Graefe's Archive for Clinical and Experimental Ophthalmology

, Volume 243, Issue 6, pp 615–618

Primary basal cell carcinoma of the caruncle with seeding to the conjunctiva

Authors

  • Jens Østergaard
    • Eye Pathology InstituteUniversity of Copenhagen
  • Jannik Boberg-Ans
    • Department of OphthalmologyRAS
  • Jan Ulrik Prause
    • Eye Pathology InstituteUniversity of Copenhagen
    • Eye Pathology InstituteUniversity of Copenhagen
Case Report

DOI: 10.1007/s00417-004-1086-5

Cite this article as:
Østergaard, J., Boberg-Ans, J., Prause, J.U. et al. Graefe's Arch Clin Exp Ophthalmol (2005) 243: 615. doi:10.1007/s00417-004-1086-5

Abstract

Background

To report the clinical and histopathological characteristics of a patient with a primary basal cell carcinoma (BCC) of the caruncle with seeding of the tumour to the conjunctiva.

Methods

Surgical excision and histological examination.

Results

A 60-year-old female presented with a lesion of the caruncle. Clinical examination revealed a pale lobulated tumour without skin involvement. Computer tomography scans showed orbital invasion. The tumour was excised. Three years later a small polypoid tumour developed in the inferior fornix of the same eye. Two and a half years later, the patient developed an orbital recurrence.

Microscopically, both neoplasms were composed of infiltrative islands of basaloid tumour cells, scattered mitoses and peripheral palisading consistent with the diagnosis of BCC.

Conclusion

This case describes a primary BCC of the caruncle with seeding to the conjunctiva.

Introduction

Basal cell carcinoma (BCC) is the most common human malignancy and accounts for 90% of eyelid cancers [7]. Primary BCC of mucous membranes is extremely rare [7] and the majority of BCCs of the conjunctiva are due to invasion from the periocular skin [4]. Only two primary BCCs of the caruncle [8, 9] and four of the conjunctiva have been reported previously [13, 5].

This case report describes a patient who developed a primary BCC of the caruncle and later a conjunctival BCC in the same eye probably due to seeding from the caruncular lesion. Furthermore, the BCC of the caruncle showed orbital invasion.

Case report

A 60-year-old Caucasian woman presented with discomfort at the medial corner of the left eye. She had no history of previous neoplasms. Clinical examination showed a pale lobulated cyst-like swelling of the caruncle (Fig. 1a). The lesion measured 3×4 mm and there were no signs of skin involvement. Dilation of the medial conjunctival blood vessels was present. The lesion was excised; however, histology proved that the removal was not radical since the lesion extended deeper than expected. Histologically, the lesion was composed of infiltrative islands of basaloid tumour cells with peripheral palisading consistent with the diagnosis of BCC (Fig. 1b). Areas within the lesion had a metatypic appearance with diffusely arranged spindle-shaped cells. The tumour exhibited scattered mitotic activity. Computer tomography (CT)- and ultrasound scans were performed. The deep part of the tumour measuring 8–10 mm was found to extend into the orbit. The remaining tumour was subsequently excised as were parts of the canaliculi and conjunctiva using frozen-section control to ensure radical removal. The surgical margins did not show tumour tissue.
Fig. 1

a Clinical photography of the primary caruncular lesion of the left eye showing a pale lobulated tumour measuring 3×4 mm. b Histological survey of the caruncular tumour stained with haematoxylin–eosin (H&E, ×30). Infiltrative islands of tumour cells (asterisks) located beneath the conjunctival epithelium (arrow). c Micrograph of the conjunctival tumour found in the inferior fornix (H&E, ×280). The infiltrative islands are composed of basaloid tumour cells. Note the mitosis (arrow) and the peripheral palisading (arrowheads). d Computer tomography scan of the recurrent lesion (arrow) invading the left orbit

Three years later, the patient was referred again because a polypoid tumour measuring 3 mm and located in the middle of the inferior fornix of the left eye had appeared. The lesion was excised, and histology showed it to be composed of infiltrative islands of basaloid tumour cells with peripheral palisading (Fig. 1c), consistent with a second BCC. The tumour was excised and tumour-free surgical margins were verified by means of microscopy.

Five-and-a-half years after the initial treatment, the patient developed horizontal diplopia caused by restriction in lateral motility. Renewed CT scans showed orbital recurrence at the location of the removed orbital extension of the caruncular BCC (Fig. 1d). The tumour was excised by anterior orbitotomy. Histology revealed a recurrence of the BCC with basaloid tumour cells located around nerves and vessels. The patient was referred to undergo supplementary orbital radiation therapy but shortly after finishing this she died unexpectedly from other causes.

