Congenital laryngeal atresia associated with esophageal atresia and tracheoesophageal fistula: a case of long-term survival

Okuyama, Hiroomi; Kubota, Akio; Kawahara, Hiroyuki; Oue, Takaharu; Tazuke, Yuko

doi:10.1016/j.jpedsurg.2006.08.040

Cited by 18 publications

(18 citation statements)

References 12 publications

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“…Indeed, in the absence of a functional tracheo-esophageal fistula, the PTD might protect the fetal lung from excessive pressure resulting in a less severe effect on fetal lung organogenesis. Without a PTD, lung organogenesis will be severely affected resulting in bilateral solid CCAM, which is lethal (Okuyama et al, 2006;Cavoretto et al, 2008).…”

Section: Discussionmentioning

confidence: 99%

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Prenatal diagnosis and pulmonary pathology in congenital high airway obstruction sequence

2009

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Section: Discussionmentioning

confidence: 99%

“…In the presence of a large functional tracheoesophageal fistula, prenatal diagnosis is impossible. In these cases, airway management at birth is difficult due to the unexpected ventilatory problems and esophageal intubation, tracheal puncture or tracheostomy is required with only few reports of long-term survivors (Okuyama et al, 2006).…”

Section: Discussionmentioning

confidence: 99%

Prenatal diagnosis and pulmonary pathology in congenital high airway obstruction sequence

2009

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“…Post-mortem examination allowed for the diagnosis of a laryngomucocoele as the cause of this upper airway obstruction and the fatal outcome is a frequent event in undiagnosed CHAOS. The absence of the other echographical parameters of the triad may be explained if it is assumed that the obstruction at 30 weeks was only partial 2 , 7. Oesophageal compression by dilated airways and increased lung volume are probably the cause of polyhydramnios due to decreased ingestion of amniotic fluid 1 , 6…”

Section: Discussionmentioning

confidence: 99%

Congenital laryngomucocoele: a rare cause for CHAOS

Cunha

Janeiro²,

Fernandes³

et al. 2009

Case Reports

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Congenital high airway obstruction syndrome (CHAOS) is a rare but life-threatening condition that results from the obstruction of the upper airways.We describe a female newborn, from a Grávida II, Para 0, 36-year-old woman, with a routine ultrasound at 30 weeks' gestation that showed polyhydramnios. She delivered a live-born female baby at 36 weeks without any dismorphic features but with respiratory distress. Attempts at endotracheal intubation were unsuccessful due to the presence of a mass obstructing the larynx. The reanimation process was stopped after 20 minutes. Post-mortem examination demonstrated the presence of a total occlusion of the larynx by a laryngomucocoele. Laryngocele, a congenital cyst of the larynx, occurs rarely and hardly ever as a cause of CHAOS. What is more, laryngomucocoele has not been previously reported as a cause of CHAOS. These conditions represent a neonatal emergency with reserved prognosis unless diagnosed antenatally allowing for a programmed ex utero intrapartum treatment (EXIT) by performing tracheostomy while maintaining the placental circulation.

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“…Congenital high airway obstruction syndrome (CHAOS) can usually be diagnosed by prenatal ultrasound, which shows certain characteristic findings including overdistended hyperechogenic lungs, an inverted diaphragm, a dilated trachea, polyhydramnios, and ascites [4]. However, when a TEF is present, there may be no such findings because the pulmonary secretion can exit through the airway, thereby permitting normal lung development [7]. TEF and CHAOS conceal each other's characteristic findings, which may result in difficulties in diagnosing CHAOS associated with TEF during the prenatal period.…”

Section: Prenatal Diagnosis and Management Of Congenital Subglottic Smentioning

confidence: 99%

“…However, if the patient has CHAOS without a proximal TEF such as Gross type C EA, endoesophageal intubation would be impossible. Only 2 survivors of CHAOS associated with EA and distal TEF have been reported in the literature [6,7]. Both patients had laryngeal atresia and EA without prenatal diagnosis.…”

Section: Prenatal Diagnosis and Management Of Congenital Subglottic Smentioning

confidence: 99%

Prenatal Diagnosis and Management of Congenital Subglottic Stenosis Associated with Congenital Esophageal Atresia Type C

et al. 2008

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Most patients with congenital esophageal atresia (EA) have congenital tracheobronchial abnormalities, which may cause respiratory distress, be difficult to treat and have a poor prognosis. The authors report a neonate with EA and congenital subglottic stenosis (SGS) who exhibited severe respiratory distress immediately after birth. After emergency endotracheal intubation with a narrow endotracheal tube, the authors performed total correction of EA and anterior cricoid split (ACS) on day 1 of age. The postoperative course was uneventful. Some reports have stated that it is difficult to make a prenatal diagnosis when SGS is associated with EA and tracheoesophageal fistula (TEF). The anterior cricoid split technique may be suitable for managing moderate SGS even in neonates with EA. Partial resection of the hypertrophic cricoid cartilage is considered effective in preventing restenosis.

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Congenital laryngeal atresia associated with esophageal atresia and tracheoesophageal fistula: a case of long-term survival

Cited by 18 publications

References 12 publications

Prenatal diagnosis and pulmonary pathology in congenital high airway obstruction sequence

Prenatal diagnosis and pulmonary pathology in congenital high airway obstruction sequence

Congenital laryngomucocoele: a rare cause for CHAOS

Prenatal Diagnosis and Management of Congenital Subglottic Stenosis Associated with Congenital Esophageal Atresia Type C

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