Lacrimal gland choristomas

Cruz, Antonio Augusto V.; Limongi, Roberto Murillo; Feijó, Eduardo Damous; Enz, Tim Jürg

doi:10.5935/0004-2749.20220029

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ORIGINAL ARTICLE • Arq. Bras. Oftalmol. 85 (02) • 2022 • https://doi.org/10.5935/0004-2749.20220029 copy

Lacrimal gland choristomas

Coristomas de glândula lacrimal

Authorship SCIMAGO INSTITUTIONS RANKINGS

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

Author/year	Cases	Sex	Age	Location	Clinical appearance	Treatment	Histopathology
Dame/1946⁽⁶6 Dame LR. Accessory lacrimal gland on the cornea. Am J Ophthalmol. 1946;29(5):579-82.⁾	1	M	28 years	Inferotemporal bulbar conjunctiva/Cornea	Mass	Excision	Normal LGT
François et al./1951⁽⁷7 François J, Rabaey M. Adenoma of the limbal conjunctiva. Br J Ophthalmol. 1951;35(4):237-41.⁾	1	F	36 years	Inferonasal limbal conjunctiva	Cystic	Excision	Adenoma admixed with normal LGT
Braun-Vallon/1955⁽⁷⁴74 Braun-Vallon S. [Strange abnormality of the lateral rectus muscle]. Bull Soc Ophtalmol Fr. 1955;4:212. French.⁾	1	F	8 years	Superotemporal epibulbar	Mass	Excision	Normal LG
Boase/1954⁽⁸8 Boase AJ. Adenoma of the conjunctiva. Br J Ophthalmol. 1954; 38(6):380-2.⁾	1	F	30 years	Medial bulbar conjunctiva	Mass	Excision	LG adenoma
Allende/1954⁽²⁹29 Allende FP. Neurofibromatosis difusa con enorme glándula lagrimal aberrante en posición córneo-conjuntival. Arch Oftalmol B Aires. 1954;29:119-205.⁾	1	M	7 years	Superotemporal bulbar conjunctiva/Cornea	Mass	Biopsy	Normal LGT
Hughes et al/1956⁽⁵⁶56 Hughes WL, Ballen PH. Ectopic lacrimal gland. AMA Arch Opthalmol. 1956;55(2):271-3.⁾	1	F	23 years	Inferotemporal Limbus/Cornea	Mass	Excision	Normal LGT
Mettier/1958⁽⁹9 Mettier SR Jr. Aberrant lacrimal gland. AMA Arch Opthalmol. 1958;60(3):488-91.⁾	1	F	21 years	Inferonasal Cornea/Sclera	Nodular	Excision	Normal LGT
Gördüren/1962⁽¹¹11 Gördüren S. Aberrant Lacrimal Gland Associated with Other Congenital Abnormalities. Br J Ophthalmol. 1962;46(5):277-80.⁾	1	M	6 years	Superotemporal bulbar conjunctiva	Nodular	Excision	Glandular acini + fibrosis
Kessing/1968⁽⁵⁸58 Kessing SV. Ectopic lacrimal gland tissue at the corneal limbus (gland of Manz?). Acta Ophthalmol (Copenh). 1968;46(3):398-403.⁾	1	F	23 years	Inferonasal limbus	Mass	Excision	Lacrimal tissue
Pfaffenbach et al/1971⁽⁶⁶66 Pfaffenbach DD, Green WR. Ectopic lacrimal gland. Int Ophthalmol Clin. 1971;11(3):149-59.⁾	2	F	7 years	Superotemporal and superonasal bulbar conjunctiva/cornea OU	Mass	Biopsy	Lacrimal tissue
	2	M	13 months	Superotemporal bulbar conjunctiva	Mass	Excision	Lacrimal tissue
First et al/1971⁽⁷⁶76 Firat T, Emüler U. Two cases of aberrant lacrimal gland. Ann Ophthalmol. 1971;3(1):50-1.⁾	2	F	11 years	Superotemporal bulbar conjunctiva	Cystic	Excision	Lacrimal tissue
	2	M	31 years	Superotemporal bulbar conjunctiva	Cystic	Excision	Lacrimal tissue
Bullock et al/1986⁽²⁴24 Bullock JD, Fleishman JA, Rosset JS. Lacrimal ductal cysts. Ophthalmology. 1986;93(10):1355-60.⁾	1	M	66 years	Left medial canthus	Cystic	Excision	Multiloculated cyst with islands of LGT
Hered et al/1987⁽²⁶26 Hered RW, Hiles DA. Epibulbar osseous choristoma and ectopic lacrimal gland underlying a dermolipoma. J Pediatr Ophthalmol Strabismus. 1987;24(5):255-8.⁾	1	F	6 months	Superotemporal episcleral	Mass	Excision	Small nests of ectopic LGT underlying a dermolipoma and osteous choristoma
