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.

Anesthesia management in neonatal congenital bronchobiliary fistula: case report and literature review.

BACKGROUND: There is very little published literature and none that discussed care in a neonate regarding anesthetic risk and management of neonate with congenital bronchobiliary fistula during thoracoscopy and thoracotomy. This article analyzes related risk factors and literature review from perioperative ventilation, circulation and other aspects of management. CASE PRESENTATION: A neonate diagnosed as congenital bronchobiliary fistula combined with severe chemical pneumonia, consolidation of the lungs, and infection was facing the risk of anaesthesia under thoracoscopy exploration surgery, who experiened more than 20 days diagnostic period before operation. Many risk factors have led to conversion from minimally invasive surgery to thoracotomy, including persistent hypoxemia, hypercapnia, difficult surgical exposure and extremly difficulty of intraoperative ventilation management. Anesthesia maintenance after conversion to open access remained problematic. Fortunately the patient showed no sign of any adverse CNS effects after 4 months of follow-up. CONCLUSIONS: The most prominent anesthesia challenges are hypoxemia, increased airway resistance, impaired ventilation, and the risk of metabolic acidosis. Close cooperation among the entire neonatal medical team is the key factors in successful management of this rare case.

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

► Persistent dry cough ► Frequent sinus congestion ► Hemoptysis.

Evaluation of endoscopic coblation treatment for obliteration of congenital pyriform sinus fistula.

BACKGROUND: Congenital pyriform sinus fistula is a very rare branchial apparatus malformation. Conventional open surgery for fistulectomy might fail to excise the lesion completely, leading to continual recurrence. The aim of this study was to provide an innovative endoscopic coblation technique for patients with pyriform sinus fistula and evaluate its intermediate-term effectiveness. METHODS: Retrospective case series with 112 patients (age range 3-36 years) between 2013 and 2016 and underwent endoscopic coblation of the sinus fistula. Data collected including patient demographics, presenting symptoms, diagnostic methods, prior and subsequent treatments, length of hospital stay, and recurrence were analyzed. RESULTS: Of the 112 cases, there were no postoperative complications observed except temporary reddish swelling in three patients. Four cases were lost to follow-up. Of 108 patients, 106 experienced no recurrence after their first endoscopic coblation of the sinus tract. The remaining two patients with recurrence in the follow-up were ultimately treated with recoblation without complications or further recurrences. Also, Endoscopic coblation can be used to treat seven patients with recurrence who had experienced open resection. In our series, median follow-up period was 1.5 years. CONCLUSION: Endoscopic coblation is an effective approach for most patients. We advocate using this minimally invasive technique as first line of treatment for pyriform sinus fistula.

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.

Fístula broncobiliar congénita: Caso dínico / Congenital broncho-biliary fistula: A case report

La fístula traqueo- o broncobiliar congénita o fístula hepatopulmonar congénita es una rara malformación con alta morbimortalidad si su diagnóstico no se realiza a tiempo. Existe una comunicación entre el sistema respiratorio (tráquea o bronquio) y el tracto biliar. Hasta la fecha, se han reportado solo 35 casos. Presentamos el caso de un neonato con neumopatía derecha y débito bilioso por el tubo endotraqueal. El diagnóstico se realizó mediante broncoscopía con radioscopía y la resección de la fístula broncobiliar fue por videotorascopía. Posteriormente, se requirió de una hepatectomía izquierda con anastomosis biliodigestiva en Y de Roux por presentar hipoplasia de colédoco.

Congenital tracheo-or-bronchobiliary fistula or congenital he-patopulmonary fistula is a rare malformation with high morbidity and mortality if the diagnosis is not made early. The tracheo-or-bronchobiliary fistula is a communication between the respiratory (trachea or bronchus) and biliary tract. To date, only 35 cases have been published worldwide. We report a case of a neonate with right pneumonia and bilious fluid in the endotracheal tube. Diagnosis was made using bronchoscopy with fluoroscopy. Videothoracoscopy was used to remove the bronchobiliary fistula. Subsequently, a left he-patectomy with Roux-en-Y biliary-digestive anastomosis was performed as bile ductus hypoplasia was present.

