
    
2.
Edmunds MR, Kipioti A, Colloby PS, Reuser TT. A case of ectopic cilia in nail-patella syndrome. Int Ophthalmol 2012;32:289–92.  Back to cited text no. 2
    
3.
Krahulík D, Karhanová M, Vaverka M, Brychtová S, Pospíšilová D. Ectopic cilia associated with an orbital dermoid cyst and sinus tract: Case report. J Neurosurg Pediatr 2015;16:203-6.  Back to cited text no. 3
    
4.
Cordon AJ, Patrinely JR, Knupp JA, Font RL. Complex choristoma of the eyelid containing ectopic cilia and lacrimal gland. Ophthalmology 1991;98:1547-50.  Back to cited text no. 4
    
5.
McCulley TJ, Yip CC, Kersten RC, Kulwin DR. An ectopic site of lacrimal gland secretion mimicking epiphora. Arch Ophthalmol 2002;120:1586–7.  Back to cited text no. 5
    
6.
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;24:433–5.  Back to cited text no. 6
    
7.
Jakobiec FA, Yoon MK. Histopathologic proof for the origin of ectopic cilia of the eyelid skin. Graefes Arch Clin Exp Ophthalmol 2013;251:985–8.  Back to cited text no. 7
    
8.
Nakra T, Blaydon SM, Durairaj VD, Shinder R. Congenital upper eyelid ectopic cilia. J Pediatr Ophthalmol Strabismus 2011;48:e16-8. doi: 10.3928/01913913-20110208-08.  Back to cited text no. 8
    
9.
MacQuillan A, Hamilton S, Grobbelaar A. Angiosomes, clefts, and eyelashes. Plast Reconstr Surg 2004;113:1400–3.  Back to cited text no. 9
    
10.
Alyahya GA, Bangsgaard R, Prause JU, Heegaard S. Occurrence of lacrimal gland tissue outside the lacrimal fossa: Comparison of clinical and histopathological findings. Acta Ophthalmol Scand 2005;83:100-3.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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