TACRD and VACTERL associations in a fetus: Case report and review of the literature

Xu, Ganqiong; Zhou, Qing; Zhang, Ming; Pu, Da-rong; Ouyang, Zhu

doi:10.1016/j.ijporl.2013.09.016

Cited by 4 publications

(3 citation statements)

References 62 publications

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“…VACTERL association is a rare and complex condition with highly heterogeneous etiology and manifestations. [2,6] Approximately 90% of VACTERL association cases occur sporadically, [7] with an empiric recurrence risk of 1% or less. [4] Most doctors require at least 3 component features for diagnosis, without clinical or laboratory-based evidence of the overlapping conditions, while others emphasize the presence of certain component features, especially TEF or anorectal malformation.…”

Section: Discussionmentioning

confidence: 99%

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VACTERL association complicated with multiple airway abnormalities

Lin

Zhong

et al. 2019

Medicine

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Introduction:VACTERL association is an acronym that includes vertebral anomalies (V), anal atresia (A), cardiac defects (C), tracheoesophageal fistula (TEF) or esophageal atresia (EA), renal anomalies (R), and limb defects (L). Airway anomalies have rarely been reported with VACTERL association.Patient concerns:A 10-month-old boy who had been diagnosed with anal atresia and received surgical corrections soon after birth consulted our institution by complaining repeated cough and fever.Diagnosis:Diagnosis of VACTERL association was finally made. Bronchoscopy and chest CT with computed tomography angiography confirmed multiple airway abnormalities including bridging bronchus, airway malacia, and complete tracheal rings.Interventions:Supplemental oxygen was provided and antibiotics was initiated.Outcomes:The patient resolved gradually and was discharged 10 days later. The follow-up showed the patient has remained well just with mild psychomotor retardation.Conclusion:Multiple airway anomalies may be seen in VACTERL association. It is worthwhile to make special note for evaluating the tracheobronchial pulmonary system by chest CT and bronchoscopy, especially patients presenting with breathing anomalies.

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Section: Discussionmentioning

confidence: 99%

“…[1] No definite etiology and pathogenesis has been proven, but a defect in mesodermal differentiation due to a variety of causes(genetic, environmental and multi-factors are implicated), in early first trimester (between 6th and 10th weeks of gestation), has been suggested. [7,8]…”

Section: Discussionmentioning

confidence: 99%

VACTERL association complicated with multiple airway abnormalities

Lin

Zhong

et al. 2019

Medicine

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“…The incidence was <1 in every 50,000 infants in a male-to-female ratio of 2:1. [2] Respiratory distress at birth and failure of intubation usually occur in TA. Affected patients could breathe via a tracheoesophageal fistula (TEF) if it is present.…”

Section: Introductionmentioning

confidence: 99%

Tracheal atresia with a cephalically developed lung bud

Lee

Yi²,

Yun³

et al. 2019

Medicine

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Rationale: Tracheal atresia (TA) involves complete or partial tracheal absence below the larynx. It involves failure of complete separation of the laryngotracheal diverticulum from the foregut at the 4th gestational week. In TA, the trachea and main bronchi generally proceed in the normal caudal directions. Patient concerns: At the gestational age of 34 weeks and 6 days, a male baby weighing 2290 g was born via cesarean section. A brisk bag was used, and mask ventilation was performed, but was not effective. Intubation was attempted; however, the endotracheal tube did not advance below the vocal cord. Diagnosis: Tracheal atresia. Interventions: An emergent open neck exploration could not reveal the trachea. On computed tomography, the tracheoesophageal fistula (TEF) started from the lower esophagus just above the gastroesophageal junction. The trachea arose upward to form the main bronchus. Outcomes: His parents signed the “Do not resuscitate” form due to poor outcome. On the 23rd day of birth, the baby expired. Lessons: Our case involved TA with tracheoesophageal fistula forming between the trachea and main bronchi in a cephalic direction as detected on computed tomography. Further, the trachea arose from the gastroesophageal junction; thus, it does not belong to any Floyd classification. Herein, we report a TA case with a cephalically developed lung bud.

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Tracheal Agenesis: Vertical Division of the Native Esophagus – A Novel Surgical Approach and Review of the Literature

Straughan

Mulcahy

Sandler

et al. 2020

Ann Otol Rhinol Laryngol

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Introduction: Tracheal agenesis (TA) is rare and usually fatal. Few survivors with concomitant tracheoesophageal fistulae (TEF) who underwent ligation of the distal esophagus with creation of a spit-fistula and neo-trachea from the proximal esophagus exist. We report a novel surgical technique whereby the esophagus is divided longitudinally to preserve a functional alimentation tract and a parallel neo-trachea. We review the literature of reported cases, including survivors beyond 12 months. Methods: Case report and literature review. Results: A female infant with prenatal polyhydramnios was born at 35 weeks gestation with immediate respiratory distress and absent cry. Oxygenation was maintained with a laryngeal mask airway. Despite a normal appearing larynx, she could not be intubated and emergent neck exploration disclosed no cervical trachea. The patient was placed on extra corporeal membranous oxygenation (ECMO), and later diagnosed with TA, Floyd Type I. Parental desire for reconstruction but refusal of a spit-fistula necessitated a novel procedure. The esophagus was divided longitudinally via a microstapler to preserve the original alimentary tract and create a parallel neo-trachea originating from the TEF and terminating as a cervical stoma. The healing process was complicated but the baby was ultimately discharged to home where she developed normally neurologically until succumbing one night to accidental decannulation at 16 months of age. Conclusion: We describe a novel surgical approach to manage TA. This includes avoiding creation of a spit fistula and preserving the native esophagus. We then survey the literature, reporting the survivorship duration and operative management of 174 reported cases of TA.

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TACRD and VACTERL associations in a fetus: Case report and review of the literature

Cited by 4 publications

References 62 publications

VACTERL association complicated with multiple airway abnormalities

VACTERL association complicated with multiple airway abnormalities

Tracheal atresia with a cephalically developed lung bud

Tracheal Agenesis: Vertical Division of the Native Esophagus – A Novel Surgical Approach and Review of the Literature

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