Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence

Abstract

BackgroundCongenital ranulas seldom occur, with bilateral presentation and prenatal diagnosis reported very rarely. We believe this is the first reported case of a neonate with a antenally diagnosed massive congenital ranula, who went on to develop a non-contiguous contralateral ranula, both contributing to obstruction in a complex paediatric airway.Case reportA female neonate was born to a non-primagravid mother via a planned elective caesarean section due to a lower facial defect and oral cyst. Antenatal aspiration of the pseudocyst was performed under ultrasound guidance with limited success. In the immediate post-natal period a poor airway was observed and the cyst was subsequently marsupialised. With the development of macroglossia secondary to oedema and tongue base collapse the airway was secured through surgical tracheostomy. A subsequent ultrasound scan revealed the presence of a second solitary cystic mass on the contralateral side. After careful excision of the contralateral pseudocyst, tongue function improved, with the resolution of a safe airway which permitted successful decannulation. A planned definitive procedure antenatally did not result in the anticipated improvement in function. However the subsequent development of a second non-contiguous pseudocyst and further surgical management resulted in a safe airway, improved masticator function and the ability to thrive.ConclusionsThe prenatal diagnosis of congenital ranulas have been seldom reported, with no reported cases of contralateral occurrence and airway obstruction from an intraoral ranula. This rare case highlights the need for a well considered contingency plan when surgery is required for a neonatal airway at risk.

Cervicofacial Actinomycosis in the Pediatric Population: Presentation and Management

“…Given the location of the dermoid cyst, there were also concerns for difficulties with speech, swallowing, mastication, and airway obstruction. 19,20 The growth of Actinomyces spp directed targeted amoxicillin therapy. Despite antibiotic treatment and multiple aspirations, the lesion continued to enlarge.…”

Prenatal Diagnosis and Intrapartum Management of Congenital Ranula

“…However, if the lesion persists after an observation time of a few months, extraction is suggested [12]. Although sclerotherapy and cryotherapy have both demonstrated success in some studies, most treatment approaches are based on excision of the ranula with or without the sublingual gland [13,14].…”

Ranula: Current Concept of Pathophysiologic Basis and Surgical Management Options

“…Ranulas diagnosed on routine antenatal large enough to warrant intrauterine decompression for a safe labour and delivery have been reported [ 12 ]. This has suggested a genetic basis for ranulas of early life including plunging ranulas which were found to be more frequent in the ethnic groups of the Maori and the Pacific Island Polynesians [ 13 , 14 ].…”