Literature DB >> 14694403

A case of laryngeal atresia (congenital high airway obstruction syndrome) with chromosome 5p deletion syndrome rescued by ex utero intrapartum treatment.

Yutaka Kanamori1, Yoshihiro Kitano, Kohei Hashizume, Masahiko Sugiyama, Tetsuya Tomonaga, Hajime Takayasu, Satoshi Egami, Keiji Goishi, Kazuhiko Shibuya, Yukiko Kawana, Genzo Marumo, Akihiko Kikuchi, Shiro Kozuma, Yuji Taketani, Yushi Sekiyama.   

Abstract

The authors report a case of laryngeal atresia (congenital high airway obstruction syndrome [CHAOS]) that was diagnosed prenatally. The patient underwent successfully tracheostomy by ex utero intrapartum treatment (EXIT). The fetal ultrasonography and magnetic resonance imaging MRI showed a typical CHAOS pattern with expanded hyperechogenic lungs, inverted diaphragms, and a dilated trachea. Recently, 3 cases of prenatally diagnosed CHAOS were reported to be treated successfully by EXIT. The clinical manifestation and course of this case was not similar to these 3 cases. The 3 previous patients did not fare as well during gestation and were delivered earlier than that in our case. In our case, fetal hydrops was seen at 23 gestational weeks, but it gradually subsided and disappeared at 30 gestational weeks. The fetus was stable and well. After delivery at 39 weeks, the baby received respiratory assistance by ventilator assistance. After 3 days, she could breath well on her own. The patient also had chromosome 5p deletion syndrome and perineal groove. More experience in treating CHAOS cases with EXIT to fully estimate its clinical course and prognosis is needed.

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Year:  2004        PMID: 14694403     DOI: 10.1016/j.jpedsurg.2003.09.041

Source DB:  PubMed          Journal:  J Pediatr Surg        ISSN: 0022-3468            Impact factor:   2.545


  6 in total

1.  Evidence for autosomal dominant inheritance in prenatally diagnosed CHAOS.

Authors:  Piet Vanhaesebrouck; Kris De Coen; Paul Defoort; Hubert Vermeersch; Geert Mortier; Linde Goossens; Koen Smets; Alexandra Zecic; Sabine Vandaele; Frans De Baets
Journal:  Eur J Pediatr       Date:  2006-04-27       Impact factor: 3.183

2.  Isolated congenital tracheal stenosis in a preterm newborn.

Authors:  Ulrich Krause; Ralph M W Rödel; Thomas Paul
Journal:  Eur J Pediatr       Date:  2011-05-18       Impact factor: 3.183

3.  Congenital high airways obstruction syndrome - first and second trimester diagnosis.

Authors:  Debra Paoletti; Meiri Robertson
Journal:  Australas J Ultrasound Med       Date:  2015-12-31

4.  [Congenital high airway obstruction syndrome (CHAOS): a case report].

Authors:  Kamal El Moussaoui; Aziz Slaoui; Aziz Baidada; Aicha Kharabch
Journal:  Pan Afr Med J       Date:  2021-01-01

5.  Case report: Antenatal diagnosis of congenital high airway obstruction syndrome - laryngeal atresia.

Authors:  Mukesh Kumar Garg
Journal:  Indian J Radiol Imaging       Date:  2008-11

6.  Prenatal diagnosis and pathology of laryngeal atresia in congenital high airway obstruction syndrome.

Authors:  Piya Chaemsaithong; Tharintorn Chansoon; Boonsri Chanrachakul; Suchin Worawichawong; Sansanee Wongwaisayawan; Patama Promsonthi
Journal:  Case Rep Radiol       Date:  2012-12-24
  6 in total

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