PC2161: Bilateral Dacryocystoceles Atypically Presenting as Respiratory Distress and Stertor

Introduction: Respiratory distress is the most common cause of admission to higher levels of care in both term and preterm infants. Here we describe a case with intermittent respiratory distress and stertorous breath sounds admitted to the NICU and ultimately diagnosed with bilateral congenital dacryocystoceles (CDC), an incredibly rare diagnosis with an atypical presentation.

Case Description: Our patient is a newborn female born at 36 weeks and 6 days gestation via spontaneous vaginal delivery to a multiparous mother. Her prenatal course was unremarkable with negative serologies including group-B-Strep status, and delivery was uncomplicated without advanced resuscitation. At six hours of life (HOL) parents relayed concern for grunting and difficulty with latch during feeds. Initial assessment noted significant nasal congestion which improved with suctioning. The remainder of the evaluation was unremarkable and she was well-appearing without desaturations, tachypnea, or increased work of breathing. She remained with her parents for routine newborn care. At 16 HOL, she was reevaluated for worsening respiratory distress and noted to have subcostal and intercostal retractions, nasal flaring, head bobbing, and grunting. Auscultation was significant for biphasic stertorous breathing and mildly decreased aeration in the lungs, but without adventitious breath sounds suggestive of parenchymal disease. After placement on monitors, she was noted to have two brief desaturations to 60%, one while feeding and one while taking a pacifier. She was then transferred to the NICU where a nasogastric tube was passed successfully through bilateral nares and started on low-flow nasal cannula. Further workup consisting of arterial blood gas, complete blood count, blood culture, and chest x-ray were unremarkable. Given the presence of biphasic low pitched stertorous breath sounds, ENT was consulted. Flexible nasopharyngoscopy revealed bilateral inferior meatal bluish cysts located at Hasner's valve inferior and lateral to the inferior turbinates (Figure 1). Non-contrasted CT face further supported the diagnosis of CDCs without mass effect, inflammation, or involvement of adjacent orbits and intraorbital contents (Figure 2). The decision was made to trial a conservative management approach with warm compresses and ocular massage. She then developed epiphora thought to be secondary to rupture during her scope. Subsequently, her respiratory symptoms resolved and she discharged home after observation without further respiratory distress or concerns for infection.

Discussion: CDCs are an incredibly rare nasolacrimal duct obstruction with the classical presentation being unilateral bluish swelling located at the medial canthal area and epiphora. We present a case of bilateral CDCs with an atypical presentation ultimately diagnosed by localizing the pathology based on biphasic stertorous breath sounds.

Conclusion: This case highlights the appropriate use of adventitious breath sounds to localize pathology and the need for a multi-disciplinary team when evaluating upper airway anomalies.