Oncocytomas (oxyphil adenomas) of lacrimal · adenomas,89 papillary cystadenomas,'0-12 adeno-lyphoma-like tumours,13 and Warthin's tumour.'4 The author here reports 3 cases of caruncular

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  • Oncocytomas (oxyphil adenomas) of the lacrimal carunicle

    Fig. 2 Case 2. An1 oncocyto11t10with a pronounced papillarylcystic configuration. Note theback to back formation of theoncocytes separated by adelicate stroma. (H & E, x 60).

    found. In addition a small cystic lesion of the rightcaruncle was noted. The patient stated that thislesion had been present for several years and wasnot causing any discomfort.

    In October 1967 she attended the OphthalmicClinic again. Her visual acuity was now right eye6/36, left eye 5/60; this was not corrected withlenses. The caruncular lesion was still present andwas apparently unchanged in size. In April 1968bilateral intracapsular lens extractions were per-

    formed. Postoperatively her corrected visual acuitywas 6/9 in both eyes. In December 1971 the patientattended the Ophthalmology Clinic again becausethe lesion of her right caruncle was increasing insize and now troubling her. The lesion was describedas being a cystic swelling with several large bloodvessels crossing its anterior surface. These appear-ances led to the tentative diagnosis of an angioma.The lesion was excised and submitted for histo-logical examination.i.,x ......t.; :

    Fig. 3 Case 2. An accessorylacrimal gland containing asecretory duct exhibitingmarked oncocytic inetaplasia,slightly right of centre.

    I (H&E, x105).

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    Pathological examinationMacroscopic appearances. The lesion was describedas a ruptured cyst measuring 5 mm in diameter.

    Microscopic examination. Histologically the cystwall was composed of granular, intensely eosino-philic cuboidal cells forming a papillary-tubularstructure. Occasional cells contained large brownishgranules in their cytoplasm. These granules did notstain by any silver impregnation technique andwere considered to be lipofuscin granules. Scantymucus-secreting cells were present throughout thelesion. There was little nuclear pleomorphism, andmitotic figures were not identified in the sectionsstudied. A scanty condensation of connective tissuesurrounded the lesion.

    Discussion

    The term oncocyte was devised by Hamperl15 todescribe an epithelial cell that was characterised byits large size and eosinophilic granular cytoplasm.The derivation of the term (Greek: Onkausthui-toswell) was thought by Hamperl to convey theessential morphological characteristic of the cell,namely its large swollen appearance. Electronmicroscopic studies5 16-18 (and case 1 of this report)have since shown that the essential feature of thesecells is the number of mitochondria within thecytoplasm. It is believed that the increase in mito-chondria in the oncocyte reflects a defect of meta-bolism in this organelle leading to a compensatoryincrease in their number.19 Though the primarymetabolic defect is not known, it is thought to arisein old 'burnt out' cells, a concept inferred from theincreasing incidence on oncocytic lesions with age.

    Benign oncocytomas arising in ocular adnexahave been described in the lacrimal gland, lacrimalsac, plica semilunaris, bulbar conjunctiva, upperand lower fornices, mucocutaneous junction, andlacrimal caruncle. Of these, the caruncular oncocy-toma is by far the commonest; however, even inthis site it is a rarity. In a study of 200 lesionsarising from the caruncle Evans20 described only 1case which may have been an oncocytoma, theprecise histological details being unavailable. Sincethat report probably only 25 cases (excluding thisreport) have been described.Oncocytomas are generally regarded as benign

    tumours. However, sporadic reports of malignantoncocytomas have appeared. Malignant oncocy-tomas of the parotid have been described.2' 23 Onecase arising from a gland in the nasal mucosa hasalso been described.'9 Four malignant oncocytomasarising in ocular adnexa have been described, 2arising in the lacrimal sac24 25 and 2 occurring in thelacrimal gland.626 In relation to the lacrimal gland

    only 1 benign oncocytoma has been described.27 Itis surprising that so few lacrimal oncocytomas(benign or malignant) have been described, espe-cially when one considers the fact that oncocyticmetaplasia is known to occur in this organ. Bockand Schlagenhauff'8 examined the lacrimal glandsof 20 necropsies and found oncocytic metaplasia in7 of them (35%). Equally surprising is the apparentexcess of malignant tumours to benign lesions. Itis possible to attribute these anomalies to the extre-mely slow growth of the benign oncocytoma.Case 3 above illustrates the extremely slow

    growth of this tumour. The lesion was present forat least 7 years before excision. Thus benign lacrimaloncocytomas may never attain sufficient size tocause symptoms and thus escape detection. Incontrast, a malignant lesion with its more rapidgrowth and invasion of adjacent tissues would beexpected to produce symptoms.

