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Case Report/Case Series |

Dacryops:  A Series of 5 Cases and a Proposed Pathogenesis

Kay Lam, MD1,2; Seymour Brownstein, MD1,2; David R. Jordan, MD1; Andre Jastrzebski, MD1,2
[+] Author Affiliations
1Department of Ophthalmology, University of Ottawa, The Ottawa Hospital, The Ottawa Hospital Research Institute, Ottawa, Ontario, Canada
2Department of Pathology, University of Ottawa, The Ottawa Hospital, The Ottawa Hospital Research Institute, Ottawa, Ontario, Canada
JAMA Ophthalmol. 2013;131(7):929-932. doi:10.1001/jamaophthalmol.2013.1885.
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Importance  Lacrimal gland ductal cysts (dacryops) are uncommon, occurring anywhere that lacrimal gland tissue is present. While dacryops has long been recognized, its pathogenesis has not been well established.

Observations  Five cases of dacryops were identified at the Ottawa Hospital over a 3-year period (2009-2012). Histopathological examination showed immunoreactivity to IgA on the luminal surface of the epithelial ductal cyst and mild chronic inflammation in the underlying stroma or lacrimal gland.

Conclusions and Relevance  Our results suggest that a multifactorial mechanism leads to dacryops including chronic inflammation, an immune response, and IgA hypersecretion with an osmotic effect, all contributing to the cyst formation. Further research on the pathogenesis is recommended.

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Figure 1.
Photographs and Computed Tomographic Images of the 5 Cases

A, A clear, cystic mass in the superotemporal fornix of the right eye of case 1. B, In case 2, a fluid-filled cyst in the right superotemporal fornix. C, A mass (arrow) in the left upper eyelid temporally in case 3. D, In case 3, the orbital lesion was cystic, with an attached periosteum. E, A computed tomographic scan of case 4 shows a hypodense, well-delineated cyst in the area of the left lacrimal gland that measured 20 × 12 × 22 mm and displaced the globe medially. F, A computed tomographic scan of case 53 reveals a large, cystic mass in the left inferior fornix causing enophthalmos of the globe.

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Figure 2.
Stainings of 5 Dacryops Cases

A, In case 2, a large cystic lesion is lined by double-layered, cuboidal epithelium with numerous apical snouts and with adjacent lacrimal gland tissue and subepithelial mild inflammatory infiltrates (hematoxylin eosin, original magnification ×160). B, In case 3, staining for IgA was positive along the entire inner surface of the cystic epithelium (original magnification ×400). C, In case 1, IgA stains strongly positive on the luminal surface of the inner epithelial cells, which showed typical apocrine features with several apical snouts (original magnification ×400). D, In contrast in case 1, IgG does not stain the epithelium (original magnification ×400). E, Cystic lining in case 4 shows transition from bilayered, ductular epithelium on the right to a stratified squamous, conjunctival-like epithelium on the left (hematoxylin eosin, original magnification ×250). F, In case 4, staining for IgA was strongly positive along the surface of the double-layer epithelium on the right and only weakly positive along the stratified squamous epithelium on the left (original magnification ×250). G, Transmission electron microscopy in case 53 demonstrates the bilayer epithelium with apocrine snouts on the surface. H, Electron-dense secretory granules in case 53 predominantly within the apical portion of the surface cells.

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