Original Research n Vascular
and Interventional Radiology
Note: This copy is for your personal non-commercial use only. To order presentation-ready copies for distribution to your colleagues or clients, contact us at www.rsna.org/rsnarights.
Fluoroscopic Balloon Dilation of Esophageal Atresia Anastomotic Strictures in Children and Young Adults: Single-Center Study of 103 Consecutive Patients from 1999 to 20111 Mandela Thyoka, MB, BS, MCS (ECSA), MRCS Ed Alex Barnacle, BM, MRCP, FRCR Samantha Chippington, BM, BS, BMedSci, MRCP, FRCR Simon Eaton, PhD David P. Drake, MA, MB, BChir, FRCS, DCH Kate M. K. Cross, FRACS Paolo De Coppi, MD, PhD Edward M. Kiely, FRCS Agostino Pierro, MD, FRCS Joseph I. Curry, FRCS Derek J. Roebuck, BMedSc, MB, BS, DMRD, FRCR, FRANZCR, MRCPH
Purpose:
To determine whether fluoroscopic balloon dilation (FBD) is a safe and effective method of treating esophageal anastomotic stricture after surgical repair in an unselected patient population.
Materials and Methods:
With ethics committee approval, records for 103 consecutive patients who underwent FBD with our interventional radiology service (1999–2011) were reviewed retrospectively. Patients underwent diagnostic contrast material– enhanced study prior to the first dilation. Dilations were performed by using general anesthesia. Outcomes were number and/or frequency of dilations, clinical effectiveness and response to dilations, esophageal perforation, requirement for surgery, and mortality. Data were expressed as mean 6 standard deviation (with range). Comparisons were conducted by using the Fisher exact test and log-rank test. The significance level was set at P , .05.
Results:
One hundred three patients (61 male patients, 59%) underwent 378 FBD sessions (median, two dilations per patient; range, 1–40 dilations). The median age at first FBD was 2.2 years (range, 0.1–19.5 years). The balloon catheter diameters ranged from 4 to 20 mm. FBD was successful in 93 patients (90%): 44 (47%) after single dilation and 49 (53%) after multiple dilations. There was no difference in the proportion of patients who required one dilation and were younger than 1 year versus those who were 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]). Ten patients (10%) required further procedures: Three underwent stent placement, three underwent esophageal stricture resection, and four underwent esophageal reconstruction. Four esophageal perforations (1%) developed after FBD. Antireflux surgery was performed in 18 patients (17%). There were no deaths.
Conclusion:
FBD for anastomotic strictures after esophageal atresia repair is feasible and acceptably safe and provides relief of symptoms in most patients (90%); however, about half require more than one dilation, and surgery is best predicted if more than 10 dilations are required.
1
From the Surgery Unit, University College London Institute of Child Health, 30 Guilford St, London WC1N 1EH, England (M.T., S.E., P.D.C., A.P.); and Departments of Radiology (A.B., S.C., D.J.R.) and Surgery (M.T., D.P.D., K.M.K.C., P.D.C., E.M.K., A.P., J.I.C.), Great Ormond Street Hospital for Children, London, England. Received October 9, 2012; revision requested December 10; revision received August 29, 2013; accepted September 17; final version accepted October 22. Address correspondence to M.T. (e-mail: [email protected]). RSNA, 2013
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radiology.rsna.org n Radiology: Volume 271: Number 2—May 2014
VASCULAR AND INTERVENTIONAL RADIOLOGY: Fluoroscopic Balloon Dilation of Esophageal Anastomotic Stricture
E
sophageal atresia (EA) is a common congenital anomaly of the esophagus, occurring in one in 2500 births (1). Although the treatment for EA has traditionally been primary or secondary surgical repair, postoperative morbidities occur, including anastomotic leak or stricture, recurrent fistula, gastroesophageal reflux (GER), tracheomalacia, and dysmotility (2– 4). In fact, anastomotic strictures are found in up to 50% of patients after EA repair (2). When symptomatic, esophageal strictures demonstrate obstructive symptoms, such as dysphagia and food bolus obstruction, and this may result in failure to thrive. The first treatment of EA strictures is dilation, which has traditionally been performed by using bougies (5). This technique may be associated with a higher rate of mucosal injury, which may contribute to worsening of the stricture. London et al (6) reported the first use of balloon dilation by using Gruentzig catheters in two patients with severe esophageal strictures; however, the first report in children was by Ball et al (7) in 1984. Balloon dilation
Advances in Knowledge nn In patients with anastomotic strictures after previous esophageal atresia repair, fluoroscopic balloon dilation (FBD) is successful in 90%. nn Half of all patients undergoing FBD will require two or more dilations to achieve complete relief. nn There is no difference in the proportion of patients who require one dilation and are younger than 1 year versus those who are 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]).
is considered advantageous over bouginage and is widely accepted as a safer and more effective alternative, as it uses radial forces to dilate the stricture, thus reducing the rate of mucosal injury (8). Balloon dilation can be achieved with endoscopic or fluoroscopic control. Several groups have described their experience with endoscopic balloon dilation. However, only a handful of reports on fluoroscopic balloon dilation (FBD) have been published so far (8–13). Antoniou et al (9) reported the largest endoscopic balloon dilation experience in 59 children with esophageal anastomotic stricture after surgical repair. Experience with FBD in children has been reported in smaller series. We hypothesized that FBD is a safe and effective method of treating esophageal anastomotic stricture after surgical repair in an unselected patient population.