Pokorny et al/1987⁽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.⁾	3	M	16 years	Superotemporal limbus and cornea	Mass	Incisional biopsy	Multiple lobules of well-formed lacrimal tissue, associated with loose fibroadipose tissue, smooth muscle and cartilage
		M	9 months	Inferotemporal limbus and cornea	Nodular	Excisional biopsy	Multiple lobules of well-formed lacrimal tissue, associated with loose fibroadipose tissue
		F	13 years	Superotemporal limbus and cornea	Mass	Partial resection	Well differentiated LG lobules associated with hyaline cartilage and smooth muscle
Kim et al/1989⁽¹1 Kim J, Chi J. Heterotopic lacrimal gland as a component of ocular choristoma. Seoul J Med. 1989;30:97-103.⁾	8	F	5 years	Lateral bulbar conjunctiva	Mass	NS	LGT associated with fat, fibrosis and smooth and skeletal muscle
		F	15 years	Superonasal bulbar conjunctiva	Mass	NS	LGT associated with fat, fibrosis and smooth and skeletal muscle
		F	10 years	Superotemporal bulbar conjunctiva	Mass	NS	LGT associated with pilosebaceous elements fibrosis and smooth and skeletal muscle and nerves
		M	12 years	Lateral bulbar conjunctiva	Mass	NS	LGT associated with sweat glands, pilosebaceous elements fibrosis and smooth and skeletal muscle
		M	19 years	Nasal bulbar conjunctiva	Mass	NS	LGT
		M	14 years	Lateral bulbar conjunctiva	Mass	NS	LGT with pilosebaceous components, fibrosis, smooth muscle and nerves
		M	2 years	Inferotemporal limbus	Mass	NS	Lacrimal tissue with fibrosis
		M	5 years	Lateral bulbar conjunctiva	Mass	NS	LGT associated with fat, fibrosis and smooth and skeletal muscle
Rao et al/1989⁽⁵⁴54 Rao VA, Kwatra V, Puri A. Cyst of ectopic (choristomatous) lacrimal gland. Indian J Ophthalmol. 1989;37(4):189-90.⁾	1	M	40 years	Lateral bulbar conjunctiva	Cystic	Excision	Lacrimal duct cyst
Duncan et al/1998⁽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.⁾	3	F	2 years	Superotemporal limbal	Mass	Excision	Complex choristomas containing lacrimal tissue
		F	9 months	Superotemporal and inferotemporal limbal	Mass	Excision	Complex choristomas containing lacrimal tissue
		M	27 months	Bilateral limbal: Superotemporal, inferotemporal and nasal	Mass	Excision	Complex choristomas containing lacrimal tissue
Patyal et al/2010⁽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.⁾	1	F	13 years	Lateral fornix	Mass	Excision	Pleomorphic adenoma
Raina et al/2010⁽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.⁾	1	M	1 months	Bilateral bulbar conjunctiva. Supero and inferotemporal, superior	Mass	Biospy	Normal glandular tissue
Ferri et al/2013⁽⁵⁵55 Ferri S, Shinder R. Inferomedial dermolipoma with ectopic lacrimal gland. Ophthal Plast Reconstr Surg. 2013;29(2):e43-4.⁾	1	F	25 years	Inferomedial bulbar conjunctiva	Mass	Excisional biopsy	Dermolipoma + LGT
Raven et al/2016⁽⁶¹61 Raven ML, Larson JC, Potter HD. Ectopic lacrimal gland underlying a caruncular nevus. Ophthalmology. 2016;123(5):1136.⁾	1	M	55 years	Caruncle	Mass	Excision	LG acini
Aldossary et al/2018⁽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.⁾	12	12 out of 15 complex choristomas		Temporal in most cases	Mass	Excision	LG acini
Total of cases	46, 34 patients with age and sex specified=16 Males/18 Females

