A rare case of congenital bronchoesophageal fistula in an adult

IntroductionWhen congenital bronchoesophageal fistulas exist without atresia of the esophagus, the diagnosis can be delayed, although symptoms may occur early following fistula development. Therefore, while they are usually found in infants, they can be extremely rarely found in adults. We herein report a rare case of bronchoesophageal fistula without atresia in an adult.CaseAn 69-year-old male presented to the outpatient clinic with a decades-long history of cough with expectoration immediately after taking food, especially liquids.Computed tomograph, esophagoscopy, and esophagography revealed the fistulous communication between the mid-esophagus and right lower lobe bronchus, with consolidation in the right lower lobe. We performed right lower lobectomy with the closure and excision of the fistula. The histopathology of the fistula revealed the mucosa to be lined by stratified squamous epithelium. There was no evidence of inflammation, granuloma, or carcinoma.ConclusionIn conclusion, despite the benign nature of this malformation, if left untreated, it can cause long-term debilitating respiratory symptoms associated with the fistula. Therefore, the diagnosis should be considered in the evaluation of recurrent lung infection.     

Congenital broncho-esophageal fistula in the adult

The case of a 62-year-old woman with a type II congenital broncho-esophageal fistula is presented. She had had recurrent pulmonary infections that were more prominent in the last 15 years. A barium swallow examination showed a communication between the esophagus and the right lower lobe. High resolution computed tomographic scan of the chest revealed right middle and lower lobe bronchiectasis. Bronchoscopy was unremarkable. At thoracotomy bronchoesophageal fistula was divided and the esophageal end was repaired in two layered fashion and reinforced by pediculed parietal pleural flap. Right middle and lower lobectomies were performed. Demonstration of the broncho-esophageal fistula and assessment of the status of the pulmonary parenchyma are important steps prior to surgery.     

Bronchoesophageal fistula with an esophageal web

Congenital bronchoesophageal fistulas in the adult age group are rare, with only approximately 20 cases having been recognized. All of these cases have been an isolated esophageal anomaly without other associated esophageal pathology. We present an interesting case of an adult with both symptomatic congenital bronchoesophageal fistula and proximal esophageal web.     

Congenital bronchoesophageal fistula in an elderly lady

We present a case report of a congenital bronchoesophageal fistula presenting late in life. Bronchoesophageal fistulae commonly present in neonates, but sometimes, as in the present case they may be clinically silent till old age. She had a relatively short symptomatic period of around eighteen months. She was diagnosed on the basis of contrast computed tomography (CT), bronchoscopy and esophagoscopy, and underwent resection.     

A case of 72-year-old female with congenital esophago-bronchial fistula

A 72-year-old female with congenital esophago-bronchial fistula is reported. She had complained of cough attack during meals and repeated pneumonia since childhood. A chest x-ray film showed chronic bronchitis in the right lower lung field. Esophagogram revealed a fistula between the esophagus and the right lower lobe bronchus. Resection of the right lower lobe and removal of the fistulous tract were successfully performed. Histological examination of the resected fistula with slight inflammatory change showed muscle layer and transitional zone between the stratified squamous epithelium of esophagus and the ciliated columnar epithelium of bronchus. This is the oldest case in the Japanese literature.     

Congenital bronchoesophageal fistula in the adult

Congenital bronchoesophageal fistulas, when not associated with esophageal atresia, are compatible with life and may persist until adulthood before diagnosis has been established. We report such a rare case of a 55-year-old Caucasian female with a history of repeated pulmonary infections, suffering from cough during the last 12 months due to a mass in the right lung. A bronchoesophageal fistula (type III according to Braimbridge and Keith classification) was incidentally discovered during thoracotomy which was resected and end-sutured. Following that, a right lower lobectomy was performed. The patient had an uneventful recovery. The final diagnosis of congenital bronchoesophageal fistula was established excluding all the reasons that lead to the acquired disease. The diagnostic and therapeutic procedures are analyzed and the relevant literature is reviewed.     