Materials and Methods Ethics committee approval was obtained to undertake a retrospective review of records for 103 consecutive patients who underwent FBD for anastomotic strictures after previous EA repair with our interventional radiology service from 1999 to 2011. Patients with anastomotic strictures underwent diagnostic contrast material–enhanced study prior to the first dilation.
Esophageal Dilations Patients with symptomatic anastomotic strictures after surgical EA repair were referred to the interventional radiology service for FBD. Patients were assessed for anesthetic fitness, per American Society of Anesthesiologists score (14), and were scheduled to undergo an elective procedure. A standard protocol of ingesting nothing by mouth for a period
nn Requirement of more than 10 FBD sessions is a predictor of the need for surgery. nn Perforation rate after FBD is minimal (1.0%), and most of the perforations can be treated conservatively. Radiology: Volume 271: Number 2—May 2014 n radiology.rsna.org
Implications for Patient Care nn Although most patients respond to FBD successfully, several dilations may be needed to achieve compete relief of symptoms. nn Surgical intervention should be considered earlier in those who require multiple dilations.
Thyoka et al
of 6 hours before receiving anesthetic was observed, to reduce the risk of regurgitation of stomach contents and subsequent aspiration. Members of the interventional radiology department performed all balloon dilations in an angiography suite or operating room. All dilations were performed with general anesthesia involving endotracheal intubation, by using a standard technique (15). Patients were placed in the supine position, and the esophagus was visualized by using fluoroscopy and water-soluble contrast medium injected through a curved-tip angiographic catheter; a flexible guidewire was used to cross the esophageal stricture, and an angioplasty balloon catheter was inserted into the esophagus over the guidewire and placed across the stricture. Gradual balloon inflation was performed by using fluoroscopy to confirm that the “waist” was successfully overcome (Fig 1). The balloon size was selected by the operator, taking into account the size of the patient and the severity of the stricture, as depicted by luminal narrowing at fluoroscopy. The balloon was then deflated and withdrawn. An immediate postdilation contrast-enhanced study was performed at the discretion of the operator. Indications for this included difficulty crossing the stricture with the guidewire, excessive bleeding on the withdrawn balloon or from the
Published online before print 10.1148/radiol.13122184 Content codes: Radiology 2014; 271:596–601 Abbreviations: EA = esophageal atresia FBD = fluoroscopic balloon dilation GER = gastroesophageal reflux Author contributions: Guarantor of integrity of entire study, M.T.; study concepts/ study design or data acquisition or data analysis/interpretation, all authors; manuscript drafting or manuscript revision for important intellectual content, all authors; approval of final version of submitted manuscript, all authors; literature research, M.T., A.B., S.C., D.P.D., P.D.C., D.J.R.; clinical studies, M.T., A.B., S.C., D.P.D., K.M.K.C., E.M.K., J.I.C., D.J.R.; experimental studies, M.T.; statistical analysis, M.T., S.E., D.P.D.; and manuscript editing, M.T., A.B., S.C., D.P.D., K.M.K.C., P.D.C., E.M.K., A.P., J.I.C., D.J.R. Conflicts of interest are listed at the end of this article.
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Figure 1
Figure 1: FBD fluoroscopic images in a 2-year-old girl with an anastomotic stricture after repair of EA. (a) Preliminary contrast-enhanced image shows an anastomotic stricture (arrows) at the usual vertebral level (about T4). (b) The 14-mm balloon catheter has been advanced over the guidewire so that its radiopaque markers (arrows) are equidistant from the stricture, and the balloon catheter has been partially inflated with diluted contrast material by using a 10-mL syringe. (c) Further inflation confirms the presence of a resistant “waist.” (d) Forceful injection by hand (to about 600 kPa [6 atm]) abolishes the waist on the balloon.
effectiveness and response to dilations, esophageal perforation, requirement for surgery, and mortality.
