Document Detail


A case of congenital high airway obstruction syndrome managed by ex utero intrapartum treatment: case report and review of the literature.
MedLine Citation:
PMID:  17372858     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Congenital high airway obstruction syndrome (CHAOS) has been reported to be fatal. Ten cases of CHAOS that underwent ex utero intrapartum treatment (EXIT) procedure to secure the fetal airway have been reported. A 36-year-old woman (gravida 3, para 2) was referred to our hospital at 22 weeks of gestation. Sonography revealed large echogenic lungs, flattened diaphragm, and marked hydrops. Magnetic resonance imaging confirmed the diagnosis of CHAOS. Polyhydramnios and fetal skin edema were improved and the fetal ascitic fluid was regressed gradually. At 36 weeks of gestation, an EXIT procedure was undertaken. Fetal laryngoscopy and bronchoscopy showed complete laryngeal obstruction, and a tracheostomy was performed immediately. The infant was discharged from hospital at 6 weeks of age. Thereafter, he developed well both physically and mentally. A laryngoplasty was performed at 20 months of age using silicon sheet as a patent airway. The child has a tracheostomy, is able to phonate but does not speak, and is awaiting decannulation. Use of the EXIT procedure in CHAOS cases offers the potential for salvage and excellent long-term outcome of these fetuses that otherwise would not survive. However, management of the airway, particularly with regard to long-term reconstruction in children with CHAOS, remains challenging.
Authors:
Fumi Shimabukuro; Kaoru Sakumoto; Hitoshi Masamoto; Yoshihide Asato; Tomohide Yoshida; Akihiko Shinhama; Eiko Okubo; Aki Ishisoko; Yoichi Aoki
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Publication Detail:
Type:  Case Reports; Journal Article; Review     Date:  2007-03-19
Journal Detail:
Title:  American journal of perinatology     Volume:  24     ISSN:  0735-1631     ISO Abbreviation:  Am J Perinatol     Publication Date:  2007 Mar 
Date Detail:
Created Date:  2007-03-28     Completed Date:  2007-05-22     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8405212     Medline TA:  Am J Perinatol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  197-201     Citation Subset:  IM    
Affiliation:
Department of Obstetrics and Gynecology, Faculty of Medicine, University of the Ryukyus, Okinawa, Japan.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple
Adult
Airway Obstruction / congenital,  surgery*,  ultrasonography
Female
Fetal Diseases / surgery*
Humans
Infant, Newborn
Laryngoscopy
Larynx
Magnetic Resonance Imaging
Male
Polyhydramnios
Pregnancy
Salvage Therapy
Syndrome
Tracheostomy*
Ultrasonography, Prenatal

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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