Lacrimal gland choristomas

Coristomas de glândula lacrimal

Antonio Augusto V. Cruz Roberto Murillo Limongi Eduardo Damous Feijó Tim Jürg Enz About the authors

ABSTRACT

The purpose of this article was to report a case of intraconal lacrimal gland tissue and to review the literature on lacrimal gland choristoma. The magnetic resonance imaging findings of a biopsy-proven orbital case are also presented. A PubMed database search was performed using the key terms heterotopic, ectopic, aberrant, choristoma, and lacrimal gland to identify all the previously documented studies on lacrimal gland choristoma, in English, Spanish, and French. We classified the lacrimal gland choristoma cases classified according to the location of the lesions, clinical appearance, management, and outcome. The search targeting the period between 1887 and 2019 returned 79 articles, which were reviewed. We found a total of 113 cases of choristomas with normal lacrimal gland tissue. Only two of them were not associated with the eye or its adnexa while the remaining 111 lesions were found either on the ocular surface (n=46) or in the orbit (n=34). Intraocular choristomas were found in 18 patients, and the rest of the lesions were noted either on the eyelids (n=10) or in the lacrimal drainage system (n=3). Orbital and intraocular choristomas are the most harmful lesions as orbital choristomas are frequently associated with permanent diplopia while intraocular lacrimal gland choristomas have a poor visual prognosis and are a common cause of enucleation of the eye. In one of the reported cases, a corneal lacrimal gland choristoma had been experimentally induced by activating the FGF10 signaling pathway. Lacrimal gland choristomas are not uncommon. This peculiar type of lesion has been experimentally induced and may appear in a variety of locations associated with the globe and its adnexa.

Keywords:
Lacrimal gland; Choristoma; Prognosis

RESUMO

Descrever um caso de tecido de glândula lacrimal orbitário intracônico e revisar a literatura sobre choristomas lacrimais. Os termos heterotópico, ectópico, aberrante, choristoma e glândula lacrimal foram usados para procurar manuscritos em inglês, espanhol e francês. Todos os artigos encontrados foram revisados. Os choristomas lacrimais relatados foram classificados segundo a localização das lesões, aparência clínica, manejo e desfecho. Imagens de ressonância magnética de um novo caso orbitário comprovado por biópsia também são apresentadas. Foram revisados setenta e nove artigos, de 1887 a 2019, descrevendo 113 casos de choristomas constituídos por tecido de glândula lacrimal normal. Apenas 2 casos (2%) não estavam associados com o olho ou seus anexos. A maioria das 111 lesões restantes foram encontradas na superfície dos olhos (n=46) e nas órbitas (n=34). Os choristomas intraoculares foram encontrados em 18 pacientes. As demais lesões foram diagnosticadas nas pálpebras (n=10) e no sistema lacrimal (n=3). Os choristomas orbitais e intraoculares foram as lesões mais deletérias. Na órbita eles são frequentemente associados com diplopia permanente. Os choristomas lacrimais intraoculares imitam neoplasias e são uma causa comum de enucleação. Os choristomas lacrimais não são incomuns. Este tipo peculiar de lesão já foi induzido experimentalmente uma única vez e pode aparecer em uma variedade de locais associados ao globo e sua anexos. Os choristomas intraoculares têm um prognóstico visual ruim.

Descritores:
Glândula lacrimal; Coristoma; Prognostico

INTRODUCTION

Lacrimal gland choristomas are benign lesions formed by normal lacrimal gland tissue (LGT) present outside the lacrimal fossa. LGTs may occur in a variety of unusual places and are either heterotopic(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.), aberrant(22 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.), or ectopic(33 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.). This type of choristomas is uncommon and extremely difficult to identify in a clinical setting as they may mimic neoplastic(44 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.) or parasitic lesions(55 Pujari A, Ali MJ, Vemuganti GK, Naik MN. Intramuscular ectopic lacrimal gland cyst masquerading as myocysticercosis. Ophthal Plast Reconstr Surg. 2015;31(2):e45-7.). The knowledge related to these lesions is limited due to the inconsistency in the terminologies used to describe them. Also, the information pertaining to its epidemiology, topographic location, and neoplastic transformation is unorganized and scattered across the literature, adding to the challenge.

The purpose of this article was to describe a new case of intraconal LGT and perform a literature review of similar previous studies that were based on the location and clinical characteristics of the lacrimal gland choristomas.

METHODS

We searched the PubMed database using the key terms heterotopic, ectopic, aberrant, choristoma, and lacrimal gland (LG), for articles published between 1887 and 2019. All articles in English, Spanish, and French were reviewed and categorized based on the location of the lesions, their clinical appearance, management, and outcome. We also screened the references cited in the articles for additional reports, which were included in the literature analysis.

RESULTS

We found 79 articles from 1887 to 2019, describing 113 cases of LG choristomas(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.

2 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.

3 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.

4 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.

5 Pujari A, Ali MJ, Vemuganti GK, Naik MN. Intramuscular ectopic lacrimal gland cyst masquerading as myocysticercosis. Ophthal Plast Reconstr Surg. 2015;31(2):e45-7.

6 Dame LR. Accessory lacrimal gland on the cornea. Am J Ophthalmol. 1946;29(5):579-82.

7 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.

8 Boase AJ. Adenoma of the conjunctiva. Br J Ophthalmol. 1954; 38(6):380-2.

9 Mettier SR Jr. Aberrant lacrimal gland. AMA Arch Opthalmol. 1958;60(3):488-91.

10 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Oph­thalmol. 1960;44(10):619-25.

11 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.

12 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.

13 Baldridge M. Aberrant lacrimal gland in the orbit. Arch Ophthalmol. 1970;84(6):758-9.

14 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.

15 Zilkha A. Ectopic lacrimal gland and exophthalmos. Case report with emphasis on value of orbital venography. Radiology. 1972; 103(2):275-6.

16 Mueller EC, Borit A. Aberrant lacrimal gland and pleomorphic adenoma within the muscle cone. Ann Ophthalmol. 1979;11(4):661-3.

17 Rush A, Leone CR Jr. Ectopic lacrimal gland cyst of the orbit. Am J Ophthalmol. 1981;92(2):198-201.

18 Appel N, Som PM. Ectopic orbital lacrimal gland tissue. J Comput Assist Tomogr. 1982;6(5):1010-2.

19 Pe'er J, Ilsar M. Ectopic lacrimal gland under the nasal mucosa. Am J Ophthalmol. 1982;94(3):418-9.

20 Conway VH, Brownstein S, Chisholm IA. Lacrimal gland choristoma of the ciliary body. Ophthalmology. 1985;92(3):449-53.

