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 Table of Contents  
CASE REPORT
Year : 2023  |  Volume : 3  |  Issue : 1  |  Page : 145-147

A rare case of congenital ectopic eyelid tissue


1 Department of Orbit and Oculoplasty, Sadguru Netra Chikitsalaya, Chitrakoot, Madhya Pradesh, India
2 Department of Pathology, Central Laboratory, Indore, Madhya Pradesh, India

Date of Submission15-Jul-2022
Date of Acceptance31-Oct-2022
Date of Web Publication20-Jan-2023

Correspondence Address:
Narendra Patidar
Sadguru Netra Chikitsalya, Chitrakoot, Jankikund, Satna - 210 204, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_1731_22

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  Abstract 


Ectopic eyelid, an extremely rare entity, is a congenital disturbance in embryogenesis in which lid tissue is present in an abnormal position near the eyelid. We herein report a case of a six-year-old child with a soft mass near the lateral canthus of the left eye having cilia overlying it, with a similar family history in her father. A surgical excision biopsy was performed under general anesthesia. A histopathology report revealed stratified squamous epithelium with numerous dermal pilous units consisting of a central hair shaft and clusters of Moll's glands, suggesting eyelid tissue. Therefore, an ectopic eyelid tissue can be considered a differential diagnosis for any congenital, non-progressive, soft tissue mass near the eyelid with overlying cilia. However, histopathology is required to confirm the diagnosis.

Keywords: Cilia, ectopic, lid


How to cite this article:
Pal S, Mehta A, Agrawal PA, Patidar N, Kulsum F. A rare case of congenital ectopic eyelid tissue. Indian J Ophthalmol Case Rep 2023;3:145-7

How to cite this URL:
Pal S, Mehta A, Agrawal PA, Patidar N, Kulsum F. A rare case of congenital ectopic eyelid tissue. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Feb 1];3:145-7. Available from: https://www.ijoreports.in/text.asp?2023/3/1/145/368165



The congenital ectopic lid is a very rare condition with very few cases reported in the literature. The origin of this abnormally placed lid is not clear, but the previous theory of partial or complete substitution of the Meibomian glands was refuted and embryologic origin was suggested.[1],[2],[3]

Ectopic cilia are the congenital disturbance in the position of eyelashes, presenting with a tuft of hair in an abnormal position within the eyelid. There are various case reports of ectopic cilia reported in the literature, with diagnosis confirmed on gross appearance or histopathology.[1],[2]

Herein, we report a rare case of ectopic eyelid tissue with cilia near the lateral canthus. Similar family history is present in this case, and a diagnosis is made based on the gross appearance and histopathological report.

Permission by the parents to print identifiable photographs was obtained and archived. The case described in this report is compliant with the Declaration of Helsinki, and health insurance portability and accountability act regulations.


  Case Report Top


A six-year-old female—the first child of non-consanguineous parents—presented with a small painless, non-progressive mass, located near the lateral canthus of the left eye since birth. There was a similar history of a small mass with cilia near the lateral orbital rim of the left eye in the father since birth, with no apparent inconvenience [Figure 1]a.
Figure 1: (a) Image of the father's LE showed a small soft mass with overlying cilia. (b and c) Patient showing a small mass near the lateral canthus of the left eye; no bony fixity with overlying cilia on the surface. (d) Postoperative image of the patient

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There was no sign of any other congenital or facial anomaly. Systemic examination was clinically unremarkable. The best-corrected visual acuity (BCVA) was 6/6 in both eyes, intraocular pressure (IOP) was 19 mmHg in the right eye and 17 mmHg in the left eye, measured with a non-contact tonometer. A 4 × 4 mm non-tender, soft mass with multiple cilia present on its surface was attached to the lateral canthus with no fixity to the bone, and it was freely mobile [Figure 1]b and [Figure 1]c. There was no sign of inflammation and the overlying skin was normal in appearance. The extraocular movement was full in all gazes. Slit-lamp examination of the anterior segment was within normal limits, and dilated fundus with 90 D lens was clinically unremarkable. A provisional clinical diagnosis of an ectopic eyelid mass was made.

The patient underwent an elective excision biopsy of the mass; surgical exploration and removal of the mass were done and lateral canthus forming sutures were taken. Care was taken to remove a mass in toto to prevent a recurrence.

