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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 3  |  Page : 466-468

Unusual presentation of conjunctivo-palpebral dermoid


1 Department of Cornea and External Disease, Disha Eye Hospitals, Kolkata, West Bengal, India
2 Department of Orbit and Oculoplasty, Disha Eye Hospitals, Kolkata, West Bengal, India

Date of Submission28-Oct-2020
Date of Acceptance26-Feb-2021
Date of Web Publication02-Jul-2021

Correspondence Address:
Dr. Nibedita Das
176, Madurdaha, Jeet Solitaire, 2nd Floor, Kolkata, West Bengal - 700107
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_3326_20

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  Abstract 


A 10-year-old girl child presented with a chronic history of redness and itching in her right eye. Slit-lamp examination revealed two small reddish flat masses with few caterpillar hair-like structures in the lower palpebral tarsal conjunctiva simulating ophthalmia nodosa. However, histopathology of the excised mass showed a lesion composed of stratified squamous epithelium with hair follicles and adnexal tissues embedded in fibro collagenous substance compatible with dermoid choristoma. Different types of choristoma in bulbar conjunctiva, eyelid, and orbit have been mentioned in earlier literature. Palpebral conjunctival dermoid choristoma has been rarely described previously and the current case is a unique presentation of that entity.

Keywords: Dermoid choristoma, epibulbar choristoma, ophthalmia nodosa, palpebral choristoma


How to cite this article:
Das N, Das J. Unusual presentation of conjunctivo-palpebral dermoid. Indian J Ophthalmol Case Rep 2021;1:466-8

How to cite this URL:
Das N, Das J. Unusual presentation of conjunctivo-palpebral dermoid. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Jul 28];1:466-8. Available from: https://www.ijoreports.in/text.asp?2021/1/3/466/320071



Choristomas are the most common type of epibulbar and orbital tumors in the pediatric age group.[1] Ocular choristoma usually affects the epibulbar region whereas palpebral location is uncommon. Palpebral conjunctival choristoma may mimic different pathologies. Ocular choristoma may contain dermal adnexal structures, adipose or fibro-fatty tissue, cartilaginous tissue, and osseous tissue one or in combinations with each other. Choristoma with dermal components is mostly reported as epibulbar dermoid. Typical dermoid cyst results from sequestration of surface epithelium during embryogenesis and is lined by a stratified keratinized epithelium. In contrast, conjunctival dermoid is believed to occur from embryogenic sequestration of the conjunctiva, so lined by nonkeratinized epithelium. Dermoid choristomas in palpebral conjunctiva are rare and also can present uniquely,[2] like in our case which masqueraded as ophthalmia nodosa. This case report adheres to the ethical principles outlined in the Declaration of Helsinki and proper informed consent was obtained from the patient for publication.


  Case Report Top


A 10-year-old Indian girl presented with complaints of itching, redness, and swelling in her right lower lid for 8 months. She has been treated elsewhere with topical loteprednol 0.5% and olopatadine 1% eye drops with minimal relief. Her corrected distant visual acuity was 20/20 in both the eyes with normal anterior and posterior segment findings. The eyelid margin and eyelashes of both sides were normal. On eversion of the right lower lid, two distinct subconjunctival, flat, flesh-colored, nontender, nodular masses (measuring 5 mm × 5 mm) were noticed [Figure 1]a, which were fixed to the underlying tarsus. Few broken caterpillar hair-like foreign bodies, which were embedded within the masses [Figure 1]b led to a provisional diagnosis of ophthalmia nodosa. Adjacent conjunctiva showed a mild follicular reaction.
Figure 1: External photograph of everted right lower lid showing two separate flat nontender hyperemic granulomatous masses under the conjunctiva (a) higher magnification by slit-lamp demonstrates embedded hair-like structure marked by a white circle around it (b)

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Removal of the suspected caterpillar hair and exploration of the mass was planned under general anesthesia. After anesthesia, a chalazion clamp was applied on the lower lid and everted. With higher magnification, many delicate eyelashes were seen as they are embedded within those nodules. A probable diagnosis of dermoid was made and excision biopsy of the mass was done, and the tissue was sent for histopathology examinations. A gritty sensation was felt superficially over the mass during scraping by the No. 15 blade and the lesion was shaved off by scalpel leaving the tarsus intact. Antibiotic eye ointment (chloramphenicol 1% w/w) was applied and the wound was kept open for healing by secondary intention. The patient was doing fine without any recurrence.

Histopathology examination revealed a solid mass of dense fibro collagenous tissue covered by stratified squamous epithelium containing numerous dermal appendage structures, including hair follicles, sebaceous glands, and sweat glands. The presence of goblet cells within the overlying stratified columnar conjunctival layer was also noted [Figure 2]. These clinical and histological findings confirmed the diagnosis of conjunctival dermoid choristoma No other tissues such as adipose, cartilaginous, osseous, or lacrimal were identified within the lesion.
Figure 2: Histopathologic photomicrographs of hematoxylin and eosin (H&E) stain demonstrates solid mass of dense fibro collagenous tissue covered by stratified squamous epithelial (solid arrow) surface containing numerous dermal appendage, including hair follicles (hollow arrow) and sebaceous glands (solid asterisks). The tarsal conjunctival lining formed by nonkeratinized stratified columnar epithelium (diamond) with interspaced goblet cells (hollow star)

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There were no focal ulceration and secondary histiocytic reaction, epitheloid, or giant cells around any linear refractile foreign bodies that indicate ophthalmia nodosa.