21 Nikhil KC, Kaushik C. Ectopic lacrimal gland in the eyelids (a case report). Indian J Ophthalmol. 1985;33(1):65-6.

22 Margo CE, Naugle TC Jr, Karcioglu ZA. Ectopic lacrimal gland tissue of the orbit and sclerosing dacryoadenitis. Ophthalmic Surg. 1985;16(3):178-81.

23 Ghadially FN, Chisholm IA, Lalonde JM. Ultrastructure of an intraocular lacrimal gland choristoma. J Submicrosc Cytol. 1986;18(1):189-98.

24 Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology. 1986;93(10):1355-60.

25 von Domarus H. A lacrimal gland cyst in the orbit. J Craniomaxillofac Surg. 1987;15(2):106-9.

26 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.

27 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.

28 Kural G, Serifoglu A, Ertüre S. A case of aberrant lacrimal gland and fistula. Br J Ophthalmol. 1989;73(5):376-7.

29 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.

30 O'Donnell BA, Martin FJ, Kan AE, Filipic M. Intraocular lacrimal gland choristoma. Aust N Z J Ophthalmol. 1990;18(2):211-3.

31 Gordon AJ, Patrinely JR, Knupp JA, Font RL. Complex choristoma of the eyelid containing ectopic cilia and lacrimal gland. Ophthalmology. 1991;98(10):1547-50.

32 Boccato P, Blandamura S, Midena E, Carollo C. Orbital ectopic lacrimal gland tissue simulating a neoplasm. Report of a case with fine needle aspiration biopsy diagnosis. Acta Cytol. 1992;36(5):737-43.

33 Shields JA, Eagle RC Jr, Shields CL, De Potter P, Poliak JG. Natural course and histopathologic findings of lacrimal gland choristoma of the iris and ciliary body. Am J Ophthalmol. 1995;119(2):219-24.

34 Rowley SA, Karwatowski WS. Lacrimal gland choristoma of the ciliary body. Arch Ophthalmol. 1997;115(11):1482-3.

35 Sakurai H, Mitsuhashi N, Hayakawa K, Nozaki M, Kurosaki H, Kishi S, et al. Ectopic lacrimal gland of the orbit. J Nucl Med. 1997;38(9):1498-500.

36 Shields JA, Shields CL, Eagle RC Jr, Adkins J, De Potter P. Adenoid cystic carcinoma developing in the nasal orbit. Am J Ophthalmol. 1997;123(3):398-9.

37 West JA, Drewe RH, McNab AA. Atypical choristomatous cysts of the orbit. Aust N Z J Ophthalmol. 1997;25(2):117-23.

38 Klüppel M, Müller W, Sundmacher R. Lacrimal gland choristoma of the iris. Arch Ophthalmol. 1999;117(1):110-1.

39 Sim KT, Sullivan SC. Aberrant lacrimal gland within the tarsal plate presenting as hyperlacrimation. Eye (Lond). 1999;13(Pt 4):591-3.

40 Shields JA, Hogan RN, Shields CL, Eagle RC Jr, Weakley DR Jr. Intraocular lacrimal gland choristoma involving iris and ciliary body. Am J Ophthalmol. 2000;129(5):673-5.

41 Lee WR, Aitken DA, Kirkness CM. Diffuse hyperplasia of intratarsal ectopic lacrimal gland tissue. Arch Ophthalmol. 2002;120(12):1748-52.

42 Solaroglu I, Okutan O, Kaptanoglu E, Beskonakli E. Unilateral proptosis arising from an ectopic lacrimal gland of the orbit. J Neurosurg. 2005;103(3 Suppl):289-91.

43 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.

44 Kim BH, Henderson BA. Intraocular choristoma. Semin Ophthalmol. 2005;20(4):223-9.

45 Cho HY, Sa HS, Kang SW. Lacrimal gland choristoma of the ciliary body. Jpn J Ophthalmol. 2006;50(2):100-2.

46 Jung JY, Kim JH, Kim ST, Kim HJ, Weon YC. MR features of intraocular ectopic lacrimal tissue. AJNR Am J Neuroradiol. 2006;27(10):2196-8.

47 Kobrin EG, Shields CL, Danzig CJ, Ehya H, King RA, Shields JA. Intraocular lacrimal gland choristoma diagnosed by fine-needle aspiration biopsy. Cornea. 2007;26(6):753-5.

48 Yüceer N, Ozer E. Solitary ectopic lacrimal gland tissue of the orbit presenting with unilateral proptosis in a 5-year-old child. J Pediatr Neurosci. 2008;3(2):157-9.

49 Ranganathan D, Lenhart P, Hubbard GB, Grossniklaus H. Lacrimal gland choristoma in a preterm infant, presenting with spontaneous hyphema and increased intraocular pressure. J Perinatol. 2010;30(11):757-9.

50 Patyal S, Banarji A, Bhadauria M, Gurunadh VS. Pleomorphic adenoma of a subconjunctival ectopic lacrimal gland. Indian J Ophthalmol. 2010;58(3):245-7.

51 Lenzi R, Casani AP, Sellari-Franceschini S. Ectopic intraorbital lacrimal ductal cyst: a case report. Orbit. 2012;31(5):350-1.

52 Alsuhaibani AH. Slow-growing large pleomorphic adenoma of ectopic lacrimal gland tissue in the upper eyelid. Saudi J Ophthalmol. 2012;26(4):453-5.

53 Obi EE, Drummond SR, Kemp EG, Roberts F. Pleomorphic adenomas of the lower eyelid: a case series. Ophthal Plast Reconstr Surg. 2013;29(1):e14-7.

54 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.

55 Ferri S, Shinder R. Inferomedial dermolipoma with ectopic lacrimal gland. Ophthal Plast Reconstr Surg. 2013;29(2):e43-4.

56 Hughes WL, Ballen PH. Ectopic lacrimal gland. AMA Arch Opthalmol. 1956;55(2):271-3.

57 Bech K, Jensen OA. Mixed Tumor of the Lower Orbital Region. Arch Ophthalmol. 1965;74(2):226-8.

58 Kessing SV. Ectopic lacrimal gland tissue at the corneal limbus (gland of Manz?). Acta Ophthalmol (Copenh). 1968;46(3):398-403.

59 Braich PS, Silbert JE, Levada AJ, Schiff NR. Pseudo third cranial nerve palsy secondary to orbital ectopic lacrimal gland cyst: management with functional endoscopic sinus surgery. Ear Nose Throat J. 2014;93(2):77-9.