[Congenital broncho-biliary fistula: a case report]. / Fístula broncobiliar congénita: caso dínico.

Congenital tracheo-or-bronchobiliary fistula or congenital he-patopulmonary fistula is a rare malformation with high morbidity and mortality if the diagnosis is not made early. The tracheo-or-bronchobiliary fistula is a communication between the respiratory (trachea or bronchus) and biliary tract. To date, only 35 cases have been published worldwide. We report a case of a neonate with right pneumonia and bilious fluid in the endotracheal tube. Diagnosis was made using bronchoscopy with fluoroscopy. Videothoracoscopy was used to remove the bronchobiliary fistula. Subsequently, a left he-patectomy with Roux-en-Y biliary-digestive anastomosis was performed as bile ductus hypoplasia was present.

La fístula traqueo- o broncobiliar congénita o fístula hepatopulmonar congénita es una rara malformación con alta morbimortalidad si su diagnóstico no se realiza a tiempo. Existe una comunicación entre el sistema respiratorio (tráquea o bronquio) y el tracto biliar. Hasta la fecha, se han reportado solo 35 casos. Presentamos el caso de un neonato con neumopatía derecha y débito bilioso por el tubo endotraqueal. El diagnóstico se realizó mediante broncoscopía con radioscopía y la resección de la fístula broncobiliar fue por videotorascopía. Posteriormente, se requirió de una hepatectomía izquierda con anastomosis biliodigestiva en Y de Roux por presentar hipoplasia de colédoco.

Minimally Invasive Surgical Repair for Congenital Bronchobiliary Fistula in an Adult.

Congenital bronchobiliary fistula (CBBF) is a very rare disease and usually requires surgical intervention at a young age. We report a case of CBBF in an adult who was treated successfully with a minimally invasive endoscopic operation.

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.

[Lung perfusion scintigraphy in pediatric patients with congenital malformations]. / La scintigrafia polmonare in età neonatale e pediatrica per patologie congenite.

Congenital malformations are an heterogeneous group of disorders that can lead changes in pulmonary perfusion and then can be evaluated with lung perfusion scintigraphy. We selected five patients in which the role of lung perfusion scintigraphy emerges as a reliable and non-invasive imaging technique. Lung perfusion scintigraphy is a useful tool in pediatric patients with congenital malformations allowing an accurate evaluation of the best therapeutic strategy and its results.

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.

[Diagnosis and surgical treatment of congenital bronchoesophageal fistula in adult].

OBJECTIVE: To explore the clinical characteristics, diagnosis and surgical treatment of adult congenital bronchoesophageal fistula. METHODS: Eleven cases of adult congenital bronchoesophageal fistula that were diagnosed and surgically treated between May 1990 and August 2010 had been reviewed. There were 7 male and 4 female patients, ranging in age from 28 to 66 years (mean 48.7 years). The chief clinical presentation included coughing and sputum in 10 cases, recurrent bouts of coughing after drinking liquid food in 6 cases, hemoptysis in 6 cases, low fever in 4 cases, chest pain in 3 cases. The duration of symptoms before diagnosis ranged from 5 to 36 years (mean 16.8 years). The diagnosis of bronchoesophageal fistula was confirmed most by esophagography. Associated diseased lung was resected in all patients (lobectomy in 10 cases and pneumonectomy in 1 case). The operation included right thoracotomy in 7 cases and left thoracotomy in 4 cases. The fistula was completely resected in 10 cases. The tract was simply divided and the end was sutured in 1 case. RESULTS: The postoperative course was uneventful in 10 patients who were discharged from hospital 10 to 18 d after operation. One patient suffered from esophageal fistula and received second operation. Regular follow-up was conducted on all 11 patients, proving that 3-year survival rate was 11/11 and 5-year survival rate was 9/11. CONCLUSION: Persistence of congenital bronchoesophageal fistula into adulthood is rare. The main symptom is nonspecific coughing and bouts of coughing after drinking liquid food. The most useful diagnostic method is the esophagography. Even though it is benign disease, life-threatening complications might occur and it must be treated surgically as soon as the diagnosis is established.

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.