Congenital H-type tracheoesophageal fistula: a national multicenter study
Tóm tắt
Congenital H-type tracheoesophageal fistula (TEF) is very rare and represents <5 % of all congenital tracheoesophageal malformations. This is a national, multicenter review of our experience with isolated H-type TEF outlining clinical presentation, methods of diagnosis, associated anomalies, treatment and outcome The medical records of all patients with the diagnosis of congenital H-type TEF treated at four pediatric surgery units in Saudi Arabia were retrospectively reviewed for: age at diagnosis, sex, presenting symptoms, associated anomalies, method of diagnosis, treatment and outcome. During the study period (January 1998–December 2013), 435 infants and children with the diagnosis of esophageal atresia with or without TEF were treated. Among these, 23 (5.3 %) had isolated TEF. There were 11 males and 12 females. Their age at presentation ranged from 5 days to 3 years and 7 months but the majority (90 %) were diagnosed during their first year of life. Their clinical presentation included: chocking and coughing during feeds in 12 (52.2 %), recurrent chest infection in 16 (69.6 %) and cyanosis in 10 (43.5 %). One presented with abdominal distension also. The diagnosis was made using esophagogram. In 11 (47.8 %), a single study confirmed the diagnosis, 8 (34.8 %) required two studies while 4 (17.4 %) required three studies. Nineteen (82.6 %) had preoperative bronchoscopy and in 13 (56.5 %), a catheter was used to cannulate the fistula. All were operated through a right cervical incision except one who underwent thoracoscopic ligation and division of the fistula. In one, the fistula was only transfixed and tied without being divided. This patient developed a recurrent fistula. Two patients developed postoperative stridor secondary to recurrent laryngeal nerve palsy. In both of them, there was complete recovery. H-type TEF is very rare and commonly presents with recurrent chest infection, chocking and coughing during feeds and cyanosis. Physicians caring for these patients should be aware of this and a high index of suspicion is of paramount importance to avoid delay in diagnosis with its associated morbidity. A contrast esophagogram is valuable in confirming the diagnosis. The study however may need to be repeated. Preoperative bronchoscopy is valuable to localize and cannulate the fistula for easier access during surgery. Surgical repair is the treatment of choice and this should be performed through a right cervical incision or thoracotomy for low fistulae. Thoracoscopic ligation and division of a low H-type fistula is an alternative and less invasive approach when compared to thoracotomy.
Tài liệu tham khảo
Genty E, Attal P, Nicollas R, Roger G, Triglia JM, Garabedian EN, Bobin S (1999) Congenital tracheoesophageal fistula without esophageal atresia. Int J Pediatr Otorhinolaryngol 48:231–238
Crabbe D (2003) Isolated tracheo-oesophageal fistula. Pediatr Respir Rev 4:74–78
Biechlin A, Delattre A, Fayoux P (2008) Isolated congenital tracheoesophageal fistula. Retrospective analysis of 8 cases and review of the literature. Rev Laryngol Otol Rhinol 129:147–152
Harjai MM, Holla RG, Kale R, Sharma YK (2007) H-type tracheo-oesophageal fistula. Arch Dis Child Fetal Neonatal Ed 92(1):F40
Kirk JM, Dicks-Mireaux C (1989) Difficulties in diagnosis of congenital H-type tracheo-oesophageal fistulae. Clin Radiol 40:150–153
Stavroulias D, Ampollini L, Carbognani P, Rusca M (2011) Late presentation of congenital H-type tracheoesophageal fistula in an immunocompromised patient. Eur J Cardio-thoracic Surg 40(2):e98–e100
Hajjar WM, Iftikhar A, AlNassar SA, Rahal SM (2012) Congenital tracheoesophageal fistula: a rare and late presentation in adult patient. Ann Thorac Med 7(10):48–50
Sundar B, Guiney EJ, O’Donnell B (1975) Congenital H-type tracheo-oesophageal fistula. Arch Dis Child 50(11):862–863
