Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature

Agnathia is a rare disease characterized by the absence of a mandible. Few cases of prenatally diagnosed isolated agnathia have been reported. We present a case report and review of the literature of prenatally diagnosed agnathia. A 38-year-old woman (gravida 0, para 0) was referred to our hospital...

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Main Authors: Kazuhiro Kajiwara, Tomohiro Tanemoto, Chie Nagata, Aikou Okamoto
Format: Article
Language:English
Published: Wiley 2016-01-01
Series:Case Reports in Obstetrics and Gynecology
Online Access:http://dx.doi.org/10.1155/2016/8512351
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author Kazuhiro Kajiwara
Tomohiro Tanemoto
Chie Nagata
Aikou Okamoto
author_facet Kazuhiro Kajiwara
Tomohiro Tanemoto
Chie Nagata
Aikou Okamoto
author_sort Kazuhiro Kajiwara
collection DOAJ
description Agnathia is a rare disease characterized by the absence of a mandible. Few cases of prenatally diagnosed isolated agnathia have been reported. We present a case report and review of the literature of prenatally diagnosed agnathia. A 38-year-old woman (gravida 0, para 0) was referred to our hospital at 28 weeks and 3 days of gestation for fetal evaluation because of polyhydramnios and suspected facial anomalies. Three-dimensional ultrasonography and MRI indicated agnathia. Premature rupture of the membranes occurred before the parents could reach a decision on the postnatal treatment. We performed emergency cesarean section on the second day of the 33rd week of gestation. The neonate was deemed nonresuscitable and he died of airway obstruction shortly after birth. Because agnathia is associated with very poor prognosis, accurate prenatal diagnosis and detailed counseling should be promptly provided before unexpected delivery to the parents for the determination of postnatal treatment.
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institution Kabale University
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language English
publishDate 2016-01-01
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series Case Reports in Obstetrics and Gynecology
spelling doaj-art-ba115c8e22e94598b4aa5f02501ef7f52025-02-03T01:10:00ZengWileyCase Reports in Obstetrics and Gynecology2090-66842090-66922016-01-01201610.1155/2016/85123518512351Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the LiteratureKazuhiro Kajiwara0Tomohiro Tanemoto1Chie Nagata2Aikou Okamoto3The Jikei University School of Medicine, Nishishimbashi 25-83-3, Minato-ku, Tokyo 105-8461, JapanThe Jikei University School of Medicine, Nishishimbashi 25-83-3, Minato-ku, Tokyo 105-8461, JapanNational Center for Child Health and Development, 2-10-1 Okura, Setagaya-ku, Tokyo 157-8535, JapanThe Jikei University School of Medicine, Nishishimbashi 25-83-3, Minato-ku, Tokyo 105-8461, JapanAgnathia is a rare disease characterized by the absence of a mandible. Few cases of prenatally diagnosed isolated agnathia have been reported. We present a case report and review of the literature of prenatally diagnosed agnathia. A 38-year-old woman (gravida 0, para 0) was referred to our hospital at 28 weeks and 3 days of gestation for fetal evaluation because of polyhydramnios and suspected facial anomalies. Three-dimensional ultrasonography and MRI indicated agnathia. Premature rupture of the membranes occurred before the parents could reach a decision on the postnatal treatment. We performed emergency cesarean section on the second day of the 33rd week of gestation. The neonate was deemed nonresuscitable and he died of airway obstruction shortly after birth. Because agnathia is associated with very poor prognosis, accurate prenatal diagnosis and detailed counseling should be promptly provided before unexpected delivery to the parents for the determination of postnatal treatment.http://dx.doi.org/10.1155/2016/8512351
spellingShingle Kazuhiro Kajiwara
Tomohiro Tanemoto
Chie Nagata
Aikou Okamoto
Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
Case Reports in Obstetrics and Gynecology
title Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
title_full Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
title_fullStr Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
title_full_unstemmed Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
title_short Prenatal Diagnosis of Isolated Agnathia-Otocephaly: A Case Report and Review of the Literature
title_sort prenatal diagnosis of isolated agnathia otocephaly a case report and review of the literature
url http://dx.doi.org/10.1155/2016/8512351
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AT chienagata prenataldiagnosisofisolatedagnathiaotocephalyacasereportandreviewoftheliterature
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