Author/year	Cases	Sex	Age	Location	Clinical appearance	Treatment	Histopathology
Christensen et al/1952⁽²2 Christensen L, Anderson ED. Aberrant intraocular adenomata and epithelization of the anterior chamber. AMA Arch Opthalmol. 1952; 48(1):19-29.⁾	1	M	2 weeks	Iris, ciliary body, sclera	Cystic	Enucleation	Cystic glandular implants in the sclera with ducts communicating with subconjunctival space and cyst of the anterior chamber
Bruce/1952⁽⁶⁹69 Bruce GM. Aberrant glandular tissue in the iris. Trans Am Acad Ophthalmol Otolaryngol. 1952;56(1):47-51.⁾	1	M	2 months	Iris	Mass	Excision	Normal lacrimal tissue
Hunter/1960⁽¹⁰10 Hunter WS. Aberrant intra-ocular lacrimal gland tissue. Br J Ophthalmol. 1960;44(10):619-25.⁾	1	M	9 months	Iris, ciliary bod, cornea	Cystic	Enucleation	Normal LGT
Dallachy/1961⁽³3 Dallachy R. Ectopic Lacrimal Glandular Tissue within the Eyeball. Br J Ophthalmol. 1961;45(12):808-15.⁾	1	F	9 months	Iris, ciliary body	Nodular	Excision followed by enucleation	Normal LGT
Green et al/1967⁽¹²12 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.⁾	1	NS	11 months	iris, ciliary body	Nodular	Enucleation	Normal glandular tissue
Morgan et al/1972⁽¹⁴14 Morgan G, Mushin A. Ectopic intraocular lacrimal gland tissue. Br J Ophthalmol. 1972;56(9):690-4.⁾	1	M	9 months	Ciliary body	Cystic	Enucleation	LGT in the ciliary body. Cyst of LGT within the anterior chamber
Conway et al/1985⁽²⁰20 Conway VH, Brownstein S, Chisholm IA. Lacrimal gland choristoma of the ciliary body. Ophthalmology. 1985;92(3):449-53.⁾	1	F	22 months	Ciliary body, choroid, superficial sclera	Mass/Cystic	Enucleation	LG choristoma
Ghadially et al/1986⁽²³23 Ghadially FN, Chisholm IA, Lalonde JM. Ultrastructure of an intraocular lacrimal gland choristoma. J Submicrosc Cytol. 1986;18(1):189-98.⁾	1	F	11 months	Ciliary body, choroid, limbal surface and anterior sclera	Cystic	Enucleation	Normal lacrimal tissue
O'Donnell et al/1990⁽³⁰30 O'Donnell BA, Martin FJ, Kan AE, Filipic M. Intraocular lacrimal gland choristoma. Aust N Z J Ophthalmol. 1990;18(2):211-3.⁾	1	M	19 months	Iris	Nodular	Biopsy	Normal LG
Shields et al/1995⁽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.⁾	1	F	19 months	Iris, ciliary body	Cystic	Iridocycletomy	Glandular mass with an irregular cystic structure
Rowley et al/1997⁽³⁴34 Rowley SA, Karwatowski WS. Lacrimal gland choristoma of the ciliary body. Arch Ophthalmol. 1997;115(11):1482-3.⁾	1	M	5 years	Iris, epibulbar	Cystic/Mass	Incisional biopsy	Ectopic LGT
Klüppel et al/199⁽³⁸38 Klüppel M, Müller W, Sundmacher R. Lacrimal gland choristoma of the iris. Arch Ophthalmol. 1999;117(1):110-1.⁾	1	F	2 months	Iris	Cystic	Excision	Lacrimal serous gland tissue
Shields et al/2000⁽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.⁾	1	M	At birth	Iris, ciliary body	Mass	Iridocyclectomy	Benign LGT
Cho et al/2006⁽⁴⁵45 Cho HY, Sa HS, Kang SW. Lacrimal gland choristoma of the ciliary body. Jpn J Ophthalmol. 2006;50(2):100-2.⁾	1	F	10 months	Ciliary body	Cystic/mass	Partial sclerocyclectomy	Normal LG with cystic spaces
Jung et al/2006⁽⁴⁶46 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.⁾	1	F	2 months	Choroid	Cystic	Incisional biopsy	Ectopic lacrimal tissue
Kobrin et al/2007⁽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.⁾	1	M	6 years	Iris	Mass	FNAB	Features characteristic of a benign glandular epithelium consistent with LG acini
Ranganathan et al/2010⁽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.⁾	1	F	6 weeks	Iris/ciliary body	Mass/Multi cystic	Enucleation	LGT with dilated ducts
Tauziède-Espariat et al/2016⁽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.⁾	1	F	12 months	Ciliary body	Mass	Iridocyclectomy, enucleation	LGT
Total of cases	18, 8 Males/9 Females/1 Not stated