60 Misra S, Bhandari A, Misra N, Gogri P, Mahajan S. Pleomorphic adenoma of a deep orbital ectopic lacrimal gland. Orbit. 2016;35(5):295-7.

61 Raven ML, Larson JC, Potter HD. Ectopic lacrimal gland underlying a caruncular nevus. Ophthalmology. 2016;123(5):1136.

62 Ali MJ, Mishra DK, Naik MN. Ectopic lacrimal gland in a complex choristoma involving the lacrimal sac fossa. Saudi J Ophthalmol. 2017;31(3):162-4.

63 Wang Y, Jakobiec FA, Zakka FR, Lee NG. A Lacrimal gland choristoma of the lacrimal sac. Ophthal Plast Reconstr Surg. 2018;34(1):e3-6.

64 Singh S, James C, Curragh DS, Selva D. Lacrimal gland choristoma in lacrimal sac as a probable cause of nasolacrimal duct obstruction. Clin Exp Ophthalmol. 2019;47(5):675-7.

65 Wajda BN, Mancini R, Evers B, Nick Hogan R. A rare case of atypical pleomorphic adenoma arising from periocular ectopic lacrimal gland. Int Ophthalmol. 2019;39(7):1617-9.

66 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.

67 Suneetha N, Nirmala V, Thomas R, Battu R, Job R, Rawoof A. Isolated myositis: A rare presentation of intraorbital ectopic lacrimal gland. Indian J Ophthalmol. 1999;47:124-6.

68 Guy JR, Quisling RG. Ectopic lacrimal gland presenting as an orbital mass in childhood. AJNR Am J Neuroradiol. 1989;10(5 Suppl):S92.

69 Bruce GM. Aberrant glandular tissue in the iris. Trans Am Acad Ophthalmol Otolaryngol. 1952;56(1):47-51.

70 Mindlin A, Lamberts D, Barsky D. Mixed lacrimal gland tumor arising from ectopic lacrimal gland tissue in the orbit. J Pediatr Ophthalmol. 1977;14(1):44-7.

71 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.

72 Evans PA. Adenoma in an Eyelid. Br J Ophthalmol. 1964;48(4):234-5.

73 Jain BS. Pleomorphic Adenoma. Report of Two Uncommon Cases. Am J Ophthalmol. 1964;57(4):672-5.

74 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.

75 Boudet B, Bertezene. [Exophthalmos caused by lacrimal adenoma in an ectopic position (angiography of the orbit)]. Bull Soc Ophtalmol Fr. 1964;64:624-7. French.

76 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.

77 Jacobs L, Sirkin S, Kinkel W. Ectopic lacrimal gland in the orbit identified by computerized axial transverse tomography. Ann Ophthalmol. 1977;9(5):591-3.
-7878 Kao SC, Yeh LK, Tsai CC, Hsu WM. Ectopic lacrimal gland cyst of the orbit. Zhonghua Yi Xue Za Zhi (Taipei). 2000;63(4):334-8.). LGT was reported outside the limits of the eye and its adnexa in only two cases. As shown in tables 1 to 4, the remaining 111 cases were associated with either the eyeball (n=64%, 57.6%)(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.

2 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.

3 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.
-44 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.,66 Dame LR. Accessory lacrimal gland on the cornea. Am J Ophthalmol. 1946;29(5):579-82.

7 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.

8 Boase AJ. Adenoma of the conjunctiva. Br J Ophthalmol. 1954; 38(6):380-2.

9 Mettier SR Jr. Aberrant lacrimal gland. AMA Arch Opthalmol. 1958;60(3):488-91.

10 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Oph­thalmol. 1960;44(10):619-25.

11 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.
-1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,1414 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.,2020 Conway VH, Brownstein S, Chisholm IA. Lacrimal gland choristoma of the ciliary body. Ophthalmology. 1985;92(3):449-53.,2323 Ghadially FN, Chisholm IA, Lalonde JM. Ultrastructure of an intraocular lacrimal gland choristoma. J Submicrosc Cytol. 1986;18(1):189-98.,2424 Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology. 1986;93(10):1355-60.,2626 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.,2727 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.,2929 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.,3030 O'Donnell BA, Martin FJ, Kan AE, Filipic M. Intraocular lacrimal gland choristoma. Aust N Z J Ophthalmol. 1990;18(2):211-3.,3333 Shields JA, Eagle RC Jr, Shields CL, De Potter P, Poliak JG. Natural course and histopathologic findings of lacrimal gland choristoma of the iris and ciliary body. Am J Ophthalmol. 1995;119(2):219-24.,3434 Rowley SA, Karwatowski WS. Lacrimal gland choristoma of the ciliary body. Arch Ophthalmol. 1997;115(11):1482-3.,3838 Klüppel M, Müller W, Sundmacher R. Lacrimal gland choristoma of the iris. Arch Ophthalmol. 1999;117(1):110-1.,4040 Shields JA, Hogan RN, Shields CL, Eagle RC Jr, Weakley DR Jr. Intraocular lacrimal gland choristoma involving iris and ciliary body. Am J Ophthalmol. 2000;129(5):673-5.,4343 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.,4545 Cho HY, Sa HS, Kang SW. Lacrimal gland choristoma of the ciliary body. Jpn J Ophthalmol. 2006;50(2):100-2.

46 Jung JY, Kim JH, Kim ST, Kim HJ, Weon YC. MR features of intraocular ectopic lacrimal tissue. AJNR Am J Neuroradiol. 2006;27(10):2196-8.
-4747 Kobrin EG, Shields CL, Danzig CJ, Ehya H, King RA, Shields JA. Intraocular lacrimal gland choristoma diagnosed by fine-needle aspiration biopsy. Cornea. 2007;26(6):753-5.,4949 Ranganathan D, Lenhart P, Hubbard GB, Grossniklaus H. Lacrimal gland choristoma in a preterm infant, presenting with spontaneous hyphema and increased intraocular pressure. J Perinatol. 2010;30(11):757-9.,5050 Patyal S, Banarji A, Bhadauria M, Gurunadh VS. Pleomorphic adenoma of a subconjunctival ectopic lacrimal gland. Indian J Ophthalmol. 2010;58(3):245-7.,5454 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.