    HISTOGENESIS OF CARUNCULARONCOCYTOMASThe histogenesis of the caruncular oncocytoma isstill open to discussion. Anatomically the lacrimalcaruncle is a piece of modified skin. It is covered bystratified squamous epithelium and contains hairfollicles, sebaceous glands, and sweat glands. Itdiffers from skin in that it contains accessory lacri-mal glands similar to those of Krause.These glands are usually in the centre of the

    caruncle and open by a sinuous duct near the plicasemilunaris.29 Three possible sites of origin areproposed-the surface epithelium, the conjunctiva,and the accessory lacrimal glands. Other suggestedsites of origin such as sweat glands2 or mucousglands'3 seem improbable, for oncocytomas are notdescribed arising from these structures in othersites.The accessory lacrimal glands or ducts have

    been proposed as the site of origin by severalauthors' 2 5 6 10-12 in view of their histologicalappearances (see below). Greer8 described 2 casesof caruncular oncocytoma which appeared to arisefrom the surface epithelium. Case 2 above similarlyshowed close apposition to the surface epithelium.However, in this example oncocytic metaplasia waspresent in the duct of an accessory lacrimal glandpresent near the tumour, though a direct continua-tion between the 2 could not be demonstrated.Forbes and Crawford" described a case in which adilated secretory duct displaying oncocytic meta-plasia was in direct continuity with an oncocytoma.A possible explanation for the existance of a lesionin apparent continuity with the surface epitheliumas described by Greer8 and the author (case 2) isthat the lesion could have arisen from the epithelium

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  • Onicocytomas (oxyphil adetiomas) of iic lacrimalcarlncle9

    of a secretory duct immediately beneath the carun-cular epithelium. Briggs and Font6 described focaloncocytic metaplasia in the ducts and acini ofaccessory lacrimal glands present in the region ofthe caruncle. Radnot and Lapis5 examined anoncocytoma with the transmission electron micro-scope and found, particularly at the periphery ofthe lesion, epithelial cells morphologically identicalto those of accessory lacrimal glands. It wouldseem unlikely that oncocytomas arise from thecaruncular epithelium proper, as this is a stratifiedsquamous epithelium similar to skin, and, so far asthe author is aware, oncocytomas have not beendescribed arising from cutaneous structures else-where in the body.

    It is possible that caruncular oncocytomas arisefrom the conjunctiva immediately adjacent to thecaruncle. In support of this argument Briggs andFont6 described 2 oncocytomas apparently arisingfrom the bulbar conjunctiva near the plica semi-lunaris. If oncocytomas were to arise mainly fromthe conjunctiva, then one should anticipate oncocy-tic changes described more frequently in the con-junctival epithelium, but this is not the case. More-over, in relation to lesions described arising fromthe plica semilunaris, it is to be noted that this isthe site of drainage of the secretory duct of thecaruncular lacrimal tissue. It would appear that themajority, if not all, caruncular oncocytomas arisefrom accessory lacrimal glands or their secretoryducts. This belief is supported by the similar histo-logical appearances of oncocytomas occurring atthe caruncle and thoEe occurring in other secretoryglands, namely the major salivary glands.

    CLINICAL ASPECTSTo the clinician the caruncular oncocytoma is ararity. Nevertheless the diagnosis should be con-sidered when he sees a caruncular swelling, particu-larly if the lesion appears cystic or has a brightcherry red colour. The lesion appears to occurmainly in postmenopausal females. In the 28 casesrecorded, including this report, 5 patients have beenmale (in one case the sex of the patient was notgiven), a ratio of 5 6:1 females to males. It usuallyhas an extremely slow rate of growth and oftenpresents as a chance finding.

    The author thanks Miss M. A. C. Jones and Mr C. A. L.Palmer for allowing the publication of their clinical material;Professor L. Henry and Mr A. Stanworth for their help andencouragement; Miss F. Bishop and Mrs S. Porter for theirassistance with the technical preparation of the specimens.