Author/year	Cases	Sex	Age (yrs)	Location	Clinical findings	Imaging	Treatment	Histopathology	Outcome
Jain/1964⁽⁷³73 Jain BS. Pleomorphic Adenoma. Report of Two Uncommon Cases. Am J Ophthalmol. 1964;57(4):672-5.⁾	1	M	14	Superotemporal extraconal	Proptosis	X-Rays	Excision	Pleomorphic adenoma	NS
Boudet et al/1964⁽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.⁾	1	F	61	Inferotemporal intraconal	Proptosis/diplopia	Orbital angiography	Excision	Pleomorphic adenoma	Cured/diplopia on downgaze
Bech et al/1965⁽⁵⁷57 Bech K, Jensen OA. Mixed Tumor of the Lower Orbital Region. Arch Ophthalmol. 1965;74(2):226-8.⁾	1	M	50	Inferonasal orbital rim	Palpable mass	None	Excision	Pleomorphic adenoma	Cured
Green et al/1967⁽¹²12 Green WR, Zimmerman LE. Ectopic lacrimal gland tissue. Report of eight cases with orbital involvement. Arch Ophthalmol. 1967; 78(3):318-27.⁾	8	F	45	Extraconal, medial wall	Proptosis, vertical dystopia	None	Excision	Atrophic lacrimal tissue with moderate chronic inflammatory infiltrate	Favorable
		M	44	Extraconal lateral wall. Attached to the orbital lobe of the lacrimal gland	Proptosis, diplopia	X-ray	Excision	Atrophic lacrimal tissue with fibrous stroma	Persistent diplopia
		F	16	Extraconal superonasal. Normal lacrimal tissue/Adenocarcinoma	Proptosis, diplopia, palpable mass	None	2 Biopsies	Autopsy after accidental death: ectopic LGT with cysts, areas of adenocarcinoma	Accidental car death
		F	42	Extraconal inferotemporal and orbital floor l	Retrobulbar pain, limitation of motility, Proptosis	None	Excision	Lobules of glandular tissue with lymphocytic infiltration	NS
		F	11	Beneath the superior orbital rim between the LG and the trochlea. Second biopsy: intraconal	Ptosis, proptosis, limitation of motility. Palpable mass beneath the superior orbital rim	X-ray	3 Biopsies	Intraconal lesion with lobules of LG with acute and chronic inflammation	Vision loss, proptosis, eye motility limitation, corneal clouding
		M	15	Intraconal superotemporal	Proptosis, vision loss	US	Excision	Glandular tissue lymphocytic infiltration, fibroadipose connective tissue	Optic nerve atrophy
		M	46	Apical extraconal tissue from a large tumor involving the LG	Proptosis, slightly reduced acuity	Arteriography	2 Biopsies	LGT with cystic areas	Proptosis reduction
		M	11	Extraconal posterior	Proptosis, vision loss, choroidal folds	US	Biopsy	Lobules of LGT with moderate atrophy and chronic inflammatory infiltrate	Residual exophthalmos
Baldrige et al/1970⁽¹³13 Baldridge M. Aberrant lacrimal gland in the orbit. Arch Ophthalmol. 1970;84(6):758-9.⁾	1	M	18	Extraconal lateral from the rim to the apex	Proptosis, esotropia	X-ray	Excision	Normal lacrimal gland tissue	Residual esotropia
Zilkha/1972⁽¹⁵15 Zilkha A. Ectopic lacrimal gland and exophthalmos. Case report with emphasis on value of orbital venography. Radiology. 1972; 103(2):275-6.⁾	1	F	46	Extraconal lateral	Proptosis, diplopia, adduction limitation	X-ray, carotid angiography, orbital venography	Excision	Normal LGT with lymphocytic infiltration	Residual proptosis, diplopia improvement
Mindlin et al/1977⁽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.⁾	1	M	21	Extraconal medial	Proptosis, choroidal folds	X-ray, US, orbital arteriography	Excision	Pleomorphic adenoma	Residual macular changes
Jacobs et al/1977(77)	1	F	69	Intraconal inferolateral	Proptosis, visual loss, diplopia	CT	Excision	Normal GLT	Diplopia
Müeller et al/1979⁽¹⁶16 Mueller EC, Borit A. Aberrant lacrimal gland and pleomorphic adenoma within the muscle cone. Ann Ophthalmol. 1979;11(4):661-3.⁾	1	M	31	Intraconal lateral to the optic nerve	Proptosis, choroidal folds, slight visual acuity loss	X-ray	Excision	Normal glandular tissue + areas of pleomorphic adenoma	Cured