55 Ferri S, Shinder R. Inferomedial dermolipoma with ectopic lacrimal gland. Ophthal Plast Reconstr Surg. 2013;29(2):e43-4.
-5656 Hughes WL, Ballen PH. Ectopic lacrimal gland. AMA Arch Opthalmol. 1956;55(2):271-3.,5858 Kessing SV. Ectopic lacrimal gland tissue at the corneal limbus (gland of Manz?). Acta Ophthalmol (Copenh). 1968;46(3):398-403.,6161 Raven ML, Larson JC, Potter HD. Ectopic lacrimal gland underlying a caruncular nevus. Ophthalmology. 2016;123(5):1136.,6666 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.,6969 Bruce GM. Aberrant glandular tissue in the iris. Trans Am Acad Ophthalmol Otolaryngol. 1952;56(1):47-51.,7171 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.,7474 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.,7676 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.,7979 Aldossary MM, Alkatan HM, Maktabi AM. Epibulbar complex and osseous choristoma: clinicopathological study with interesting associations. Ann Med Surg (Lond). 2018;36:135-41.), orbit (n=34, 30.6%)(55 Pujari A, Ali MJ, Vemuganti GK, Naik MN. Intramuscular ectopic lacrimal gland cyst masquerading as myocysticercosis. Ophthal Plast Reconstr Surg. 2015;31(2):e45-7.,1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,1313 Baldridge M. Aberrant lacrimal gland in the orbit. Arch Ophthalmol. 1970;84(6):758-9.,1515 Zilkha A. Ectopic lacrimal gland and exophthalmos. Case report with emphasis on value of orbital venography. Radiology. 1972; 103(2):275-6.

16 Mueller EC, Borit A. Aberrant lacrimal gland and pleomorphic adenoma within the muscle cone. Ann Ophthalmol. 1979;11(4):661-3.

17 Rush A, Leone CR Jr. Ectopic lacrimal gland cyst of the orbit. Am J Ophthalmol. 1981;92(2):198-201.
-1818 Appel N, Som PM. Ectopic orbital lacrimal gland tissue. J Comput Assist Tomogr. 1982;6(5):1010-2.,2121 Nikhil KC, Kaushik C. Ectopic lacrimal gland in the eyelids (a case report). Indian J Ophthalmol. 1985;33(1):65-6.,2222 Margo CE, Naugle TC Jr, Karcioglu ZA. Ectopic lacrimal gland tissue of the orbit and sclerosing dacryoadenitis. Ophthalmic Surg. 1985;16(3):178-81.,2525 von Domarus H. A lacrimal gland cyst in the orbit. J Craniomaxillofac Surg. 1987;15(2):106-9.,3232 Boccato P, Blandamura S, Midena E, Carollo C. Orbital ectopic lacrimal gland tissue simulating a neoplasm. Report of a case with fine needle aspiration biopsy diagnosis. Acta Cytol. 1992;36(5):737-43.,3535 Sakurai H, Mitsuhashi N, Hayakawa K, Nozaki M, Kurosaki H, Kishi S, et al. Ectopic lacrimal gland of the orbit. J Nucl Med. 1997;38(9):1498-500.

36 Shields JA, Shields CL, Eagle RC Jr, Adkins J, De Potter P. Adenoid cystic carcinoma developing in the nasal orbit. Am J Ophthalmol. 1997;123(3):398-9.
-3737 West JA, Drewe RH, McNab AA. Atypical choristomatous cysts of the orbit. Aust N Z J Ophthalmol. 1997;25(2):117-23.,4242 Solaroglu I, Okutan O, Kaptanoglu E, Beskonakli E. Unilateral proptosis arising from an ectopic lacrimal gland of the orbit. J Neurosurg. 2005;103(3 Suppl):289-91.,4848 Yüceer N, Ozer E. Solitary ectopic lacrimal gland tissue of the orbit presenting with unilateral proptosis in a 5-year-old child. J Pediatr Neurosci. 2008;3(2):157-9.,5151 Lenzi R, Casani AP, Sellari-Franceschini S. Ectopic intraorbital lacrimal ductal cyst: a case report. Orbit. 2012;31(5):350-1.,5757 Bech K, Jensen OA. Mixed Tumor of the Lower Orbital Region. Arch Ophthalmol. 1965;74(2):226-8.,5959 Braich PS, Silbert JE, Levada AJ, Schiff NR. Pseudo third cranial nerve palsy secondary to orbital ectopic lacrimal gland cyst: management with functional endoscopic sinus surgery. Ear Nose Throat J. 2014;93(2):77-9.,6060 Misra S, Bhandari A, Misra N, Gogri P, Mahajan S. Pleomorphic adenoma of a deep orbital ectopic lacrimal gland. Orbit. 2016;35(5):295-7.,6868 Guy JR, Quisling RG. Ectopic lacrimal gland presenting as an orbital mass in childhood. AJNR Am J Neuroradiol. 1989;10(5 Suppl):S92.,7070 Mindlin A, Lamberts D, Barsky D. Mixed lacrimal gland tumor arising from ectopic lacrimal gland tissue in the orbit. J Pediatr Ophthalmol. 1977;14(1):44-7.,7373 Jain BS. Pleomorphic Adenoma. Report of Two Uncommon Cases. Am J Ophthalmol. 1964;57(4):672-5.,7575 Boudet B, Bertezene. [Exophthalmos caused by lacrimal adenoma in an ectopic position (angiography of the orbit)]. Bull Soc Ophtalmol Fr. 1964;64:624-7. French.,7777 Jacobs L, Sirkin S, Kinkel W. Ectopic lacrimal gland in the orbit identified by computerized axial transverse tomography. Ann Ophthalmol. 1977;9(5):591-3.,7878 Kao SC, Yeh LK, Tsai CC, Hsu WM. Ectopic lacrimal gland cyst of the orbit. Zhonghua Yi Xue Za Zhi (Taipei). 2000;63(4):334-8.), or less commonly, the eyelids (n=10, 9.0%)(3131 Gordon AJ, Patrinely JR, Knupp JA, Font RL. Complex choristoma of the eyelid containing ectopic cilia and lacrimal gland. Ophthalmology. 1991;98(10):1547-50.,3939 Sim KT, Sullivan SC. Aberrant lacrimal gland within the tarsal plate presenting as hyperlacrimation. Eye (Lond). 1999;13(Pt 4):591-3.,4141 Lee WR, Aitken DA, Kirkness CM. Diffuse hyperplasia of intratarsal ectopic lacrimal gland tissue. Arch Ophthalmol. 2002;120(12):1748-52.,5252 Alsuhaibani AH. Slow-growing large pleomorphic adenoma of ectopic lacrimal gland tissue in the upper eyelid. Saudi J Ophthalmol. 2012;26(4):453-5.,5353 Obi EE, Drummond SR, Kemp EG, Roberts F. Pleomorphic adenomas of the lower eyelid: a case series. Ophthal Plast Reconstr Surg. 2013;29(1):e14-7.,6565 Wajda BN, Mancini R, Evers B, Nick Hogan R. A rare case of atypical pleomorphic adenoma arising from periocular ectopic lacrimal gland. Int Ophthalmol. 2019;39(7):1617-9.,7272 Evans PA. Adenoma in an Eyelid. Br J Ophthalmol. 1964;48(4):234-5.,7373 Jain BS. Pleomorphic Adenoma. Report of Two Uncommon Cases. Am J Ophthalmol. 1964;57(4):672-5.). Out of the 64 cases related to the globe,18 (28.1%) were intraocular uveal choristomas(22 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.

