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Войти в исследования Pediatrics and neonatology · 2025
Diagnostic and surgical challenges in atypical forms of esophageal atresia: A case series.
СтатьяКлинический случайПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Shchapov Nikolay F, Ekimovskaya Ekaterina V, Kulikov Denis V, Shatova Svetlana M, Sergeeva Svetlana V
Аннотация
BACKGROUND: Esophageal atresia (EA) is a rare malformation (∼1 in 2500-3000 live birth) typically involving a blind upper pouch and distal tracheoesophageal fistula (TEF) (≈86 % of cases). Atypical forms (such as duplications, vascular rings, or functional obstructions) lie outside standard classifications and they are very uncommon. These variants can confound diagnosis and require adapted strategies.
METHODS: We retrospectively reviewed 43 EA cases (2018-2021) and identified three with atypical anatomic or functional presentations. Clinical, radiologic, and endoscopic findings were analyzed. Each case demanded a bespoke diagnostic and surgical plan.
RESULTS: Case 1: A neonate with EA/TEF had a long esophageal duplication with an internal septum. The septum was divided and the esophagus anastomosed; although the repair was intact, the infant later died from aspiration pneumonia. Case 2: EA repair was complicated by a right aortic arch (≈6 % of EA/TEF patients). We tunneled the distal esophagus behind the arch for a thoracoscopic anastomosis. A mild stricture was dilated, and the infant tolerated full feeds. Case 3: Initial imaging suggested EA, but endoscopy showed a continuous but narrowed esophagus (likely a motility disorder). Tube feeding led to normalization by 1 month, avoiding surgery. Two infants survived with good feeding outcomes.
CONCLUSION: These cases (≈7 % of our EA cohort) illustrate that atypical EA variants require individualized management. Detailed imaging and endoscopy were critical to identify unusual anatomy. Tailored thoracoscopic techniques (such as septum resection and retro-arch anastomosis) restored continuity in two infants, while recognizing a motility obstruction in the third obviated surgery. Awareness of atypical anatomy is essential to improve outcomes.
THE LEVEL OF EVIDENCE: IV.
METHODS: We retrospectively reviewed 43 EA cases (2018-2021) and identified three with atypical anatomic or functional presentations. Clinical, radiologic, and endoscopic findings were analyzed. Each case demanded a bespoke diagnostic and surgical plan.
RESULTS: Case 1: A neonate with EA/TEF had a long esophageal duplication with an internal septum. The septum was divided and the esophagus anastomosed; although the repair was intact, the infant later died from aspiration pneumonia. Case 2: EA repair was complicated by a right aortic arch (≈6 % of EA/TEF patients). We tunneled the distal esophagus behind the arch for a thoracoscopic anastomosis. A mild stricture was dilated, and the infant tolerated full feeds. Case 3: Initial imaging suggested EA, but endoscopy showed a continuous but narrowed esophagus (likely a motility disorder). Tube feeding led to normalization by 1 month, avoiding surgery. Two infants survived with good feeding outcomes.
CONCLUSION: These cases (≈7 % of our EA cohort) illustrate that atypical EA variants require individualized management. Detailed imaging and endoscopy were critical to identify unusual anatomy. Tailored thoracoscopic techniques (such as septum resection and retro-arch anastomosis) restored continuity in two infants, while recognizing a motility obstruction in the third obviated surgery. Awareness of atypical anatomy is essential to improve outcomes.
THE LEVEL OF EVIDENCE: IV.