On gross examination, the specimen consisted of a skin-covered smooth, oval mass with overlying cilia along with surrounding conjunctiva. A histopathological examination revealed keratinized squamous epithelium with numerous dermal pilous units containing central hair shafts and also a cluster of sweat glands of Moll lined by apocrine epithelium found in the subcutaneous fat [Figure 2]. These findings were consistent with the diagnosis of eyelid tissue. The postoperative period was uneventful, without any evidence of recurrence [Figure 1]d.
Figure 2: (a) H and E-stained section (400×) shows squamous epithelium with associated hair follicle having hair shaft at the center (black arrow). (b) H and E-stained section (100×) shows numerous dermal pilous units with central hair shafts (black arrow) clusters of glands lined by apocrine epithelium, and gland of Moll (red arrow) in subcutaneous fat

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  Discussion Top


Normal embryology of the eyelid suggests that it develops from both secondary mesenchyme (mesoderm invaded by cranial neural crest cells) and surface ectoderm.[4],[5] Generally, the surface ectoderm gives rise to the conjunctiva, skin epithelium, that is, simple squamous epithelium called the periderm cells and underlying basal layer, hair follicles, gland of Zeis, gland of Moll, and Meibomian glands, while tarsal plate, levator muscle, orbicularis oculi, orbital septum, and tarsal muscle of Muller develop from the mesenchyme.[6] The process of eyelid fusion starts at the beginning of week 8 when there is a gap between the upper and lower eyelid folds. The flattened periderm cells undergo morphogenetical and proliferative change into rounded or cuboidal periderm cells. The leading edge of these proliferating cells makes contact with the advancing edge of the opposing periderm cell.[6] Only the periderm and epidermal layers are involved in eyelid fusion; eyelid mesenchyme remains unfused for the development of future adnexa of both eyelids.[6] However, during the 13th week, the eyelash Anlagen develops lateral outgrowths,[6] which differentiate into the holocrine sebaceous glands of Zeis and modified sweat glands of Moll.[6] Some studies have demonstrated that upper and lower eyelids first meet at the lateral end of palpebral fissure with fusion progressing medially in a zipper-like fashion.[6] With the available embryological evidence, it is likely suggestive that our patient had surface ectodermal derivative eyelid tissue near the lateral canthi.

Ectopic cilia have been reported in the literature, but limited literature on the ectopic lid has been found. The origin of abnormally placed cilia is not clear, but the previous theory of partial or complete substitution of the Meibomian gland has been refuted[1],[2] and an embryologic origin has been suggested.[3]

In 1985, Prasanna[7] reported a case of the congenital ectopic lid with a right oro-nasal-ocular cleft and no relevant family history. In 2011, Baghestani and Banihashemi[8] reported a case of ectopic cilia with positive family history in the paternal grandfather. Similarly, in our case, there was a mass near the lateral orbital rim in the father since birth. This suggests the possibility of an autosomal dominant pattern of inheritance.[8] Twitty[9] showed that neural crest cells possessed well-defined inherent potential properties so that such cells that were pre-destined to form a lower eyelid could well migrate abnormally to produce the features of an eyelid in an unexpected position.[9] Ectopic eyelid morphogenesis could be explained hypothetically on an embryological basis. Based on our literature search, we found that ectopic eyelid tissue without any congenital or facial anomaly is relatively rare and its presentation near the lateral canthus and association with a positive family history has been rarely reported.


  Conclusion Top


Ectopic eyelid, a rare congenital condition, is usually asymptomatic. It can present as a small, painless mass near the eyelid, with or without family history. Diagnosis can be confirmed by histopathological examination of the excised mass. General and systemic examinations are a must to rule out any congenital or facial anomaly.

Abbreviations: RE = Right eye; LE = Left eye; WNL = Within normal limits; H/P = Histopathology; IOP = Intraocular pressure; H and E = Hematoxylin and eosin.

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.
Dalgleish R. Ectopic cilia. Br J Ophthalmol 1966;50:592-4.  Back to cited text no. 1
    
2.
Owen RA. Ectopic cilia. Br J Ophthalmol 1968;52:280.  Back to cited text no. 2
    
3.
MacQuillan A, Hamilton S, Grobbelaar A. Angiosomes, clefts, and eyelashes. Plast Reconstr Surg 2004;113:1400-3.  Back to cited text no. 3
    
4.
Hayashi Y, Liu CY, Jester JJ, Hayashi M, Wang IJ, Funderburgh JL, et al. Excess biglycan causes eyelid malformation by perturbing muscle development and TGF-alpha signaling. Dev Biol 2005;277:222-34.  Back to cited text no. 4
    
5.
Barishak YR. Embryology of the eye and its adnexae. Dev Ophthalmol 1992;24:1-142.  Back to cited text no. 5
    
6.
Tawfik HA, Abdulhafez MH, Fouad YA, Dutton JJ. Embryologic and fetal development of the human eyelid. Ophthalmic Plast Reconstr Surg 2016;32:407-14.  Back to cited text no. 6
    
7.
Prasanna M. Congenital accessory ectopic eyelid: Case report. Br J Plast Surg 1985;38:429-31.  Back to cited text no. 7
    
8.
Baghestani S, Banihashemi SA. Ectopic cilia in a 14-year-old boy. Pediatr Dermatol 2011;28:55-6.  Back to cited text no. 8
    
9.
Twitty VC. The developmental analysis of specific pigment patterns. J Exp Zool 1945;100:141.  Back to cited text no. 9
    


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