Surgical treatment of benign tracheoesophageal fistula and bronchoesophageal fistula in the adults

This article deals with experiences of surgical treatment of benign tracheoesophageal fistula and bronchoesophageal fistula in seven adults. Among them 2 cases were congenital, 2 cases were infective and 3 cases were traumatic. There were 5 males and 2 females. Their average age ranged from 24 to 53 years. The modes of operation were as follows: (1) resection of fistula, and repairing of leak in 3 cases; (2) pneumonectomy and repairing of leak in 2 cases; (3) esophagectomy through right thoracotomy and cervical reconstruction of the retrosternal esophagogastrostomy was performed in one case; (4) repairing of leak with pedicled esophagus wall in one case. Follow-up shows that the surgical treatment gives satisfactory results in these patients.     

Benign acquired bronchoesophageal fistula in an adult

A 45-year-old woman was admitted to our hospital with a diagnosis of bronchoesophageal fistula, after a barium esophagography performed in an annual medical check-up had demonstrated an esophageal diverticulum in the middle of the thoracic esophagus, having a narrow bridge between the right intermediate bronchi. The patient had a history of tuberculosis in her childhood, and chest radiography showed multiple calcified hilar lymph nodes. The fistula was observed on gastroenteroscopy and on bronchofiberscopy. The patient was treated surgically, with ligation and resection of the fistula. The fistulous tract was attached to a calcified hilar lymph node, and both the diverticulum and fistula were concluded to have been caused by the tuberculous lymphadenitis in her childhood. It is very uncommon to have bronchoesophageal fistula, caused by tuberculosis in childhood, diagnosed and treated surgically at more than forty years later.     

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.     

Congenital anal fistula with normal anus

Three patients with a congenital anovestibular/perineal fistula were treated at the Montreal Children's Hospital. Two females (one of East Indian and the other of Japanese origin) had anovestibular fistulae that became symptomatic in the first few months of life. The third patient, a boy of Korean descent, presented at 9 months of age with a chronically draining perineal fistula. During surgery, a small fistula tract was easily dissected out and excised. Microscopic examination showed a well-preserved rectal mucosa throughout the tract. Most male patients described to date had anourethral fistulae, often accompanied by other major anomalies such as esophageal atresia or renal malformations. We believe our patient is the first male to be described with a congenital perineal fistula; this suggests that some fistula-in-ano in male infants may be due to a congenital sinus that secondarily becomes infected and drains to the skin.     

成人先天性食管支气管瘘的诊断及外科治疗

目的 探讨成人先天性食管支气管瘘的临床诊断及外科治疗.方法 回顾性分析1990年5月至2010年8月确诊并手术的11例成人先天性食管支气管瘘患者的临床资料.其中男性7例,女性4例,年龄28～66岁,平均48.7岁.主要临床表现为间断咳嗽、咳痰10例,进流食呛咳6例,咯血6例,长期反复肺部感染伴低热4例,胸痛3例.病史5～36年,平均16.8年.本组患者术前均经食管造影,或同时结合食管镜、支气管镜检查证实为食管支气管瘘,术后病理进一步确诊.6例行右下肺叶切除术,1例行右中下肺叶切除术,3例行左下肺叶切除术,1例行左全肺切除术,瘘管的食管残端均经结扎、缝扎等处理.结果 10例术后痊愈出院,住院时间10～18 d,平均12.3 d.1例术后第8天发生食管瘘,经再次手术后治愈.术后11例患者均定期随访.3年生存率11/11,5年生存率9/11.结论 成人先天性食管支气管瘘临床少见,容易误诊,多体位食管造影是确诊的主要手段;外科手术是惟一的治愈方法.     

成人先天性食管支气管瘘的诊断及外科治疗

目的 探讨成人先天性食管支气管瘘的临床诊断及外科治疗.方法 回顾性分析1990年5月至2010年8月确诊并手术的11例成人先天性食管支气管瘘患者的临床资料.其中男性7例,女性4例,年龄28～66岁,平均48.7岁.主要临床表现为间断咳嗽、咳痰10例,进流食呛咳6例,咯血6例,长期反复肺部感染伴低热4例,胸痛3例.病史5～36年,平均16.8年.本组患者术前均经食管造影,或同时结合食管镜、支气管镜检查证实为食管支气管瘘,术后病理进一步确诊.6例行右下肺叶切除术,1例行右中下肺叶切除术,3例行左下肺叶切除术,1例行左全肺切除术,瘘管的食管残端均经结扎、缝扎等处理.结果 10例术后痊愈出院,住院时间10～18 d,平均12.3 d.1例术后第8天发生食管瘘,经再次手术后治愈.术后11例患者均定期随访.3年生存率11/11,5年生存率9/11.结论 成人先天性食管支气管瘘临床少见,容易误诊,多体位食管造影是确诊的主要手段;外科手术是惟一的治愈方法.     