Chueh H, Kim MJ, Jung JA (2008) A case of acute respiratory distress syndrome associated with congenital H-type tracheoesophageal fistula and gastroesophageal reflux. Korean J Pediatr. 51:892–895
Brookes JT, Smith MC, Smith RJ, Bauman NM, Manaligod JM, Sandler AD (2007) H-type congenital tracheoesophageal fistula: University Of Iowa experience 1985 to 2005. Ann Otol Rhinol laryngol. 116:363–368
Andrassy RJ, Ko P, Hanson BA, Kubota E, Hays DM, Mahour GM (1980) Congenital tracheo-esophageal fistula without esophageal atresia. A 22-year experience. Am J Surg 140:731–733
Nq J, Antao B, Bartram J, Raqhavan A, Shawis R (2006) Diagnostic difficulties in the management of H-type tracheoesophageal fistula. Acta Radiol 47:801–805
Karnak I, Senocak ME, Hicsonmez A, Buyukpamukcu N (1997) The diagnosis and treatment of H-Type tracheoesophageal fistula. J Pediatr Surg 32:1670–1674
Riazulhaq M, Elhassan E (2012) Early recognition of H-Type tracheoesophageal fistula. APSP J Case Rep 3(1):4
Le SD, Lam WW, Tam PK, Cheng W, Chan FL (2001) H-type tracheo-oesophageal fistula: appearance on three-dimensional computed tomography and virtual bronchoscopy. Pediatr Surg Int 17(8):642–643
Gunlemez A, Anik Y, Elemen L, Tugay M, Gökalp AS (2009) H-type tracheoesophageal fistula in an extremely low birth weight premature neonate: appearance on magnetic resonance imaging. J Perinatol 29(5):393–395
Beasley SW, Myers NA (1988) The diagnosis of congenital tracheo-oesophageal fistula. J Pediatr Surg 23:415–417
Spitz L, Kiely EM, Morecroft JA, Drake DP (1994) Oesophageal atresia: at-risk groups for the 1990s. J Pediatr Surg 29:723–725
Lamb DS (1873) A fatal case of congenital tracheoesophageal fistula. Phila Med Times 3:705
Killen DA, Greenlee HB (1965) Transcervical repair of H-type congenital tracheo-esophageal fistula: review of the literature. Ann Surg 162(1):145–150
Pigna A, Gentili A, Landuzzi V, Lima M, Baroncini S (2002) Bronchoscopy in newborns with esophageal atresia. Pediatr Med Chir 24(4):297–301
Antabak A, Luetic T, Calela D, Romic I (2014) H-type tracheoesophageal fistula in a newborn: determining the exact position of fistula by intra-operative guidewire placement. J Neonatal Surg 3(3):36–38
Amat F, Heraud MC, Scheye T, Canavese M, Labbe A (2012) Flexible bronchoscopic cannulation of an isolated H-type tracheoesophageal fistula in a newborn. J Pediatr Surg 47(10):e9–e10
Goyal A, Potter F, Losty PD (2005) Transillumination of H-type tracheoesophageal fistula using flexible miniature bronchoscopy: an innovative technique for operative localization. J Pediatr Surg 40:e33–e34
Haller JO, Berdon WE, Levin TL, Iyer KV (2004) Tracheoesophageal fistula (H-type) in neonates with imperforate anus and the VATER association. Pediatr Radiol 34(1):83–85
Imperatori CJ (1939) Congenital tracheo-esophageal fistula without atresia of the esophagus. Arch Otolaryngol 30:352
Parolini F, Morandi A, Macchini F et al (2014) Cervical/thoracotomic/thoracoscopic approaches for H-type congenital trachea-esophageal fistula: a systemic review. Int J Pediatr Otorhinolaryngol 78(7):985–989
Mattei P (2012) Double H-type tracheoesophageal fistulas idenrified and repaired in 1 operation. J Pediatr Surg 47(11):e11–e13
Tzifa KT, Maxwell EL, Chait P et al (2006) Endoscopic treatment of congenital H-Type and recurrent tracheoesophageal fistula with electrocautery and histoacryl glue. Int J Pediatr Otorhinolaryngol 70(5):925–930
Bhatnagar V, Lal VR, Sriniwas M, Agarwala S, Mitra DK (1999) Endoscopic treatment of tracheoesophageal fistula using electrocautery and the Nd:YAG laser. J Pediatr Surg 34(3):464–467
Allal H, Montes-Tapia F, Andina G (2004) Thoracoscopic repair of H-type tracheoesophageal fistula in the newborn: a technical case report. J Pediatr Surg 39:1568–1570
Aziz GA, Schier F (2005) Thoracoscopic ligation of a tracheoesophageal H-type fistula in a newborn. J Pediatr Surg 40(6):35–36