3 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.
-44 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.,1010 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Oph­thalmol. 1960;44(10):619-25.,1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,1414 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.,2020 Conway VH, Brownstein S, Chisholm IA. Lacrimal gland choristoma of the ciliary body. Ophthalmology. 1985;92(3):449-53.,2323 Ghadially FN, Chisholm IA, Lalonde JM. Ultrastructure of an intraocular lacrimal gland choristoma. J Submicrosc Cytol. 1986;18(1):189-98.,3030 O'Donnell BA, Martin FJ, Kan AE, Filipic M. Intraocular lacrimal gland choristoma. Aust N Z J Ophthalmol. 1990;18(2):211-3.,3333 Shields JA, Eagle RC Jr, Shields CL, De Potter P, Poliak JG. Natural course and histopathologic findings of lacrimal gland choristoma of the iris and ciliary body. Am J Ophthalmol. 1995;119(2):219-24.,3434 Rowley SA, Karwatowski WS. Lacrimal gland choristoma of the ciliary body. Arch Ophthalmol. 1997;115(11):1482-3.,3838 Klüppel M, Müller W, Sundmacher R. Lacrimal gland choristoma of the iris. Arch Ophthalmol. 1999;117(1):110-1.,4040 Shields JA, Hogan RN, Shields CL, Eagle RC Jr, Weakley DR Jr. Intraocular lacrimal gland choristoma involving iris and ciliary body. Am J Ophthalmol. 2000;129(5):673-5.,4545 Cho HY, Sa HS, Kang SW. Lacrimal gland choristoma of the ciliary body. Jpn J Ophthalmol. 2006;50(2):100-2.

46 Jung JY, Kim JH, Kim ST, Kim HJ, Weon YC. MR features of intraocular ectopic lacrimal tissue. AJNR Am J Neuroradiol. 2006;27(10):2196-8.
-4747 Kobrin EG, Shields CL, Danzig CJ, Ehya H, King RA, Shields JA. Intraocular lacrimal gland choristoma diagnosed by fine-needle aspiration biopsy. Cornea. 2007;26(6):753-5.,4949 Ranganathan D, Lenhart P, Hubbard GB, Grossniklaus H. Lacrimal gland choristoma in a preterm infant, presenting with spontaneous hyphema and increased intraocular pressure. J Perinatol. 2010;30(11):757-9.,6969 Bruce GM. Aberrant glandular tissue in the iris. Trans Am Acad Ophthalmol Otolaryngol. 1952;56(1):47-51.) and 46 (71.9%) were epibulbar lesions(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.,77 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.

8 Boase AJ. Adenoma of the conjunctiva. Br J Ophthalmol. 1954; 38(6):380-2.
-99 Mettier SR Jr. Aberrant lacrimal gland. AMA Arch Opthalmol. 1958;60(3):488-91.,1111 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.,2424 Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology. 1986;93(10):1355-60.,2626 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.,2727 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.,2929 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.,4343 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.,5050 Patyal S, Banarji A, Bhadauria M, Gurunadh VS. Pleomorphic adenoma of a subconjunctival ectopic lacrimal gland. Indian J Ophthalmol. 2010;58(3):245-7.,5454 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.

55 Ferri S, Shinder R. Inferomedial dermolipoma with ectopic lacrimal gland. Ophthal Plast Reconstr Surg. 2013;29(2):e43-4.
-5656 Hughes WL, Ballen PH. Ectopic lacrimal gland. AMA Arch Opthalmol. 1956;55(2):271-3.,5858 Kessing SV. Ectopic lacrimal gland tissue at the corneal limbus (gland of Manz?). Acta Ophthalmol (Copenh). 1968;46(3):398-403.,6161 Raven ML, Larson JC, Potter HD. Ectopic lacrimal gland underlying a caruncular nevus. Ophthalmology. 2016;123(5):1136.,7171 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.,7474 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.,7676 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.,7979 Aldossary MM, Alkatan HM, Maktabi AM. Epibulbar complex and osseous choristoma: clinicopathological study with interesting associations. Ann Med Surg (Lond). 2018;36:135-41.). Interestingly, 38 (82.6%) of the 46 epibulbar lesions were found in the lateral half of the globe(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.,66 Dame LR. Accessory lacrimal gland on the cornea. Am J Ophthalmol. 1946;29(5):579-82.,77 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.,1111 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.,2626 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.,2727 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.,2929 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.,4343 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.,5050 Patyal S, Banarji A, Bhadauria M, Gurunadh VS. Pleomorphic adenoma of a subconjunctival ectopic lacrimal gland. Indian J Ophthalmol. 2010;58(3):245-7.,5454 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.,5656 Hughes WL, Ballen PH. Ectopic lacrimal gland. AMA Arch Opthalmol. 1956;55(2):271-3.,6666 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.,7171 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.,7474 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.,7676 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.,7979 Aldossary MM, Alkatan HM, Maktabi AM. Epibulbar complex and osseous choristoma: clinicopathological study with interesting associations. Ann Med Surg (Lond). 2018;36:135-41.), involving the superotemporal quadrant(11 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.,1111 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.,2626 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.,2727 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.,2929 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.,4343 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.,6666 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.,7171 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.,7474 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.,7676 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.) in majority of the cases (n=15, 32.6%). Among the 34 orbital cho­ristomas, 16 (50%) were diagnosed on the lateral aspect of the intra- or extraconal orbital compartment(12,13,15,16,18,22,25,32,42,48,57,60,67,73,75). Kural, in his article, described the case of a 9-year-old boy with a lacrimal fistula in the middle of the temporal fossa, which was surgically explored and an extraorbital LG was removed(2828 Kural G, Serifoglu A, Ertüre S. A case of aberrant lacrimal gland and fistula. Br J Ophthalmol. 1989;73(5):376-7.). Pe'er reported the finding of normal LGT in the nasal mucosa, in his study(1919 Pe'er J, Ilsar M. Ectopic lacrimal gland under the nasal mucosa. Am J Ophthalmol. 1982;94(3):418-9.). The occurrence of ectopic LGT within the lacrimal drainage system is extremely rare with only three cases reported in the lacrimal sac so far(6262 Ali MJ, Mishra DK, Naik MN. Ectopic lacrimal gland in a complex choristoma involving the lacrimal sac fossa. Saudi J Ophthalmol. 2017;31(3):162-4.,6363 Wang Y, Jakobiec FA, Zakka FR, Lee NG. A Lacrimal gland choristoma of the lacrimal sac. Ophthal Plast Reconstr Surg. 2018;34(1):e3-6.,6565 Wajda BN, Mancini R, Evers B, Nick Hogan R. A rare case of atypical pleomorphic adenoma arising from periocular ectopic lacrimal gland. Int Ophthalmol. 2019;39(7):1617-9.).