  Discussion Top


The term Choristoma (Greek choristos = separated, oma = tumor) means a benign congenital overgrowth of histologically normal mature tissue in an abnormal location and differs from another congenital overgrowth “hamartoma,” which is composed of cytologically disorganized normal mature tissue indigenous to the location. In literature, choristoma are mostly described in the head and neck region.[1] They are the most common type of epibulbar and orbital tumors found in the pediatric age group.[1] In a series of 302 cases, choristoma represented 10% to 33% of all pediatric conjunctival tumors.[3]

Ocular choristomas have four major histopathologic groups: dermoid/dermal, lipodermoid, single-tissue choristoma, and complex choristoma.[1] Dermoid has collagenous connective tissue, covered by squamous epithelium with associated skin appendages. Lipodermoids have adipose tissue with dermis-like connective tissue. Single-tissue choristomas consist of one type of ectodermal and mesodermal tissue (lacrimal or other glands, nerve, brain fat, cartilage, bone, and teeth). Complex choristomas are composed of tissues of different origins. Other choristomatous growths present in cystic forms are dermoid cysts and epidermoid cysts. Histology of our case revealed masses with tissue elements suggesting components of dermal tissue. Choristomas of cartilaginous, osseous, odontogenic, dermolipoma, phacomatous, and complex type.[1],[4],[5],[6],[7] have all been described in palpebral conjunctiva with several reports of involvement of the lower eyelid.[7] Ocular choristoma's most frequent site is epibulbar in the superotemporal quadrant near the superior and lateral rectus muscles. Palpebral locations are unusual and they are even rarer concerning the palpebral conjunctiva,[4],[5],[6],[7],[8] and they may present as either sessile or pedunculated mass. Most of the time clinical diagnosis of conjunctival palpebral dermoids is challenging either by allergic or infective conjunctivitis-like presentation or their unusual location.

Choristomas in the palpebral conjunctiva clinically resemble with chalazion, epithelial inclusion cysts, dermoid, dermolipoma, pyogenic granuloma, papilloma, and hamartomas. Li et al. described a complex choristoma presented as chalazion with pyogenic granuloma.[6] In our case, the initial diagnosis of ophthalmia nodosa was because of nodular appearance with central caterpillar hair-like structure and symptomatic relief by topical steroids. Recently, Saleh et al.[9] has presented a similar nodular appearance as our case but the diagnosis was type 3 ocular manifestation of caterpillar hair as classified by Cadera et al.[10]

Choristoma may have familial history and also coexist with coloboma, Goldenhar syndrome, or epidermal nevus syndromes, or other systemic abnormality, which would then require additional investigation.[1] We found no such history in this case.

Choristomas are mostly treated by surgical excision when symptomatic or causing cosmetic concern. Malignant transformation is not reported of choristoma.[11]


  Conclusion Top


It is suggested that dermoid choristoma should be included in the differential diagnosis of lower eyelid masses. The present case highlights an unusual presentation that mimicked ophthalmia nodosa.

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.
Mansour AM, Barber JC, Reinecke RD, Wang FM. Ocular choristomas. Surv Ophthalmol 1989;33:339-58.  Back to cited text no. 1
    
2.
Kim G, Mifflin MD, Mamalis N, Moshirfar M. Conjunctival eyelashes: A rare presentation of dermoid. J Ophthalmic Vis Res 2014;9:106-8.  Back to cited text no. 2
  [Full text]  
3.
Elsas FJ, Green WR. Epibulbar tumors in childhood. Am J Ophthalmol 1975;79:1001-7.  Back to cited text no. 3
    
4.
Ojha PR, Deshpande AH, Gargade CB, Nigam JS. Epipalpebral conjunctival chondroid choristoma: Interesting developmental anomaly presenting in an adult. Indian J Ophthalmol 2017;65:613-4.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Chan S, Stewart CM. Epibulbar osseous choristoma: A rare find in the superior conjunctival tarsal plate of the upper eyelid. Ophthalmic Plast Reconstr Surg 2020;36:e34-6.  Back to cited text no. 5
    
6.
Li J, Zeng J, Shinder R. Atypical presentation of a complex eyelid choristoma mimicking a chalazion with pyogenic granuloma. Ophthalmology 2016;123:1308.  Back to cited text no. 6
    
7.
Verb SP, Roarty JD, Black EH, Poulik J, Grynspan D. Phakomatous choristoma: A rare orbital tumor presenting as an eyelid mass with obstruction of the nasolacrimal duct. J AAPOS 2009;13:85-7.  Back to cited text no. 7
    
8.
Blenc AM, Gómez JA, Lee MW, Torres FX, Linder JS. Phakomatous choristoma: A case report and review of the literature. Am J Dermatopathol 2000;22:55-9.  Back to cited text no. 8
    
9.
Saleh S, Brownstein S, Kapasi M, O'Connor M, Blanco P. Ophthalmia nodosa secondary to caterpillar-hair-induced conjunctivitis in a child. Can J Ophthalmol 2020;55:e56-9.  Back to cited text no. 9
    
10.
Cadera W, Pachtman MA, Fountain JA, Ellis FD, Wilson FM 2nd. Ocular lesions caused by caterpillar hairs (ophthalmia nodosa). Can J Ophthalmol 1984;19:40-4.  Back to cited text no. 10
    
11.
Herdiana TR, Takahashi Y, Valencia MRP, Ana-Magadia MG, Ishikawa E, Kakizaki H. Epibulbar osseous choristoma within a dermolipoma: Case report and literature review. Orbit 2019;38:407-11.  Back to cited text no. 11
    


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