成人先天性食管支气管瘘的诊断及外科治疗

目的 探讨成人先天性食管支气管瘘的临床诊断及外科治疗.方法 回顾性分析1990年5月至2010年8月确诊并手术的11例成人先天性食管支气管瘘患者的临床资料.其中男性7例,女性4例,年龄28～66岁,平均48.7岁.主要临床表现为间断咳嗽、咳痰10例,进流食呛咳6例,咯血6例,长期反复肺部感染伴低热4例,胸痛3例.病史5～36年,平均16.8年.本组患者术前均经食管造影,或同时结合食管镜、支气管镜检查证实为食管支气管瘘,术后病理进一步确诊.6例行右下肺叶切除术,1例行右中下肺叶切除术,3例行左下肺叶切除术,1例行左全肺切除术,瘘管的食管残端均经结扎、缝扎等处理.结果 10例术后痊愈出院,住院时间10～18 d,平均12.3 d.1例术后第8天发生食管瘘,经再次手术后治愈.术后11例患者均定期随访.3年生存率11/11,5年生存率9/11.结论 成人先天性食管支气管瘘临床少见,容易误诊,多体位食管造影是确诊的主要手段;外科手术是惟一的治愈方法.     

Bronchoesophageal fistula after endovascular repair of ruptured aneurysm of the descending thoracic aorta

Aortoesophageal fistula secondary to thoracic aneurysm is rare and is usually fatal without prompt surgical intervention. A 79-year-old man with significant comorbidities and previous cancer surgery was admitted on an emergency basis because of the suspicion of a ruptured thoracic aortic aneurysm. Computed tomographic scan followed by angiography demonstrated a ruptured thoracic aneurysm with aortoesophageal fistula. An endovascular stent graft repair was performed with successful exclusion of both aneurysm and fistula. On postoperative day 6, dyspnea and an isolated episode of hemoptysis occurred. Endoscopy revealed the presence of a bronchoesophageal fistula, which necessitated double exclusion of the esophagus and feeding jejunostomy. At 6 months, clinical, bronchoscopic, and computed tomographic scan follow-up showed complete sealing of the aneurysm and resolution of the bronchoesophageal fistula. At 9 months, the patient was still alive but refused to undergo substernal gastric bypass in an attempt to restore oral feeding. Endovascular repair seems promising as an emergent and palliative treatment of aortoesophageal fistula. To the best of our knowledge, this is the first case in which a bronchoesophageal fistula developed after successful endovascular repair of aortoesophageal fistula. The pathogenesis of this complications remains unclear.     

成人先天性食管支气管瘘的诊断及外科治疗

目的 探讨成人先天性食管支气管瘘的临床诊断及外科治疗.方法 回顾性分析1990年5月至2010年8月确诊并手术的11例成人先天性食管支气管瘘患者的临床资料.其中男性7例,女性4例,年龄28～66岁,平均48.7岁.主要临床表现为间断咳嗽、咳痰10例,进流食呛咳6例,咯血6例,长期反复肺部感染伴低热4例,胸痛3例.病史5～36年,平均16.8年.本组患者术前均经食管造影,或同时结合食管镜、支气管镜检查证实为食管支气管瘘,术后病理进一步确诊.6例行右下肺叶切除术,1例行右中下肺叶切除术,3例行左下肺叶切除术,1例行左全肺切除术,瘘管的食管残端均经结扎、缝扎等处理.结果 10例术后痊愈出院,住院时间10～18 d,平均12.3 d.1例术后第8天发生食管瘘,经再次手术后治愈.术后11例患者均定期随访.3年生存率11/11,5年生存率9/11.结论 成人先天性食管支气管瘘临床少见,容易误诊,多体位食管造影是确诊的主要手段;外科手术是惟一的治愈方法.

Abstract：

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.     