Table 1
Epibulbar lacrimal choristomas
Table 2
Intraocular lacrimal choristomas
Table 3
Orbital lacrimal choristomas
Table 4
Eyelid lacrimal choristomas

The overall female-to-male ratio was 1:1. The age of the patients at the time of surgical management of the LG choristomas correlated with the location of the lesion. The intraocular choristomas were managed at a median age of 10.5 months, whereas the epibulbar, orbital, and eyelid lesions were addressed much later in life, at a median age of 13, 18, and 43.5 years, respectively. Orbital choristomas were especially difficult to differentiate from inflammatory and neoplastic conditions. In all the reported cases, the LG choristomas were confirmed only after biopsy and histopathological examination.

Case Presentation

A 30-year-old male presented to the University Hospital of Ribeirão Preto for a follow-up examination after having undergone lateral orbitotomy elsewhere 2 years ago. According to him, the procedure had been performed to treat right eye proptosis, secondary to a large orbital mass. His past medical history was significant for multiple steroid injections into the right orbit for the management of a presumed orbital hemangioma during childhood. Examination of the right eye revealed no proptosis and unrestricted ocular motility. The funduscopic and visual field examinations were normal. Magnetic resonance imaging (MRI) showed multiple, large intraconal cysts in the right orbit. T1- and T2-weighted MRI revealed well-circumscribed masses around the right optic nerve with high signal intensity compared to the vitreous on T1W and T2W images (Figure 1). Histopathological evaluation of the sections of specimen obtained from the previous orbital surgery revealed ducts and acini of benign LGT (Figure 2) and absence of atypical cells, mitotic activity, and necrosis. No further intervention was planned as the patient was asymptomatic.

Figure 1
T2 - weighted magnetic resonance images of normal lacrimal tissue within the intraconal space of the right orbit. Notice the isolated tissue lobe in the axial (A) and coronal (B) slices. A large apical componente of the lesion is seen in the sagittal slice (C).

Figure 2
Histopathology showing normal lacrimal gland tissue (Hematoxylin-eosin x 200).

DISCUSSION

The occurrence of ectopic LGTs and intraocular lacrimal gland choristomas, as well as their progression into tumors, has been known since the 19th century. In 1891, Schirmer reported a case of conjunctival adenoma located between the insertion point of the medial rectus muscle and plica semilunaris(77 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.). Puech is considered to be the first to have reported a choroidal adenoma in an adult female in 1887(1010 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Oph­thalmol. 1960;44(10):619-25.,1414 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.). A large number of LG choristoma cases have been published since these early reports. Although the present review is highly representative of the studies reported in the literature, it is not exhaustive. Some ancient papers could not be retrieved, and manuscripts written in European languages were not included. The analysis of the literature review described in this article is similar to that of a recent publication on the same subject(8181 Nagendran S, Alsamnan M, Strianese D, Malhotra R. Ectopic Lacrimal gland tissue: A systematic review. Ophthalmic Plast Reconstr Surg. 2020;36(6):540-4.) where it has been presented in a more condensed form.

A question is whether ectopic LGT lesions are choristomas. The term choristoma is derived from the Greek word choristos (separated)-a tumor-like growth of microscopically normal tissue presents in an abnormal location(8282 Mansour AM, Barber JC, Reinecke RD, Wang FM. Ocular choristomas. Surv Ophthalmol. 1989;33(5):339-58.). The LG is located in the orbital lacrimal fossa. Accessory LGs are present in the superior and inferior conjunctival fornix (glands of Krause), along the nonmarginal border of the tarsal plates (glands of Wolfring), and in the plica semilunaris and the caruncle(8383 Bron AJ, Tripathi RC, Tripathi B, editors. Wolff's Anatomy of the eye and orbit. London: Chapman & Hall Medical; 1997. The ocular appendages: eyelids, conjunctiva and lacrimal apparatus. p. 30-72.). Thus, LGT lesions should be considered choristomas only if found outside these locations. Table 2 presents the cases of intraocular lacrimal choristomas found at different locations, as reported by several authors over the years. Ectopic LGT lesions might be a result of changes in the accessory lacrimal glands as in the case reported by Boase and Bullock who found an "ectopic" lacrimal cyst involving the caruncle or the medial aspect of the conjunctiva near the plica(88 Boase AJ. Adenoma of the conjunctiva. Br J Ophthalmol. 1954; 38(6):380-2.,2424 Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology. 1986;93(10):1355-60.).

The second important characteristic of an ectopic choristoma is that it is distinctly separated from the anatomical structure it is derived from. A definite clea­vage between a superolateral mass of LGT and the LG should be evident for consideration of an LGT as ectopic(8484 Henderson J, Campbell R, Farrow G, Garrity J. Ectopic lacrimal gland. In: Henderson J, Campbell R, Farrow G, Garrity J, editors. Orbital tumors. New York: Raven Press; 1994. p. 417-8.). Ectopic LGTs have been noted to be most commonly located in the epibulbar conjunctiva. However, in some of the epibulbar LGT cases reported previously, it is not clear if the superolateral masses that were considered as ectopic were actually detached from the LG(1111 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.,2626 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.,2727 Pokorny KS, Hyman BM, Jakobiec FA, Perry HD, Caputo AR, Iwamoto T. Epibulbar choristomas containing lacrimal tissue. Clinical distinction from dermoids and histologic evidence of an origin from the palpebral lobe. Ophthalmology. 1987;94(10):1249-57.,2929 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.,4343 Duncan JL, Golabi M, Fredrick DR, Hoyt CS, Hwang DG, Kramer SG, et al. Complex limbal choristomas in linear nevus sebaceous syndrome. Ophthalmology. 1998;105(8):1459-65.,5454 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.,6161 Raven ML, Larson JC, Potter HD. Ectopic lacrimal gland underlying a caruncular nevus. Ophthalmology. 2016;123(5):1136.,6666 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.,7171 Raina UK, Chaudhuri Z. Ectopic lacrimal glandular tissue in a neonate with giant congenital melanocytic nevi and cutis verticis gyrata. J Pediatr Ophthalmol Strabismus. 2010;47 Online:e1-4.,7474 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.,7676 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.).

