Detection of H-type bronchoesophageal fistula in a newborn: A case report and literature review.

RATIONALE: Congenital tracheoesophageal fistula (TEF) is a rare developmental malformation. The H subtype accounts for approximately 4% of TEFs. Unlike other TEFs, the H-type is not accompanied by esophageal atresia and has nonspecific clinical symptoms, and its specific anatomical abnormalities are not always readily apparent. Furthermore, none of the currently available diagnostic methods for H-type TEF have absolute sensitivity, resulting in misdiagnoses, and accurate diagnoses are often delayed even until adulthood; in our case, we detected a congenital bronchoesophageal fistula, which is even more rare than regular H-type TEF, through a technique that was not previously reported for newborns, involving bronchoscopy, with methylene blue injected through an esophagoscope. We believe that we have provided this kind of case first in newborns.Furthermore, because there is not one literature summarizing the clinical symptoms and the effective methods up to now, we still are not clear which detective method is more efficient or accurate, especially in newborns, so it is very necessary to summarize and compare for improving the early diagnosis of TEFs; our study makes a significant contribution to the literature because we collated previously reported cases, including the clinical features and the usefulness and success rates of major tests, which will be very helpful for the early diagnosis of TEFs. PATIENT CONCERNS: A newborn male presented with an array of nonspecific clinical symptoms from birth, leading to pneumonia and mechanical ventilation. Oral feeding led to an improvement in most but not all symptoms, which returned when oral feeding was resumed. A second round of confirmatory tests was still unable to detect the cause. DIAGNOSIS: The diagnosis of H-type bronchoesophageal fistula was established through a technique that was not previously reported for newborns, involving bronchoscopy, with methylene blue injected through an esophagoscope. INTERVENTIONS: The surgery was performed after diagnosis, and the bronchoesophageal fistula was successfully repaired. OUTCOMES: The patient was discharged on postoperative day 7, and his status was reported to be normal at a follow-up visit 8 months after surgery. LESSONS: H-type TEF is a rare congenital abnormality, and its early diagnosis is highly difficult, especially bronchoesophageal fistula. Increased oral saliva and air-filled stomachs are characteristic manifestations. Bronchoscopy combined with esophagoscopy can improve the rate of early diagnosis. A combination of tests can improve the detection rate.

36-year-old man • persistent dry cough • frequent sinus congestion • hemoptysis • Dx?

► Persistent dry cough ► Frequent sinus congestion ► Hemoptysis.

Thoracoscopic Surgery for a Congenital Bronchoesophageal Fistula With Pulmonary Sequestration in an Adult Woman.

A congenital bronchoesophageal fistula with pulmonary sequestration is rare in adults. Here, we report the case of an adult woman having congenital bronchoesophageal fistula with intralobar pulmonary sequestration who successfully underwent thoracoscopic resection and showed a good postoperative course.

Intralobar pulmonary sequestration associated with congenital broncho-esophageal fistula.

Pulmonary sequestration and congenital broncho-esophageal fistula are both rare malformations; nonetheless, in the presence of recurrent pulmonary infection, the possibility that one or the other may be involved should not be disregarded. To our knowledge, the concurrence of intralobar pulmonary sequestration and congenital broncho-esophageal fistula is very rare. Herein, we report a case of intralobar pulmonary sequestration combined with congenital broncho-esophageal fistula, both of which were successfully treated with surgery.

Two unusual cases of adult onset congenital bronchoesophageal fistulas treated with fistula division.

Adult onset congenital bronchoesophageal fistula is a very rare entity. We report 2 cases of adult onset type II congenital bronchoesophageal fistula between the distal thoracic esophagus and the lower lobe superior segmental bronchi surgically treated through a right and left thoracotomy, respectively. In both cases the fistula was transected and sutured with no parenchyma resection. Both patients had an uneventful recovery. Resection of the underlying parenchyma during surgery for bronchoesophageal fistula is not always necessary as the lung can heal in time after performing just fistulectomy.

Anesthetic management of congenital broncho-esophageal fistula in an adult.

Broncho-esophageal fistula (BEF) are quite rare in adults, more so the congenital variety. The common causes of BEF in adults include infections, trauma, and malignancies. We report a rare case of congenital BEF manifesting in adulthood with repeated pulmonary infections. We emphasize mainly on the preoperative preparation and perioperative management of this patient. It is essential to have a high index of suspicion to diagnose congenital BEF in adults. Anesthesiologists play an important role in successful management of these cases.

[Congenital broncho-esophageal fistula in an elderly patient with lung abscess].

A 60-year-old man who had been treated for pneumonia by another hospital was suspected to have a broncho-esophageal fistula, and was referred to our hospital for surgical treatment. Chest computed tomography( CT) demonstrated a lung abscess. Upper gastrointestinal endoscopic examination demonstrated an esophageal diverticulum. He underwent the resection of the fistula and left lower lobectomy of lung under left thoracotomy. Operative findings showed no severe adhesion around fistula, suggesting its congenital origin, which was also pathologically supported later. He suffered from postoperative empyema by anaerobe infection, which was successfully controlled by open drainage.