An adult congenital bronchoesophageal fistula with massive hemoptysis; report of a case

A 67-year-old female was referred to our hospital because of bronchoesophageal fistula detected by upper gastro-intestinal series for cancer screening. The patient has had a history of coughing on liquid ingestion since childhood and she has been hospitalized 4 times for treatment of pneumonia during the past 20 years. While waiting the treatment, she was emergently admitted to the hospital because of massive hemoptysis. Transcatheter embolization of feeding arteries including the right inferior phrenic artery successfully controlled her hemoptysis. After reembolization of the feeding arteries for preventing massive hemorrhage during operation, posterolateral thoracotomy was performed. Surgical findings disclosed the bronchoesophageal fistula without inflammatory changes. She underwent fistulectomy combined resection of the middle and lower lobes which were destroyed by the repeated pneumonia. This case was considered type I congenital bronchoesophageal fistula according to Braimbridge and Keith classification because of the presence of diverticular projection which connected to the bronchus. Early diagnosis and rapid treatment are thought to be important for treating this disease.     

成人先天性气管、支气管食管瘘的外科治疗

目的 探讨成人先天性气管、支气管食管瘘的外科治疗经验.方法 回顾性分析1969年至2007年10月,14例成人先天性气管、支气管食管瘘的外科治疗资料.瘘管多位于食管下段和下叶肺叶支气管之间.手术方法主要为瘘管切除或瘘管加肺叶切除.结果 术后症状均好转,无围术期死亡者.结论 成人先天性气管、支气管食管瘘可经食管造影、支气管镜检等确诊;手术治疗效果良好.     

An adult case of congenital esophagobronchial fistula accompanied with bronchial branch of the vagal nerve

A case of congenital esophagobronchial fistula in a 73-year-old woman was described. The patient had presented with repeated attacks of pulmonary infection from her childhood and had a several years history of recurrent cough after eating or drinking. Preoperative examinations including esophagogram by barium swallow revealed existence of a fistula between middle thoracic esophagus and right B6 bronchus. The fistula was recognized as Type II of Braimbridge and Keith's classification. The fistula was surgically removed with the atelectatic right lower lobe. Neither adhesion nor enlargement of lymph nodes was found in the surrounding tissues. Because the bronchial branch of vagus was found along the fistula, this lesion was strongly suggested of congenital origin. Histological observations of the resected fistula showed no chronic inflammatory changes, though muscular layers and transitional zone between squamous epithelium and bronchial epithelium were present. The differential diagnosis between the congenital and acquired types is sometimes very difficult. The existence of the bronchial branch of vagus along the fistula means that the fistula is a congenital type, definitely.     

Congenital respiratory-esophageal fistula in the adult. Report of nine cases and review of the literature.

Congenital tracheoesophageal or bronchoesophageal fistulas, if not associated with esophageal atresia, may not appear initially until adult life. Nine such cases (two tracheoesophageal and seven bronchoesophageal) are reported. The chief presenting symptoms were recurrent bouts of coughing, after drinking, and hemoptysis. In the majority of cases the duration of symptoms exceeded 15 years. The diagnosis was confirmed in seven patients by esophagography, in one patient by bronchoscopy, and in one patient the fistula was discovered incidentally during thoracotomy. The esophageal opening of the fistula was in the lower third in seven patients and in the middle third in two. Bronchoesophageal fistulas communicated with a segmental bronchus in four patients and with a main or lobar bronchus in three. Treatment involved excision of the fistula (five patients) or division and suturing (four patients). Postoperative follow-up revealed no long-term sequelae except persistent chronic respiratory failure in one patient. The respiratory failure had developed before treatment of the fistula. The analysis of this series and a review of the literature underline the high index of suspicion required in all cases of chronic cough and lung suppuration, to diagnose this benign condition before life-threatening complications occur.     

Thoracoscopic approach for congenital bronchoesophageal fistula in an adult

We present a case of a congenital bronchoesophageal fistula in an adult male who underwent video-assisted thoracic surgery for a resection of the fistula. The patient had not suffered from any serious respiratory infection since the adolescence. However, at 49 years old, the patient experienced persistent cough and back pain. An abnormal shadow in the right lower lobe was observed on a chest X-ray. Chest computed tomography scanning indicated bronchiectasia in the lower superior segment and an abnormal air duct in the posterior mediastinum. Esophagography revealed a 4-cm-long and 1-cm-diameter fistula between the midesophagus and the right lower lobe. Esophagoscopy and bronchoscopy revealed the orifice of the fistula. Three-dimensional computed tomography scanning demonstrated that there was no abnormal artery supplying blood to the affected lung. He underwent video-assisted thoracic surgery, and was uneventfully discharged. Thoracoscopy offered excellent anatomical visualization of the fistula and safe surgical resection.