A critical analysis of the published cases also raises doubts about whether all the reported lesions within the orbit represent true choristomas. Green et al. described a case wherein the excised tissue was attached to the orbital lobe of the LG(1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.). Kao reported a case of an inferior dacryops (rare, benign lacrimal gland/duct cyst) that was similar that of the accessory LGs usually found in the inferior fornix(7878 Kao SC, Yeh LK, Tsai CC, Hsu WM. Ectopic lacrimal gland cyst of the orbit. Zhonghua Yi Xue Za Zhi (Taipei). 2000;63(4):334-8.). The orbital images of superotemporal lesions found in some other cases do not show a clear disunion between the LGTs labeled as ectopic and the LG(3232 Boccato P, Blandamura S, Midena E, Carollo C. Orbital ectopic lacrimal gland tissue simulating a neoplasm. Report of a case with fine needle aspiration biopsy diagnosis. Acta Cytol. 1992;36(5):737-43.,6060 Misra S, Bhandari A, Misra N, Gogri P, Mahajan S. Pleomorphic adenoma of a deep orbital ectopic lacrimal gland. Orbit. 2016;35(5):295-7.).

Only two other studies(1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,8585 Alyahya GA, Bangsgaard R, Prause JU, Heegaard S. Occurrence of lacrimal gland tissue outside the lacrimal fossa: comparison of clinical and histopathological findings. Acta Ophthalmol Scand. 2005;83(1):100-3.) on LG choristomas have described the distribution of lesions similar to that presented in our review. Alyahya et al analyzed sections of 61 LG choristoma specimen spanning 50 years, collected from the files of the Eye Pathology Institute of Copenhagen. They compared the details on location and clinical diagnosis of the lesion specimen with their histological findings. Out of the 61 lesions, 43 (70.5%) were found on the superotemporal portion of the bulbar conjunctiva(8585 Alyahya GA, Bangsgaard R, Prause JU, Heegaard S. Occurrence of lacrimal gland tissue outside the lacrimal fossa: comparison of clinical and histopathological findings. Acta Ophthalmol Scand. 2005;83(1):100-3.). Although these studies did not provide any clinical information, they clearly specified the lo­cation of the lesions.

Knowledge of the LG's embryonic development is essential for speculating about the origin of lacrimal gland choristomas. LG formation consists of three stages(8686 de la Cuadra-Blanco C, Peces-Peña MD, Mérida-Velasco JR. Morphogenesis of the human lacrimal gland. J Anat. 2003;203(5):531-6.). During stage 1, LG appears as a thickening of the superior fornix epithelium and surrounding mesenchymal condensation at O'Rahilly stages 19-20 (crown-rump length 17-20 mm, 48-51 days). Stage 2 marks an epithelial bud formation at O'Rahilly stages 21-23 (crown-rump length 23-28 mm, 52-57 days). Stage 3 of LG development occurs during the fetal period (weeks 13-16) and is characterized by gland maturity.

The embryonic development of LG is based on the general process of branching morphogenesis as it has a tubulo-acinar branching structure (lobules formed by acini and intralobular, interlobular, and excretory ducts) similar to a bunch of grapes(8787 Schechter JE, Warren DW, Mircheff AK. A lacrimal gland is a lacrimal gland, but rodent's and rabbit's are not human. Ocul Surf. 2010;8(3):111-34.,8888 Hogan BL. Morphogenesis. Cell. 1999;96(2):225-33.). LG development depends on the interaction between the surface ectoderm and surrounding mesenchyme, which is facilitated by the exchange of chemical signals between groups of cells. This occurs through specific signaling pathways such as fibroblast growth factor 10 (FGF10), homeobox transcription factor Barx2, bone morphogenetic protein 7 (BPM7), and the canonical Wnt signaling family(8989 Carlson B, editor. Human embryology and developmental biology. Philadelphia: Saunders; 2014. Molecular basis for embryonic development p. 58-74.

90 Yao Y, Zhang Y. The lacrimal gland: development, wound repair and regeneration. Biotechnol Lett. 2017;39(7):939-49.
-9191 Garg A, Zhang X. Lacrimal gland development: from signaling interactions to regenerative medicine. Dev Dyn. 2017;246(12):970-80.).

Mesenchymal FGF10 expression is crucial for LG induction(9292 Makarenkova HP, Ito M, Govindarajan V, Faber SC, Sun L, McMahon G, et al. FGF10 is an inducer and Pax6 a competence factor for lacrimal gland development. Development. 2000;127(12):2563-72.

93 Qu X, Pan Y, Carbe C, Powers A, Grobe K, Zhang X. Glycosaminoglycan-dependent restriction of FGF diffusion is necessary for lacrimal gland development. Development. 2012;139(15):2730-9.
-9494 Govindarajan V, Ito M, Makarenkova HP, Lang RA, Overbeek PA. Endogenous and ectopic gland induction by FGF-10. Dev Biol. 2000;225(1):188-200.) as it promotes epithelial gland proliferation by directly activating FGF receptor-2 IIIb in the conjunctival epithelium. Barx2, BPM7, and the canonical Wnt signaling family are responsible for the branching morphogenesis. Barx2, which is expressed in the epithelium, is necessary for branching elongation(9595 Tsau C, Ito M, Gromova A, Hoffman MP, Meech R, Makarenkova HP. Barx2 and Fgf10 regulate ocular glands branching morphogenesis by controlling extracellular matrix remodeling. Development. 2011;138(15):3307-17.). BMP7 is expressed mainly in the mesenchyme, where it promotes the proliferation and aggregation of the cells, and a lack of this molecule impairs bud formation(9696 Dean C, Ito M, Makarenkova HP, Faber SC, Lang RA. Bmp7 regulates branching morphogenesis of the lacrimal gland by promoting mesenchymal proliferation and condensation. Development. 2004;131(17):4155-65.). The canonical Wnt cascade, on the contrary, is induced in the gland epithelium, inhibiting branching(9797 Dean CH, Miller LA, Smith AN, Dufort D, Lang RA, Niswander LA. Canonical Wnt signaling negatively regulates branching morphogenesis of the lung and lacrimal gland. Dev Biol. 2005;286(1):270-86.).

