Semilobar Holoprosencephaly with Congenital Oropharyngeal Stenosis in a Term Neonate.
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.
Division of Neonatology, National Center for Child Health and Development, Tokyo, Japan.
Kenji
K
Hishikawa
Division of Neonatology, National Center for Child Health and Development, Tokyo, Japan.
Hideshi
H
Fujinaga
Division of Education for Clinical Research, National Center for Child Health and Development, Tokyo, Japan.
Chie
C
Nagata
Division of Pulmonology, Department of Medical Subspecialties, National Center for Child Health and Development, Tokyo, Japan.
Masataka
M
Higuchi
Division of Neonatology, National Center for Child Health and Development, Tokyo, Japan.
Yushi
Y
Ito
congenital pharyngeal stenosis; holoprosencephaly; oropharyngeal stenosis