© 2008 American College of Veterinary Ophthalmologists

Veterinary Ophthalmology

(2008)

11

, 3, 202–206

Blackwell Publishing Inc

CASE REPORT

Bilateral corneoconjunctival dermoids and nasal choristomas in a calf

Denise K. Brudenall,* Daniel A. Ward,† Larry A. Kerr‡ and Shelley J. Newman§

*

Animal Eye Services, Cnr Kessels Road and Springfield St, Macgregor, QLD 4109, Australia;

†

University of Tennessee, Department of Small Animal Clinical Sciences; and

‡

Department of Large Animal Clinical Sciences, C247 Veterinary Teaching Hospital, Knoxville, TN 37996-4544, USA;

§

University of Tennessee, Department of Pathobiology, College of Veterinary Medicine, Knoxville, TN 37996-4542, USA

Abstract

A 5-day-old Angus

×

Hereford calf presented for bilateral haired masses on the eyes and nasolabial planum since birth. The masses were surgically excised from the inferonasal cornea and bulbar conjunctiva of both eyes and the left and right side of the dorsomedial nasolabial planum. Histopathology of the excised tissue confirmed bilateral corneoconjunctival dermoids with ectopic lacrimal glands, and bilateral nasal choristomas and ectopic nasal glandular tissue. Surgery was curative and healing was uneventful. Bilateral ocular dermoids in combination with nasal choristomas and ectopic glandular tissue have not been documented previously in cattle.

Key Words:

bovine, corneoconjunctival dermoid, embryology, nasal choristoma

Address communications to:

Daniel A. Ward

Tel.: (865) 974-8387 Fax: (865) 974-5554 e-mail: dward@utk.edu

INTRODUCTION

Choristomas are benign congenital overgrowths of normaltissue in an abnormal location.

1

Dermoids are choristoma-tous malformations that often occur in the ocular region.

2

Dermoids may affect the eyelids, conjunctiva, nictitans andcornea

3

and most commonly present unilaterally. Bilateralocular dermoids have been reported in cattle,

4–14

humans,

15,16

sheep,

17

a dog

18

and a camel.

19

Ocular dermoids are rare incattle,

7,9–11

with the prevalence estimated between 0.002%and 0.4%.

14,20

This paper reports a case of bilateral corneo-conjunctival dermoids in a calf in conjunction with bilateralnasal choristomas.

CASE REPORT

History and initial clinical findings

A 5-day-old Angus

×

Hereford heifer calf was presentedwith an abnormal appearance of both eyes since birth. Ocularfindings were similar in both eyes. A large fleshy mass wasattached to the inferonasal cornea with a large number oftemporally and superiorly directed hairs arising from the surfaceof the mass (Fig. 1a). The calf exhibited moderate blepha-rospasm and watery discharge of both eyes. Superficial cornealulceration was present in the inferotemporal cornea of botheyes as a result of trichiasis. The nictitans of each eye appearedto be displaced anteriorly by its respective corneal mass, butthe inferior extent of the corneal masses could not be deter-mined without general anesthesia. A firm nodule was present

in the right nictitans, just ventral to the central leading margin.No other ocular abnormalities were detected in either eye. Abroad-based, haired skin mass was also present on the left andright dorsomedial aspect of the nasolabial planum, causingpartial obstruction of the nostrils (Fig. 1b). The clinical diag-nosis was bilateral corneal dermoids and nasal choristomas.

Surgical procedure

The calf was admitted to the University of Tennessee,College of Veterinary Medicine 5 days after initial examination.The calf weighed 50 kg, was in good bodily condition andno further abnormalities were detected on physical examina-tion, complete blood count and serum chemistries. Generalanesthesia was induced with an intravenous valium-ketaminecombination, the calf was intubated, and anesthesia main-tained with isoflurane and oxygen. The corneal dermoidsextended slightly beyond the inferonasal limbus and thenmerged with a second mass of lightly haired tissue within theinferonasal bulbar conjunctiva of both eyes. The nictitans ofboth eyes was poorly developed, the left nictitans beingparticularly rudimentary. The dermoids were excised bysuperficial lamellar keratectomy that was extended intothe inferonasal conjunctiva (Fig. 2). The nasal masses wereexcised by electrocautery. All excised tissue was submittedfor histopathology. Topical 1% atropine sulfate ointment(Atropine sulfate ophthalmic ointment USP 1%, Fougera &Co., Melville, NY, USA) was applied twice daily to both eyesfor 5 days and bacitracin-neomycin-polymixin ophthalmicointment (Trioptic-P, Altanta Inc., Melville, NY, USA) applied

© 2008 American College of Veterinary Ophthalmologists,

Veterinary Ophthalmology

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twice daily to both eyes for 2 weeks postoperatively. Ocularand nasal healing was uneventful, with prominent corneo-conjunctival granulation tissue 6 days postoperatively, whichlargely resolved by the 6-week follow-up examination.