Thoracoscopic management of a communicating bronchopulmonary foregut malformation in a 23-month-old child.

A communicating bronchopulmonary foregut malformation is a rare anomaly characterized by a fistula between an isolated portion of respiratory tissue and the esophagus or stomach. We describe the thoracoscopic management of a communicating bronchopulmonary foregut malformation in a 23-month-old boy by division of an anomalous bronchus and right upper lobectomy.

Aspiration via congenital broncho-esophageal fistula after lobectomy.

Aspiration via a congenital broncho-esophageal fistula in an adult thoracotomy patient has not been previously reported. Repeated aspiration and subsequent respiratory failure if the fistula is not recognized could be life-threatening in these postoperative patients. We describe one such critical case, in which a broncho-esophageal fistula was discovered weeks after aspiration and the onset of respiratory failure after left lower lobectomy. This unusual case suggests that repeated localized pulmonary infections can indicate a broncho-esophageal fistula and that further investigations should be performed, including detailed history.

Congenital bronchoesophageal fistula in adults.

AIM: To study the clinical characteristics, diagnosis and surgical treatment of congenital bronchoesophageal fistulae in adults. METHODS: Eleven adult cases of congenital bronchoesophageal fistula diagnosed and treated in our hospital between May 1990 and August 2010 were reviewed. Its clinical presentations, diagnostic methods, anatomic type, treatment, and follow-up were recorded. RESULTS: Of the chief clinical presentations, nonspecific cough and sputum were found in 10 (90.9%), recurrent bouts of cough after drinking liquid food in 6 (54.6%), hemoptysis in 6 (54.6%), low fever in 4 (36.4%), and chest pain in 3 (27.3%) of the 11 cases, respectively. The duration of symptoms before diagnosis ranged 5-36.5 years. The diagnosis of congenital bronchoesophageal fistulae was established in 9 patients by barium esophagography, in 1 patient by esophagoscopy and in 1 patient by bronchoscopy, respectively. The congenital bronchoesophageal fistulae communicated with a segmental bronchus, a main bronchus, and an intermediate bronchus in 8, 2 and 1 patients, respectively. The treatment of congenital bronchoesophageal fistulae involved excision of the fistula in 10 patients or division and suturing in 1 patient. The associated lung lesion was removed in all patients. No long-term sequelae were found during the postoperative follow-up except in 1 patient with bronchial fistula who accepted reoperation before recovery. CONCLUSION: Congenital bronchoesophageal fistula is rare in adults. Its most useful diagnostic method is esophagography. It must be treated surgically as soon as the diagnosis is established.

Fístula broncoesofágica congénita de diagnóstico tardío / Congenital bronchoesophageal fistula of late diagnosis

Presentamos el caso de una mujer de 52 años con el antecedente de infecciones respiratorias de repetición desde la infancia, que presenta una fístula broncoesofágica (FBE) congénita diagnosticada de forma incidental a raíz de la aparición de disfagia en los últimos meses. Los estudios radiológicos y manométricos demuestran la asociación con megaesófago y acalasia. Se confirmó el diagnóstico de FBE en bronquio principal izqdo. gracias a un tránsito con gastrografín, esofagoscopia y fibrobroncoscopia. El tratamiento fue quirúrgico mediante toracotomía posterolateral izquierda, procediendo a la resección del trayecto fistuloso e invaginación del muñón con sutura de muscularis esofágica (AU)

We report the case of a 52-year-old woman with a history of recurrent respiratory infections, which presents a congenitalbronchoesophageal fistula (BEF) incidentally diagnosed after the appearance of dysphagia in the last months. Theradiological and manometric studies demonstrate the association with megaesophagus and achalasia. Diagnosis of BEFm in the left main bronchus was confirmed thanks to an esophagogram, esophagoscopy and fiberoptic bronchoscopy. The surgical treatment was carried out through a left posterolateral thoracotomy, proceeding to resection of the fistula and invagination of the stump with suture of esophageal muscularis (AU)

[Congenital bronchoesophageal fistula in an adult; an undiscribed mechanism of symptom tolerance].

We present a case of a congenital bronchoesophageal fistula in a 61-year-old woman. She was referred to hospital because of postprandial heart burn. Three-dimensional (3D) computed tomography (CT) demonstrated an anastomosis between her right intermediate bronchus and esophagus. In spite of direct communication between her bronchus and esophagus, she has never suffered severe infection. We visualized the orifice of fistula closed with mucosal flap in swallowing by means of a bronchofiberscope. The delay of a diagnosis was explained by symptom tolerance. Some theories as to the symptom tolerance are found in literatures, but we supposed to find an undiscribed mechanism; closure of the orifice in swallowing. The fistula was surgically closed.