Rush et al/1981⁽¹⁷17 Rush A, Leone CR Jr. Ectopic lacrimal gland cyst of the orbit. Am J Ophthalmol. 1981;92(2):198-201.⁾	1	M	7	Extraconal superonasal	Fullness below the superior medial orbital rim	CT	Excision	Cyst containing normal LG	NS
Appel et al/1982⁽¹⁸18 Appel N, Som PM. Ectopic orbital lacrimal gland tissue. J Comput Assist Tomogr. 1982;6(5):1010-2.⁾	1	M	63	Extra-and intraconal lateral	Proptosis, optic atrophy	CT	Biopsy	Normal LGT + varix	NS
Margo et al/1985⁽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.⁾	1	M	28	Extra- and intraconal superolateral	Proptosis	CT	Incisional biopsy	Lacrimal tissue adjacent to the lacrimal fossa and deep in the orbit	Favorable
V. Domarus/1987⁽²⁵25 von Domarus H. A lacrimal gland cyst in the orbit. J Craniomaxillofac Surg. 1987;15(2):106-9.⁾	1	F	2	Extraconal lateral	Proptosis, medial dystopia	CT	Excision	LG cyst	Cured
Guy et al/1989⁽⁶⁸68 Guy JR, Quisling RG. Ectopic lacrimal gland presenting as an orbital mass in childhood. AJNR Am J Neuroradiol. 1989;10(5 Suppl):S92.⁾	2	M	1.3	Intraconal (superior, lateral and medial)	Proptosis	CT	Biopsy	Lacrimal gland tissue	NS
	2	F	0.5	Intraconal	Proptosis, exposure keratitis	CT	Biopsy	Lacrimal gland tissue	NS
Bocato et al/1991⁽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.⁾	1	F	68	Extraconal superolateral	Proptosis	CT/ US	FNAB	Lobules of ectopic lacrimal gland	Stable
Sakurai et al/1997⁽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.⁾	1	M	9	Extraconal superior	Proptosis, Restricted eye motility, Increased intraocular pressure	CT/ MRI/SPECT/ PET scan	Incisional biopsy	Normal lacrimal gland tissue	Stable
Shields et al/1997⁽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.⁾	1	M	26	Extraconal superonasal	Subcutaneous swelling superonasal	CT/MRI	Biopsy	Adenoid cystic carcinoma	Exenteration
West et al/1997⁽³⁷37 West JA, Drewe RH, McNab AA. Atypical choristomatous cysts of the orbit. Aust N Z J Ophthalmol. 1997;25(2):117-23.⁾	1	F	20	Inferomedial	Hypertropia	CT	Excision	Cyst with lacrimal gland tissue	Cured
Suneetha et al/1999⁽⁶⁷67 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.⁾	1	M	12	Extraconal Lateral rectus/ periorbita	Adduction Limitation	CT/US	Excision	Nodular lesion with lacrimal gland tissue	Cured
Kao et al/2000⁽783⁾	1	M	33	Extraconal anterior infero-medial	None	CT/US	Excision	Dacryops	Cured
Solaroglu et al/2005⁽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.⁾	1	M	4	Extraconal lateral	Proptosis, orbital pain. Restricted eye motility	CT	Excision	LG lobules	Favorable
Yüceer et al/2008⁽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.⁾	1	F	5	Intraconal lateral	Proptosis, Restricted eye mobility	CT/MRI	Incisional biopsy by transcranial approach	Normal LGT	Lateral rectus paresis
Lenzi et al/2012⁽⁵¹51 Lenzi R, Casani AP, Sellari-Franceschini S. Ectopic intraorbital lacrimal ductal cyst: a case report. Orbit. 2012;31(5):350-1.⁾	1	M	61	Intraconal adherent to medial rectus muscle	Diplopia		Excision	Cyst of ductal origin	Cured
Braich et al/2014⁽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.⁾	1	F	1.1	Extraconal inferonasal	Cystic lesion adduction limitation Exotropia	MRI	Excision	Ectopic LGT with cysts	Persistent adduction limitation
Pujari et al/2015⁽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.⁾	1	F	15	Cyst adherent to the Inferior rectus muscle	Progressive proptosis and diplopia	US/CT	Excision	LG cyst	Cured
Misra et al/2016⁽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.⁾	1	M	60	Superolateral orbit	Proptosis	CT	Excision	Pleomorphic adenoma	Cured
Total of case	34, 20 Males/14 Females