Histopathologic examination

Histopathology was similar for the tissue excised fromboth eyes. The corneal lesion exhibited a moderately welldelineated but nonencapsulated raised mass comprised ofmoderately hyperplastic, keratinizing stratified squamousepithelium overlying a thick collagenous stroma, which mergedwith conjunctival tissue containing submucosal glandulartissue. The corneal mass contained numerous, large, well-developed hair follicles and adnexal structures superficially.

Figure 3. Corneoconjunctival dermoid. Within the deeper portions of the corneal dermoid, there is a parallel arrangement of abortive hair follicular development characterized by the presence of a hair bulb (arrow) and an associated apocrine gland (arrowhead). H&E; bar = 25 µm.

Figure 4. Nasal choristoma. Large regularly arranged hair follicles and adnexa, with deeper glandular tissue (asterix). H&E; bar = 100 µm.

Figure 1. Initial presentation. (a) Right eye. Note the long hairs protruding from the corneal dermoid and the nodular lesion of the central leading margin of the third eyelid. (b) Prominent broad-based, haired lesions on the nasolabial planum.

Figure 2. Corneoconjunctival dermoid of left eye at surgery. The corneal portion of the dermoid has been excised and reflected anteriorly; the conjunctival portion is still attached at the inferonasal aspect.

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A band of abortive hair follicles and adnexa was identified ashaphazardly arranged clusters of epithelium, in the absenceof hair bulbs, intermingled with tortuous lumina of apocrineglands beneath the productive follicles (Fig. 3). This wasaccompanied by myxomatous stroma and a minimal neutro-philic and eosinophilic inflammatory infiltrate. The sub-mucosal glandular tissue within the conjunctival portion ofthe mass exhibited abundant mucus production, as identifiedon periodic acid Schiff (PAS), mucicarmine and alcian blue2.5 staining, in addition to the presence of serous demilunes,thus making it consistent with ectopic lacrimal gland.

Histopathology of the excised nasal tissue was similar andcharacterized by moderately well-delineated but nonencap-sulated, raised masses. These were comprised of moderatelyhyperplastic, keratinizing stratified squamous epitheliumoverlying large regularly arranged hair follicles and adnexa(Fig. 4). Clusters of compound tubulo-alveolar glands show-ing moderate mitotic activity were located deep to the baseof the hair follicles. These glands, arranged as acinar structuresaround columnar lined ducts, exhibited features of mixedglands (serous and mucus producing glands, as confirmed byPAS, mucicarmine and alcian blue 2.5 staining) and wereconsidered to be consistent with nasal vestibular glands.Morphologic diagnosis was bilateral corneoconjunctivaldermoids with ectopic lacrimal glands, and bilateral nasalchoristomas with ectopic nasal vestibular glands.

DISCUSSION

The precise developmental mechanisms involved in thepathogenesis of ocular dermoids are not known.

7,21

Metaplasiaof mesenchyme (of primarily neural crest origin), resultingin abnormal differentiation of the surface ectoderm, isconsidered the most likely mechanism.

22

The resultingdermoid consists of ectodermal elements (keratinized epithe-lium, hairs, sebaceous and apocrine glands), and mesenchymalelements (fibrous tissue, fat and cartilage) combined indifferent proportions.

23

Ocular dermoids in cattle are not common, with anestimated prevalence of 0.002%–0.4%.

14,20

Ocular dermoidshave been reported in cattle of many breeds worldwide,with a similar low prevalence in all breeds other than theHereford.

7,24

The apparent predisposition in Herefords islargely based on a report by Barkyoumb and Leipold of 74affected Hereford and Hereford-cross calves from one regionof the USA, the majority of calves being bilaterally affected.The dermoid locations in that study were reported indecreasing order as limbus, third eyelid, canthus, eyelid andconjunctiva.

7

There are otherwise few reports of bilateralocular dermoids in calves, each describing single or lownumbers of animals,

4–6,8–14

and only one reporting a nasaltumor-like growth.

5

Of these bilateral cases, inferonasalcorneoconjunctival dermoids were most commonly reported,followed by nasal canthal dermoids.

4–6,8–14

Ocular dermoids may be associated with other congenitalocular or multiorgan abnormalities.

