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Br J Ophthalmol: first published as 10.1136/bjo.70.11.864 on 1 November 1986. Downloaded from

British Journal of Ophthalmology, 1986, 70, 864-866

Adenosquamous carcinoma of the : a case report

A G E NYLANDER AND H R ATTA From the Eye, Ear and Throat Hospital, Shrewsbury

SUMMARY A 76-year-old Caucasian woman presented with a caruncle tumour, histologically diagnosed as mixed adenosquamous carcinoma. Complete excision was performed, but this necessitated removing parts of the . No local recurrence or distant metastasis was found after six months' follow-up, and spontaneous recanalisation of both canaliculi occurred. This tumour is believed to be a primary adenosquamous carcinoma of the caruncle and possibly the first reported case. Long term follow up is planned to detect late metastasis, but complete excision of the tumour and its favourable site should yield a good prognosis.

A 76-year-old Caucasian woman presented with a Complete excision of the swelling was performed painless swelling in the inner corner of her right eye. two weeks later. This necessitated the excision of This had been slowly growing over a period of several parts of both lacrimal canaliculi, but no reparative copyright. months. On examination, a firm round swelling with surgery was performed at that time. Postoperative an ulcerated top was present in the area of the right recovery was uneventful, with good cosmetic results caruncle, measuring 2x2 cm in diameter (Fig. 1). (Fig. 2). On biomicroscopy the lesion appeared to compress both the lacrimal canaliculi.. The patient, however, PATHOLOGICAL FINDINGS did not complain of epiphora. No regional lympha- A white, ovoid, fleshy mass about 1-0 cm in denopathy was present. Further ocular examination, maximum dimension was partly covered by and

the medical history, and physical examination pro- with fat attached to the deep margin. Microscopic http://bjo.bmj.com/ vided no additional information. examination showed the mass to consist of a Corrcspondcnce to Dr A G E Nylander, West of England Eye carcinoma arranged in sheets and trabeculae (Fig. 3). Infirmary, Magdalen Street, Exeter EX2 4HT There were interconnected small cystic spaces in the on September 27, 2021 by guest. Protected

Fig. I Right eye, showing a round mass occupying the Fig. 2 Appearance ofthe right eyefollowing excision ofthe region ofthecaruncle. tumour. No evidence ofepiphora. 864 Br J Ophthalmol: first published as 10.1136/bjo.70.11.864 on 1 November 1986. Downloaded from

Adenosquamous carcinoma ofthe lacrimal caruncle 865

.1~ PI PMc i#e- .a - -. Fig. 3 Caruncle arranged in sheets and trabeculae with occasional mitosis. (Haematoxylin and eosin, x 234). Fig. 5 Subcutaneous border ofthe tumour withfibrous pseudocapsule in the lowerpart ofthepicture. centre of the tumour and a possible punctum opening (Haematoxylin and eosin, x 40.) on to normal skin covering the surface (Fig. 4). The deep margin had a fairly well defined and rounded border with a fibrous pseudocapsule (Fig. 5) in which 'adenosquamous' carcinoma probably arising in were a few chronic inflammatory cells. The appear- glands of the caruncle. ance suggested that the tumour had arisen in a gland copyright. opening into the skin surface. FO I LOW-U P The tumour was composed mainly of clear cells In view of the pathological findings the patient was with moderate nuclear pleomorphism and a fully investigated to exclude the presence of systemic moderate number of mitoses. In some places there malignancy. Full physical examination, chest x-ray, was partial glandular differentiation and in others full blood count, and liver function tests showed no squamous changes (Fig. 6). Stains for mucins were abnormality. A six-month follow-up showed no negative, and there was only a little glycogen. Classi- fication proved difficult, but descriptively it was an http://bjo.bmj.com/ on September 27, 2021 by guest. Protected

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Fig. 6 There is trabecular carcinoma in the upperpart with occasional acinarspaces. The lowercentreshows Fig. 4 Surface ofthe tumour with a punctum opening on to keratinising tumour (arrowed). (Haematoxylin and eosin, theskin (arrowed). (Haematoxylin and eosin, x 40.) x 105.) Br J Ophthalmol: first published as 10.1136/bjo.70.11.864 on 1 November 1986. Downloaded from

