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

BACKGROUND: Congenital 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 contr...

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Autores principales: George, Manish M., Mirza, Omar, Solanki, Kohmal, Goswamy, Jay, Rothera, Michael P.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4430706/
https://www.ncbi.nlm.nih.gov/pubmed/25984300
http://dx.doi.org/10.1016/j.amsu.2015.04.006
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author George, Manish M.
Mirza, Omar
Solanki, Kohmal
Goswamy, Jay
Rothera, Michael P.
author_facet George, Manish M.
Mirza, Omar
Solanki, Kohmal
Goswamy, Jay
Rothera, Michael P.
author_sort George, Manish M.
collection PubMed
description BACKGROUND: Congenital 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 REPORT: A 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. CONCLUSIONS: The 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.
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spelling pubmed-44307062015-05-15 Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence George, Manish M. Mirza, Omar Solanki, Kohmal Goswamy, Jay Rothera, Michael P. Ann Med Surg (Lond) Case Report BACKGROUND: Congenital 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 REPORT: A 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. CONCLUSIONS: The 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. Elsevier 2015-04-14 /pmc/articles/PMC4430706/ /pubmed/25984300 http://dx.doi.org/10.1016/j.amsu.2015.04.006 Text en © 2015 Published by Elsevier Ltd on behalf of IJS Publishing Group Limited. https://creativecommons.org/licenses/by-nc-nd/4.0/This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License (https://creativecommons.org/licenses/by-nc-nd/4.0/) , which allows reusers to copy and distribute the material in any medium or format in unadapted form only, for noncommercial purposes only, and only so long as attribution is given to the creator.
spellingShingle Case Report
George, Manish M.
Mirza, Omar
Solanki, Kohmal
Goswamy, Jay
Rothera, Michael P.
Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title_full Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title_fullStr Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title_full_unstemmed Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title_short Serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
title_sort serious neonatal airway obstruction with massive congenital sublingual ranula and contralateral occurrence
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4430706/
https://www.ncbi.nlm.nih.gov/pubmed/25984300
http://dx.doi.org/10.1016/j.amsu.2015.04.006
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