A rare case of bilateral congenital dacryocele.

Sir,

Congenital dacryocele is an uncommon congenital anomaly characterized by bluish gray cystic swelling of lacrimal sac seen just below the medial canthal tendon.[1] It is also referred to as congenital dacryocystocele, amniotocele, and mucocele. The condition is usually unilateral and has a female preponderance. It is typically present at birth and occurs due to concomitant obstruction of lacrimal system below the lacrimal sac and at the level of canalicular opening in the sac. There occurs entrapment of fluid nature of which is debatable. Some reports point to the source as amniotic fluid; however, some studies propose it to be mucus produced by intraluminal goblet cells.[2] Diagnosis can be made on the basis of time of onset, appearance, ultrasound, computed tomography, or magnetic resonance imaging to differentiate it from meningoencephalocele, capillary hemangioma, and dermoid cyst. The swelling resolves spontaneously in majority of cases with conservative management, but when self-resolution fails to occur, early probing is warranted to prevent risk of dacryocystitis.

The authors would like to bring to notice a rare case of bilateral congenital dacryocele encountered in our clinical practice. A 5-day-old female child born at term was brought to the OPD with bilateral tense blue cystic swelling below the medial canthus [Figure 1]. The swelling was more marked on the left side. There was no history of watering and the swelling appeared to be nontender. An otorhinolaryngologist opinion was sought and intranasal extension of swelling was ruled out. Diagnosis of uncomplicated bilateral congenital dacryocele was made and in the absence of evidence of respiratory distress child was initially advised conservative management in form of compressive massage. One week later, there was complete resolution of the swelling [Figure 2] and the child remained asymptomatic even at 6 months.

Figure 1

Bilateral congenital dacryocele, more pronounced on the left side

Figure 2

One week later, complete resolution of swelling following conservative management

Cases of bilateral dacryocele are rarely reported in literature.[23] The few reported bilateral cases have demonstrated coexistent intranasal mucocele requiring surgical management. In such cases, respiratory distress occurs especially during feeding due to nasal obstruction, and therefore a thorough nasal check-up, rhinoscopy particularly of inferior nasal meatus is warranted. Our case had a rare presentation of bilateral involvement in the absence of intranasal extension.

Several case studies have shown successful treatment of uncomplicated congenital dacryocele with conservative management alone,[4] which includes antibiotics, warm compresses, and sac massage. Since tear production does not start immediately after birth, children usually do not have epiphora but complications in form of dacryocystitis, preseptal cellulitis, intranasal involvement have been seen in as high as 60% of cases requiring hospitalization and subsequent surgical management.[2] Probing alone is not sufficient and intubation to ensure prolonged patency of lacrimal system is needed. In cases with intranasal extension, early surgical intervention is mandatory and includes marsupialization of the cyst to prevent its recurrence.

Because of strong association of bilateral dacryocele with intranasal mucocele and a high rate of complications we recommend in all cases of congenital dacryocele, a prompt nasal examination and close follow-up to aid in appropriate decision-making and management.

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