LAUSR

PURPOSE Since the original description of "dacryadenoma" by Jakobiec and colleagues, the data on this unusual epibulbar lacrimal gland lesion remain sparse. The aim of this study was to characterize clinically, morphologically, and immunohistochemically this isolated epibulbar lacrimal gland lesion. DESIGN Retrospective observational case series. METHODS Institutional pathology records between 2000 and 2019 were searched for all cases of isolated epibulbar lacrimal gland lesions. Tissue from three normal lacrimal glands and one complex choristoma were included for comparative analysis. Clinical, histopathologic, and immunohistochemical findings were recorded. RESULTS Four patients with isolated epibulbar lacrimal gland lesions, 2 males and 2 females, with a median age of 18 years (range, 12-57) were identified. All patients presented with recent onset of unilateral pink-to-orange, well-circumscribed subepithelial juxtaforniceal (3/4,75%) or nasal (1/4,25%) bulbar conjunctival nodules, which were asymptomatic (3/4,75%) or associated with foreign body sensation (1/4,25%). When compared to the normal lacrimal gland and complex choristoma, all isolated epibulbar lacrimal gland lesions were composed predominantly of variably dilated, branching tubular structures with pseudo-apocrine snouts and either totally absent (2/2,50%) or rare (2/2,50%) ducts and rare acinar zymogen granules (3/4,75%). CONCLUSION Our study confirms that a subset of isolated epibulbar lacrimal gland lesions differ morphologically and immunohistochemically from normal lacrimal gland tissue and the lacrimal gland in a complex choristoma. These differences range from subtle to overt, suggesting that isolated epibulbar lacrimal gland lesions may have originated from precursor cellular elements indigenous to the conjunctiva (hamartia) and grew into disorganized lacrimal gland tissue.