Author/year	Cases	Sex	Age (years)	Location	Clinical findings	Imaging	Treatment	Histopathology	Outcome
Evans/1964⁽⁷²72 Evans PA. Adenoma in an Eyelid. Br J Ophthalmol. 1964;48(4):234-5.⁾	1	F	50	Lower eyelid	Firm mass on nasal aspect of the lower lid	None	Excision	Pleomorphic adenoma	NS
Jain/1964⁽⁷³73 Jain BS. Pleomorphic Adenoma. Report of Two Uncommon Cases. Am J Ophthalmol. 1964;57(4):672-5.⁾	1	NS	48	In the middle of the upper eyelid	Nodular mass/Mechanical ptosis	None	Excision	Pleomorphic adenoma	NS
Gordon et al/1991⁽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.⁾	1	F	2	Upper eyelid	Ectopic cilia and lacrimal fistula	None	Excision	Lobules of LG associated with ectopic cilia	Cured
Sim et al/1999⁽³⁹39 Sim KT, Sullivan SC. Aberrant lacrimal gland within the tarsal plate presenting as hyperlacrimation. Eye (Lond). 1999;13(Pt 4):591-3.⁾	1	F	15	Upper eyelid	Watering fistula	Gallium-67 uptake	Excision	Intratarsal lacrimal tissue	Cured
Lee et al/2002⁽⁴¹41 Lee WR, Aitken DA, Kirkness CM. Diffuse hyperplasia of intratarsal ectopic lacrimal gland tissue. Arch Ophthalmol. 2002;120(12):1748-52.⁾	1	F	58	Upper eyelid OU	Nodular tarsal areas	None	Tarsectomy	Lacrimal tissue	Cured
Alsuhaibani/2012⁽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.⁾	1	F	75	Upper eyelid	Upper eyelid swelling	CT	Excision	Pleomorphic adenoma	Cured
Obi et al/2013⁽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.⁾	3	M	12	Medial lower eyelid	Mass	None	Excision	Pleomorphic adenoma	Cured
		M	52	Medial lower eyelid	Nodular lesion	None	Excision	Pleomorphic adenoma	Cured
		M	64	Central lower eyelid	Mass	None	Excision	Pleomorphic adenoma	Cured
Wajda et al/2019⁽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.⁾	1	F	35	Upper eyelid	Mass	CT	Excision	Pleomorphic adenoma	NS
Total	10, 4 Males/5 Females/1 Not stated

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[1] Corresponding author: Roberto Murillo Limongi. E-mail: rmurillousp@hotmail.com