Management of a double H-type tracheoesophageal fistula without esophageal atresia: a case report

IF 0.4 Q4 PEDIATRICS Annals of Pediatric Surgery Pub Date : 2023-12-01 DOI:10.1186/s43159-023-00274-8
Chiraz Regaieg, Taycir Cheikhrouhou, Mahdi Ben Dhaou, Mariam Ammar, Nadia Kolsi, Riadh Mhiri, Afef Ben Thabet, Nadia Hmida
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Abstract

Double congenital tracheoesophageal fistula (TEF) without esophageal atresia, known as double H-type fistula, is an extremely uncommon malformation. Due to its nonspecific symptoms and the small size of the fistula in infancy, H-type tracheoesophageal fistula is rarely diagnosed in the neonatal period and is also challenging to repair. We report a rare case of double H-type tracheoesophageal fistula in a 2-day-old newborn, identified with a time-lapse and repaired separately. The diagnosis was established through repeated bronchoscopy, with methylene blue injected through an esophagoscope. A right thoracotomy was performed to undertake the surgical repair after cannulating the fistula with a vascular guide wire. Five months later, the infant, who had a history of recurrent episodes of coughing and shortness of breath, was diagnosed with acute respiratory distress, and a second TEF was detected by bronchoscopy. A cervical incision was used to repair the second H-type fistula. Double H-type tracheoesophageal fistula should be considered while correcting a tracheoesophageal fistula, especially if the symptoms continue after the first operation. Repeated radiological contrast studies and bronchoscopy with methylene blue injected through an esophagoscope help the early diagnosis.
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双h型气管食管瘘无食管闭锁1例
双先天性气管食管瘘(TEF)无食道闭锁,称为双h型瘘,是一种极为罕见的畸形。由于h型气管食管瘘的症状非特异性,且其在婴儿期瘘管体积小,因此在新生儿期很少被诊断出来,其修复也具有挑战性。我们报告一个罕见的病例双h型气管食管瘘在一个2天大的新生儿,确定与延时和单独修复。通过反复支气管镜检查确定诊断,并通过食管镜注射亚甲基蓝。在导管插管后行右开胸手术修复。5个月后,这名有反复咳嗽和呼吸短促病史的婴儿被诊断为急性呼吸窘迫,并通过支气管镜检查发现第二次TEF。采用宫颈切口修复第二个h型瘘管。在纠正气管食管瘘时应考虑双h型气管食管瘘,特别是首次手术后症状持续的情况下。反复放射造影检查和支气管镜通过食管镜注射亚甲基蓝有助于早期诊断。
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来源期刊
CiteScore
0.60
自引率
0.00%
发文量
74
审稿时长
9 weeks
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