The evolving knowledge of the molecular mechanisms involved in LG morphogenesis rules out the old theory that lacrimal gland choristomas are a result of an aberrant migration of cells(22 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.). In fact, it has already been experimentally demonstrated that activation of the FGF10 signaling pathway can induce LGT formation within the cornea(9494 Govindarajan V, Ito M, Makarenkova HP, Lang RA, Overbeek PA. Endogenous and ectopic gland induction by FGF-10. Dev Biol. 2000;225(1):188-200.). Thus, it can be safely concluded that lacrimal gland choristomas result from localized abnormal expression of the signaling molecules associated with LG morphogenesis. A similar conclusion was drawn by Milman et al. who analyzed the immunohistochemical characteristics of lacrimal hamartomas(9898 Milman T, Jakobiec FA, Lally SE, Shields JA, Shields CL, Eagle RC Jr. Lacrimal Gland Hamartoma (Formerly Termed Dacryoadenoma). Am J Ophthalmol. 2020;217:189-97.). They also noted that these lesions are derived from indigenous conjunctival precursor cells activated by mesenchymal influences.

The clinical management of lacrimal gland choristomas and their impact on vision depend on the location of the LGT. Epibulbar lesions can be easily excised without serious impairment of visual function. Choristomas in the eyelids also present as solitary masses which can be easily managed. However, orbital choristomas are very difficult to diagnose and remove and can severely impair vision. As noted in table 2, 8 out of 34 patients had persistent diplopia(1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,1313 Baldridge M. Aberrant lacrimal gland in the orbit. Arch Ophthalmol. 1970;84(6):758-9.,1515 Zilkha A. Ectopic lacrimal gland and exophthalmos. Case report with emphasis on value of orbital venography. Radiology. 1972; 103(2):275-6.,4848 Yüceer N, Ozer E. Solitary ectopic lacrimal gland tissue of the orbit presenting with unilateral proptosis in a 5-year-old child. J Pediatr Neurosci. 2008;3(2):157-9.,5959 Braich PS, Silbert JE, Levada AJ, Schiff NR. Pseudo third cranial nerve palsy secondary to orbital ectopic lacrimal gland cyst: management with functional endoscopic sinus surgery. Ear Nose Throat J. 2014;93(2):77-9.,7575 Boudet B, Bertezene. [Exophthalmos caused by lacrimal adenoma in an ectopic position (angiography of the orbit)]. Bull Soc Ophtalmol Fr. 1964;64:624-7. French.,7777 Jacobs L, Sirkin S, Kinkel W. Ectopic lacrimal gland in the orbit identified by computerized axial transverse tomography. Ann Ophthalmol. 1977;9(5):591-3.), two patients suffered vision loss(1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.), and one patient had to be exenterated due to malignant transformation(3636 Shields JA, Shields CL, Eagle RC Jr, Adkins J, De Potter P. Adenoid cystic carcinoma developing in the nasal orbit. Am J Ophthalmol. 1997;123(3):398-9.).

Intraocular development of LGT poses the highest risk for vision loss. Based on the information in the literature, 9 out of 18 affected eyes were enucleated(22 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.

3 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.
-44 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.,1010 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Oph­thalmol. 1960;44(10):619-25.,1212 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.,1414 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.,2020 Conway VH, Brownstein S, Chisholm IA. Lacrimal gland choristoma of the ciliary body. Ophthalmology. 1985;92(3):449-53.,2323 Ghadially FN, Chisholm IA, Lalonde JM. Ultrastructure of an intraocular lacrimal gland choristoma. J Submicrosc Cytol. 1986;18(1):189-98.,4949 Ranganathan D, Lenhart P, Hubbard GB, Grossniklaus H. Lacrimal gland choristoma in a preterm infant, presenting with spontaneous hyphema and increased intraocular pressure. J Perinatol. 2010;30(11):757-9.), mainly due to the difficult differential diagnosis of intraocular malignancies such as medulloepithelioma(44 Tauziède-Espariat A, Roche O, Dufier JL, Putterman M. Recurrent lacrimal gland choristoma of the ciliary body in an infant mimicking a medulloepithelioma. Eur J Ophthalmol. 2016;26(6):e145-8.). The tear fluid is composed of electrolytes, metabolites, lipids, mucins, and a large number of proteins. Recent proteomic studies have identified more than 1,516 proteins in tears(9999 Aass C, Norheim I, Eriksen EF, Thorsby PM, Pepaj M. Single unit filter-aided method for fast proteomic analysis of tear fluid. Anal Biochem. 2015;480:1-5.). Therefore, intraocular tear fluid production by ectopic LGT could contribute to the inflammation(3434 Rowley SA, Karwatowski WS. Lacrimal gland choristoma of the ciliary body. Arch Ophthalmol. 1997;115(11):1482-3.) or increase in ocular pressure, which has been verified in several studies(1414 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.,3030 O'Donnell BA, Martin FJ, Kan AE, Filipic M. Intraocular lacrimal gland choristoma. Aust N Z J Ophthalmol. 1990;18(2):211-3.,3333 Shields JA, Eagle RC Jr, Shields CL, De Potter P, Poliak JG. Natural course and histopathologic findings of lacrimal gland choristoma of the iris and ciliary body. Am J Ophthalmol. 1995;119(2):219-24.,3838 Klüppel M, Müller W, Sundmacher R. Lacrimal gland choristoma of the iris. Arch Ophthalmol. 1999;117(1):110-1.,4747 Kobrin EG, Shields CL, Danzig CJ, Ehya H, King RA, Shields JA. Intraocular lacrimal gland choristoma diagnosed by fine-needle aspiration biopsy. Cornea. 2007;26(6):753-5.,4949 Ranganathan D, Lenhart P, Hubbard GB, Grossniklaus H. Lacrimal gland choristoma in a preterm infant, presenting with spontaneous hyphema and increased intraocular pressure. J Perinatol. 2010;30(11):757-9.).

  • Funding: This study received no specific financial support.

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Publication Dates

  • Publication in this collection
    10 Sept 2021
  • Date of issue
    2022

History

  • Received
    06 May 2020
  • Accepted
    12 Oct 2020
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