Discussion

BCC of the skin is overall the most common malignancy and is predominantly found in sun-exposed areas of elderly fair-skinned individuals [7]. About 90% of the eyelid cancers are BCCs, of which the majority is located on the lower eyelid and medial canthus [7]. BCC spreads by local invasion and, thus, BCC of the conjunctiva is most commonly found to originate from secondary invading BCCs [4]. In a recent series of 2,455 conjunctival lesions, six of six BCCs were found to be secondary to a skin lesion [4].

Only two illustrated case reports on primary caruncular BCCs have previously been published [8, 9]. One of these was in a 74-year-old Australian male with a history of sun exposure and multiple malignant neoplasms of the skin [9]. The lesion was a vascular multilobulated pink nodule [9]. The second case was that of a German 24-year-old male who had worked outdoors for the last 8 years as a road construction worker [8]. That lesion was a whitish nodule with a reddish centre and fine vessels surrounding the lesion [8]. In the present case, malignancy was not suspected since the lesion presented as a cyst-like swelling of the caruncle. At the time of presentation, there were no signs of skin involvement, which supports the diagnosis of a primary BCC of the caruncle. The history of the patient contained no information of excessive sun exposure.

Only four well-illustrated primary BCCs of the conjunctiva have been reported [13, 5]. Clinically, conjunctival BCC tends to mimic papillomas [5]. Of the previous reports, one was located at the nasal- and one at the temporal limbus, and both were described as nodular [3, 5]. The two other tumours were located in the palpebral aperture between the plica and limbus and were described as pedunculated [1, 2] (Table 1).
Table 1

Summary of reports on primary basal cell carcinoma of the caruncle and conjunctiva

Location

Age in years/gender

Symptoms (duration)

Clinical appearance

Treatment

Recurrence (follow-up)

Author (year)

Caruncle

74/Male

Growth of lesion (6 months)

Multilobulated nodule, vascularized, pink, 7×5 mm

Excision, 5-mm margins

Not stated (not stated)

Poon et al. (1997) [9]

Caruncle

24/Male

Growth of lesion (3 months)

Nodule, vascularized, white, red center, 3×5 mm

Excision

No (14 months)

Meier et al. (1998) [8]

Caruncle

60/Female

Growth of lesion, discomfort (weeks)

Lobulated, cyst like nodule, vascularized, pale, 3×4 mm

(1) Incomplete excision; (2) complete excision, cryosection control

Yes (51/2 years)

Present study

Conjunctiva-medial

82/Male

Growth of lesion (months)

Pedunculated nodule, fleshy 4 mm

Excision

No (2 months)

Aftab et al. (1973) [1]

Conjunctiva-medial

69/Female

Growth of lesion (6 months)

Pedunculated nodule, fleshy, lobulated surface, 2×11/2 in.

Excision

No (not stated)

Apte et al. (1975) [2]

Conjunctiva-limbal

66/Male

Growth of lesion (4 months)

Multilobulated nodule, fleshy, yellow-pink, 4×3×2 mm

Excision, 2-mm margins

No (1 year)

Husain et al. (1993) [5]

Conjunctiva-limbal

69/Male

Burning/stinging (weeks)

Nodule, 6×6 mm

Enucleation, intraocular invasion

No (8 years)

Cable et al. (2000) [3]

The present conjunctival BCC may either be a de novo lesion or a recurrence caused by seeding from the caruncular BCC. The tumour developed in the same eye, at a location not exposed to actinic rays and within 3 years after primary treatment, which is a reasonable time frame for the development of a recurrence. We thus consider the conjunctival BCC a seeding from the primary caruncular BCC. Moreover, patients who have developed a periocular BCC once have an increased risk of developing multifocal BCC [10].

The existence of primary BCCs of mucous membranes has been controversial [7], but in contrast to other mucous membranes the conjunctiva and the caruncle are exposed to actinic rays. Based on the microscopic appearance, BCCs could originate from basal cells of the epithelium or infundibular cells of the hair follicles [7]. Others believe that BCCs develop from pluripotent stem cells [7]. The fact that the caruncle is skin derived, originating from the lower eyelid, and harbours adnexal elements such as hair and sebaceous glands might explain why BCCs occasionally develop at this site.

Finally, this case was complicated by orbital invasion and recurrence. BCC is the third most frequent invading orbital malignancy (10%), only next to choroidal malignant melanoma (55%) and squamous cell carcinoma (15%) [6]. Orbital invasion by BCCs can be fatal due to intracranial extension [7]. Although BCC most often is restricted to lateral growth, the tumour cells in the orbit in this case exhibited a perineural and perivascular growth pattern which could have facilitated the tumour to traverse the subcaruncular fatty tissue. This case illustrates the importance of accurate clinical diagnosis and careful primary surgery handling a possible malignant pericaruncular lesion.

Copyright information

© Springer-Verlag 2004