25

The medial and lateral

nasal processes, optic vesicle and first and second branchialarches are in close relationship on the lateral side of theembryonic head and might all be adversely influenced bythe same stimulus.

26

Ectopic lacrimal tissue may appear incombination with an ocular dermoid as in this case reportand previously reported in cattle,

7

or alone as an epibulbarchoristoma.

27

The ectopic lacrimal tissue in this calf mayhave represented misplacement of the nictitans gland oradditional lacrimal tissue development.

The primitive adnexal units situated beneath the productivehair follicles in the deeper connective tissue frequently failedto show luminal development, resulting in a band of abortivehair follicles that has not been previously described in der-moids. Also not previously reported in cattle was the prominentmitotic activity noted in the glandular tissue of the nasalchoristomas, suggesting ongoing proliferative activity. Choris-tomas are known to grow, with the rate accelerated by trauma,irritation or puberty, in humans.

1,25

Malignant degenerationis rare in ocular choristomas but transformation of ectopiclacrimal tissue into adenocarcinoma and pleomorphic adenomahas been reported in humans.

25

Goldenhar’s syndrome is a congenital disorder characterizedby epibulbar dermoids and/or lipodermoids, preauricularskin tags, and facial and vertebral abnormalities in humans.

28

Goldenhar’s syndrome is variable in expression both in later-ality and in the extent of ocular and adnexal involvement.

29

Cardiovascular and urogenital abnormalities have also beenreported in combination with Goldenhar’s syndrome.

25,28

Congenital heart disease has been reported in associationwith bilateral corneal and nasal canthal dermoid in a Guernseycalf.

4

Barkyoumb and Leipold described cardiac defects(Tetralogy of Fallot and patent ductus arteriosus), polycystickidney disease and small tissue masses protruding into theexternal nares in some of 74 calves reported with oculardermoids, although they did not specify the number of calvesaffected and whether calves showed one or a combinationof the three abnormalities.

7

Bhatt

et al

. reported a tumor-likegrowth at the entrance of the left nasal passage in associationwith bilateral corneal and eyelid dermoids in a Rathi calf.

5

Histopathologic examination was not performed on thenasal masses in either of these two reports.

Nasal skin tags have been reported in association withGoldenhar’s syndrome.

28,30

Ide

et al

. identified seromucinous-secreting glandular tissue within a nasal tag in a child,which they classified as an aberrant salivary gland from theoral cavity.

30

Ide

et al

. hypothesized that delayed closure ofthe palate during embryogenesis could have permitted upwardgrowth of the salivary gland from the oral cavity. The calf inour report showed multicellular, compound tubulo-alveolarglands within both nasal choristomas. In cattle the surfaceof the nasolabial planum is kept moist by secretions fromnasolabial and nasal vestibular glands.

31

Nasolabial glandsin cattle are tubular merocrine (sweat) glands, and nasalvestibular glands are mixed tubuloacinar glands.

31

The nasalchoristoma glands in this report, although similar in appear-ance to the conjunctival ectopic lacrimal tissue, exhibited

© 2008 American College of Veterinary Ophthalmologists,

Veterinary Ophthalmology

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, 202–206

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more abundant mucus production and were consistent withaberrant nasal vestibular glandular tissue.

The combination of congenital ocular and nasal abnor-malities in this calf is compatible with the intimate earlydevelopmental origin of the optic and nasal regions and acommon abnormality in neural crest migration;

28

whetherthis abnormality has a genetic basis or not is less clear.Characteristics of both autosomal recessive and polygenicinheritance were found by Barkyoumb and Leipold

7

in theiranalysis of Hereford cattle with ocular dermoids. Breedpredispositions for ocular dermoids are reported in Birmancats, Dachshund, Dalmatian, Dobermann, Golden Retriever,German shepherd and Saint Bernard dogs, and Quarter-horses.

32–35

Other than the autosomally dominant ringdermoid syndrome, which presents as bilateral cornealdermoids involving the limbus for 360 degrees with noother anomalies,

36

neither corneal dermoid nor Goldenhar’ssyndrome is considered to be genetic in humans.

28,37

Noocular or nasal abnormalities were detected in the Hereforddam or the Aberdeen Angus sire of the calf in this report.The bull was a member of a small, relatively closed herd of30 cattle, and the calf in this report was the third calf pro-duced by this dam and sire. No ocular or nasal abnormalitieswere present in the previous calves of this mating or in anyother calves on this farm.

ACKNOWLEDGMENTS

The authors wish to thank Anik Vasington for assistancewith preparation of the photomicrographs.

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