Lacrimal Intrasaccal Cyst.

Abstract

Dear Editor, Two cases with epiphora and medial canthal mass showed a cyst within the sac that compressed the drainage system. We describe a new sac anomaly of intrasaccal cyst, which has not yet been reported. A 31-year-old woman was referred for epiphora in her left eye for 17 years. She also presented with a 10-year history of a palpable mass below the medial canthal tendon. The system was patent on irrigation. In computed tomographic (CT) dacryocystography (DCG), a soft ovoid mass of 25 mm was found in the sac fossa, and the passage was compressed and deviated anteriorly (Fig. 1A and 1B). DCG revealed a patent but laterally deviated drainage passage (Fig. 1C). After subciliary incision, an enlarged sac was identified and dissected. After sac incision, serous fluid gushed out from a blind sac on the periosteal side of the sac. The internal common punctum and the lateral sac wall were identified after excision of the lateral wall of the cyst. The cyst and the sac shared a common wall along the entire length between them. External dacryocystorhinostomy was performed after cyst excision. Histopathologic study revealed a cyst wall lined with pseudostratified columnar and cuboidal epithelium, supporting the finding of the sac mucosa (Fig. 1D). The patient has now been free of symptoms for five years. Fig. 1 (A,B) Computed tomographic dacryocystography (DCG) demonstrates an ovoid soft tissue mass (arrowhead) anterior to the passage (arrow) in the sac fossa (case 1). (C) DCG reveals a filling defect in the sac without passage disturbance ... A 39-year-old man was referred for epiphora of two-year duration. A soft mass was found just above the left medial canthal tendon. In the CT-DCG, a 16 mm-sized soft tissue mass was identified with pressure remodeling of the adjacent bone (Fig. 1E). The DCG revealed a deviated and blocked sac on the lateral side of a space occupying filling defect (Fig. 1F). The lateral wall of the cystic mass shared a common wall with the sac. On histopathologic examination of the common wall, both sides showed the same features of pseudostratified columnar epithelium with squamous dysplasia. The patient has now been free of symptoms for five years. A intrasaccal cyst is regarded to be a different clinical entity from a sac cyst or a diverticulum because the latter anomalies are located outside of the sac. Lacrimal diverticulum forms a mass attached and connected to the sac with a narrow neck [1,2]. Lacrimal sac cyst has been described as a separated orbital cystic mass having sac epithelium, which is near to or remote from the sac [1,2]. Embryologically, the nasolacrimal apparatus develops from the surface epithelial core and canalization occurs at three months gestation throughout the length of the nasolacrimal apparatus [3]. Though an acquired condition from sac inflammation cannot be excluded, intrasaccal cyst can be considered to develop from an extra core of cells budding off the original core like other congenital drainage system anomalies. Takahashi et al. [4] first described the sac septum. They incidentally identified it in a cross section of the sac, which shared the same epithelial lining with the sac. It was also regarded to result from developmental anomalies in embryogenesis. Although there was no description of proximal or distal features of the septum, the case might represent an asymptomatic intrasaccal cyst without cyst expansion. Mansour et al. [5] reported a supernumerary blind sac that was connected to the supernumerary puncta and canaliculi, resulting in compression of the drainage pathway. The entity was different from our cases because there was no supernumerary puncta or canaliculi connected to the cyst. Lacrimal intrasaccal cysts can demonstrate different clinical courses such as symptom-free if it is not expansile or cystic mass formation with or without nasolacrimal duct obstruction. Presenting symptoms cannot differentiate the lesion and imaging studies of CT-DCG and DCG are mandatory for diagnosis. If a thick membrane is found between the lacrimal sac cavity and internal punctum during dacryocystorhinostomy, possibility of intrasaccal cyst should be considered. Endonasal marsupialization or dacryocystorhinostomy could be one of the treatment options according to the obstructive signs. Recognition of this abnormal configuration of the sac might be helpful in differential diagnosis and treatment of patients with medial canthal mass with epiphora, though it is a rare entity.