    References

    1 Radnot M. Seltene Geschwulste der CarUncula lacri-malis. Ophthalmologica 1947; 113: 270-5.

    2 Noguchi TT, Lonser ER. Oncocytoma (oxyphil-celladenoma) of the caruncle of the eyelid. Arch Pathol 1960;69: 516-9.

    3 Klein HJ. Zystisches Onkozytom der caruncula lacri-malis. Klin Monatsbl Augenheilkd 1965; 146: 343-8.

    4 Deutsch AR, Duckworth JK. Onkocytoma (oxyphilicadenoma) of the caruncle. Am J Ophthalmol 1967; 64:458-61.

    5 Radnot M, Lapis K. Ultrastructure of the caruncularoncocytoma. Ophthalmologica 1970; 161: 63-77.

    6 Briggs SL, Font RL. Oncocytic lesions of the caruncleand other ocular adenexa. Arc/h Ophthalmol 1977; 95:474-8.

    7 Nemeth B, Feher J. Oncocytoma carunculae. Ophthal-mologica 1977; 175: 1-4.

    8 Greer CH. Oxyphil adenoma of the lacrimal caruncle.Br J Ophthal/nol 1969; 53: 198-202.

    9 Dhermy P, Offret K. Adenome oxyphile a cellulesgranuleuses (oncocytome) de la caroncule. Arch Ophtal-inol (Paris) 1976; 36: 51-8.

    10 MacKenzie JR, Patience CR. Papillary cystadenoma ofthe lacrimal caruncle. J Pathol Bacteriol 1959; 78: 288-4.

    11 Forbes GB, Crawford RAD. Papillary cystadenoma oflacrimal caruncle. Br J Ophthalmol 1963; 47: 177-9.

    12 Wilkerson JA, Winquist WD. Bilateral papillary cysta-denoma of the lacrimal caruncle. Arch Pathol 1969; 88:540-52.

    13 Lennox B, Timperley WR, Murray D, Kellet HS. Adeno-lymphoma-like tumours of the lacrimal caruncle on thelarynx. J Pathol Bacteriol 1968; 96: 321-6.

    14 Oaks LW, Jenson MB. Warthin's tumour; papillarycystadenomalymphoma occurring in a human caruncle.Am J Ophthalmol 1963; 56: 459-61.

    15 Hamperl M. Onkocyten and Geschwulste der Speichel-drusen. Virchows Arch (Pathol Anat) 1931; 282: 724-36.

    16 Tandler B, Shipkey F. Ultrastructure of Warthin'stumour. J Ultrastruct Res 1964; 11: 292-305.

    17 Balogh K, Roth SI. Histochemical and electron micro-scopic studies of eosinophilic granular cells (oncocytes)in tumours of the parotid gland. Lab Invest 1965; 14:310-8.

    18 Tandler B, Hutter RVP, Erlandson RA. Ultrastructureof oncocytoma of the parotid gland. Lab ltvest 1970;23: 567-80.

    19 Hamperl H. Benign and malignant oncocytoma. Cancer1962; 15: 1019-27.

    20 Evans WM. Tumours of the lacrimal caruncle; a studyof 200 collecied cases. Arch Ophthalmol 1940; 24: 83-106.

    21 Bauer WH, Bauer JD. Classification of glandulartumours of salivary glands; study of 143 cases. ArchPathol 1953; 55: 328-46.

    22 Buxton RW, Maxwell JH, French AJ. Surgical treatmentof epithelial tumours of the parotid gland. Surg GynecolObstet 1953; 97: 401-16.

    23 Eneroth CM. Oncocytoma of major salivary glands.J Laryngol Otol 1965; 79: 1064-71.

    24 Ryan SJ, Font FL. Primary epithelial neoplasms of thelacrimal sac. Am J Ophthalmol 1973; 76: 83-8.

    25 Peretz WL, Ettinghausen SE, Gray GF. Oncocyticadenocarcinoma of the lacrimal sac. Arch Ophthallnol1978; 96: 303-4.

    26 Dorrello U. Carcinoma oncocitoria della ghiondolalacrimale. Riv oto-neuro-oftalmol 1961; 36: 452-61.

    27 Beskid M, Zorzycha M, Przypadek onkocytoma graczoloIzowego. Klin Oczna 1959; 29: 311-5.

    28 Bock J, Schlagenhauff K. Uber das Vorkommen vonOnkozyten in der menschlichen Trankudruse. Z Aiugen-heilkd 1938; 94: 224-55.

    29 Wolff E, revised Warick R. Anatomy of the Eye andOrbit. 7th ed. London: Lewis, 1976: 219-200.

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