Semilobar Holoprosencephaly with Congenital Oropharyngeal Stenosis in a Term Neonate

Abstract Background Holoprosencephaly (HPE) is often accompanied by a deficit in midline facial development; however, congenital oropharyngeal stenosis in neonates with HPE has not been reported before. We describe a case of a neonate with prenatally diagnosed semilobar HPE accompanied...

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Bibliographic Details
Main Authors: Kenji Hishikawa, Hideshi Fujinaga, Chie Nagata, Masataka Higuchi, Yushi Ito
Format: Article
Language:English
Published: Thieme Medical Publishers, Inc. 2015-04-01
Series:American Journal of Perinatology Reports
Subjects:
Online Access:http://www.thieme-connect.de/DOI/DOI?10.1055/s-0035-1548725
Description
Summary:Abstract Background Holoprosencephaly (HPE) is often accompanied by a deficit in midline facial development; however, congenital oropharyngeal stenosis in neonates with HPE has not been reported before. We describe a case of a neonate with prenatally diagnosed semilobar HPE accompanied by congenital oropharyngeal stenosis. Case Report The patient was born at 39 weeks of gestation and developed dyspnea shortly after. Laryngoscopic test revealed oropharyngeal stenosis. Nasal continuous positive airway pressure, high-flow nasal cannula, and nasopharyngeal airway did not resolve her dyspnea; tracheostomy was required. Conclusion Neonates with HPE might be at higher risk of pharyngeal stenosis because of the functional and/or anatomical abnormalities. In the case of dyspnea in neonates with HPE, laryngoscopic evaluation should be considered.
ISSN:2157-6998
2157-7005