866 A G E Nylander and H R Atta evidence of systemic malignancy and no local recur- third recurrence the following year. This was in spite rence of tumour. The lacrimal canaliculi remained of surgery and irradiation therapy. Wilkerson and patent, and the patient did not complain of epiphora. Winguist'2 reported a bilateral papillary cystadenoma occurring in a woman six months after the first Discussion appearance. In our case we believe the early detection and Caruncle tumours are relatively rare,`'' and malig- radical excision of the tumour may yield a better nant tumours are reported to be extremely rare in this prognosis for the patient. It is generally believed that region.2 The commonest tumour is the papilloma,2 4 5 their relative isolation, ease of removal, and early while sebaceous carcinoma is the commonest malig- detection and treatment usually result in an increased nant tumour.2 survival. "'" The caruncle may be the seat of any tumour that occurs in the as well as the adjacent skin We thank Mr J M Smail for allowing us to describc this case; P W of the . This is because embryologically the Leedham for reporting on the histology; and Sue Fraser for typing caruncle is developed from the lower , being this paper. cordoned off by the lower canaliculus.6 Although References the surface epithelium is mucous membrane, the caruncle bears all the skin elements, including hair 1 Luthra CL, Doxanas MT, Green WR. Lesions of the caruncie: a clinicohistopathlogic study. Surv Ophihalmol 1978; 23: 183-95. follicles, sweat glands, and sebaceous glands.7 2 Maino JH, Richer SP, McGregor D. Carunclc lesions: the use of We were unable to find any previous reports of histological evaluation in determining a definitive diagnosis. mixed adenosquamous carcinoma arising within the J Am Optom Associ 1983; 54: 521-4. caruncle region, and it is our belief that this is the 3 Wilson RP. Tumours and cysts of the lacrimal caruncle. Trans OphthalmolSoc NZ 1958-9; 11: 23-32. first reported case. Cases of adenocarcinoma and 4 Evans WH. Tumour of the lacrimal caruncle: a study of 2(X) squamous carcinoma in the caruncle have been collected cases. Arch Ophthalmol 194t); 24: 83-1t)6. reported89 but not mixed. Luthra et al.' in reviewing 5 Serra GM. Tumori della caruncola lacrimale: studio clinico ed

112 caruncle lesions found no such lesion. Ni et al."' anatomico pathologicocasistica. Boll Oculist 1928; 7: 783-864. copyright. 6 Ask F. Ueber die Entwicklung der Caruncula lacrimalis bcim studied 512 cases of malignant eyelid tumours and Menschen, nebst Bemerkungen uber die Entwicklung der found no similar lesion. Tranenrohrchen und der meibomischen Drusen. Anat Anz 19t)7; The possibility of this tumour being metastatic in 30: 197. origin was raised. However, no systemic malignancy 7 Deutsch AR, Duckworth JK. Onkocytoma (oxyphilic adenoma) 64:45-1. was a The of the caruncle. Am J Ophthalmol 1967; found after six-month follow-up. possible 8 Boniuk M, Zimmerman LE. Sebaceous carcinoma of the eyelid, routes of spread of caruncle tumour should be , caruncle and . Ophthalmology (Rochester) 1968; considered. Apart from the usual routes of spread, 72: 619-42. namely, blood stream, lymph vessels, and direct 9 Duke-Elder S. System ofophthalmology. St Louis, Mosby: 1974; of 13 (1): 453. http://bjo.bmj.com/ metastases, Kielar" has suggested the possibility 10 Ni C, Scarl SS, Kuo PK, Chu FR, Chong CS, Albert DM. intracanalicular shedding of the tumour cells as an Sebaceous ceil carcinoma of the ocular adnexa. In: Ni C, Albert additional route. In the present case both canaliculi DM, eds. Tumours ofthe eyelids and orbits: a Chinese-American appeared to be involved, and complete excision of collaborative study. Boston: Little, Brown, 1982; 22: 23-61. the tumour involved part of the canaliculi. Of 11 Kielar RA. Sebaceous carcinoma of the caruncle. South Med J 1975; 68: 347-50. interest, however, is the full recanalisation of both 12 Wilkerson AJ, Winguist PG. Casc reports: bilateral papillary canaliculi and absence of epiphora following surgery. cystadenomas of the lacrimal carunei. Arch Pathol 1969; 88: The possibility of metastases is still present. Luthra 549-52. on September 27, 2021 by guest. Protected et al.' reported a case of adeno- 13 Duke-Elder S. System ofophthalmology. St Louis: Mosby, 1973: 13 (1): 592. carcinoma that first recurred 10 years after excision followed by a second recurrence a year later, then a Acceptedforpublication 30 January 1986.