Figure 2 Figure 2: Flowchart of the study population. * = Two patients had stent complications that required resection and anastomosis of the stricture.
patient’s mouth, severe tachycardia, or abrupt complete inflation of the balloon. The major complication was defined as esophageal perforation. All patients were extubated and allowed to recover fully from anesthesia prior to introducing the oral intake of a liquid or soft diet. Technical success was defined as 598
more than 50% increase in diameter at the stricture site after FBD, and clinical success was defined as achievement of a lumen adequate for relief of obstructive symptoms, as well as weight gain, growth, and the absence of procedural complications. Outcomes were number and/or frequency of dilations, clinical
Statistical Analysis The number of dilations required was dichotomized on the basis of age at the time of dilation: those younger than 1 year and those 1 year of age and older. Data were presented as mean 6 standard deviation (with range) as appropriate, and comparisons were made by using the Fisher exact test. Survival curves were constructed by using GraphPad Prism version 4.00 for Windows (GraphPad software, San Diego, Calif; www.graphpad.com). In the survival analysis of our study population, the requirement of the second FBD was defined as the event of interest, and the survival time was defined as the time (in months) from the first to the second FBD. Patients who did not require a second FBD were censored on the basis of their study time (length of time that the subjects were a part of the study). The survival curves between the two groups (patients younger than
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Figure 3
Table 1
Thyoka et al
Figure 4
Patient Characteristics Characteristic No. of patients No. of male patients Age at first dilation (y) No. of dilation sessions One dilation More than one dilation Stricture Diameter (mm) Length (mm) Most common vertebral level
Value 103 61 (59) 2.2 (0.1–19.5) 378 44 (43) 59 (57) 5.5 (0–14) 4 (1–65) T4 (n = 53)
Note.—Data are numbers (with percentages in parentheses) or median values (with ranges in parentheses).
Table 2 Number of Strictures (n = 103) Stricture Type Single stricture Multiple strictures Two strictures Three strictures
No. of Strictures (%) 95 (92) 8 (8) 7 1
1 year versus those 1 year of age and older) were compared by using the logrank test. The significance level was set at P , .05.
Results One hundred three patients (61 male patients, 59%) were treated consecutively with FBD for anastomotic strictures after surgical repair of EA during the study period (Fig 2). In total, 378 balloon dilation sessions were performed (median, two dilations per patient; range, 1–40 dilations). The median age at first FBD was 2.2 years (range, 0.1–19.5 years). The diameters of the balloon catheters used ranged from 4 to 20 mm (Table 1).
Strictures At fluoroscopy, 95 of 103 patients (92%) had a single stricture, and eight of 103 (8%) had more than one stricture (Table 2). The median stricture
Figure 3: Bar graph shows distribution of dilation sessions.
Figure 4: Bar graph shows number of dilations versus age at first dilation.
measurements were a diameter of 5.5 mm (range, 0–14 mm) and a length of 4 mm (range, 1–65 mm). The commonest vertebral level for a stricture was T4 (n = 53, 52%).
third perforation required surgical intervention for sepsis control. The perforation rate after FBD in our study was 1.0% (four of 378 FBD sessions).
Dilation Success One or more FBD procedures produced clinical success in 93 patients (90%), while 10 patients (10%) required further interventions. In 44 patients (43%), there was complete resolution of symptoms after the first FBD, without the need for further dilations; however, 59 patients (57%) had a recurrence of symptoms that necessitated further dilations (Fig 3). Symptoms recurred at a median time of 24 months. In those requiring more than a single dilation, 15 patients (14%) needed more than five FBD procedures. There was no difference in the proportion of patients who required one dilation and were younger than 1 year versus those who were 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]) Fig 4) or the effectiveness of the index (first) FBD procedure by using the log-rank test (P = .70; Fig 5). Perforations Two patients (2%) developed up to four esophageal perforations after FBD (Fig 6). One patient had a single perforation, which responded to conservative treatment, and the second patient had three perforations at different dilation sessions. In the latter case, the first two perforations were amenable to conservative therapy; however, the
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GER Disease Eighteen patients (17%) required antireflux surgery for GER disease that was refractory to maximal medical treatment. All of these patients underwent Nissen fundoplication. Esophageal Stent Placement and Surgery Ten patients (10%) required further intervention after FBD (Fig 1). A stent was inserted in three patients for management of the stricture; however, two subsequently required reconstructive surgery owing to stent complications. Esophageal reconstructive surgery was also undertaken in another four patients (six patients underwent esophageal reconstructive surgery). Lastly, three patients underwent resection of the esophageal anastomotic stricture. All patients (n = 10) who underwent esophageal surgery did so after 10 or more dilation sessions. In these patients, the indications for surgical intervention were varied and included stent complications, uncontrolled reflux, recurrent stricture that affected quality of life, complications of FBD (perforation of the esophagus with sepsis), and failure to thrive. Mortality We observed no mortality related directly to FBD or any complication of EA in this cohort. The median follow-up 599
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Figure 5
Figure 5: Kaplan-Meier curves illustrate the long-term effectiveness of the first FBD, as determined by the requirement of a second dilation. (a) Curve generated for all patients. (b) Curves generated for patients younger than 1 year and those 1 year of age and older at first dilation.
Figure 6
Figure 6: Fluoroscopic image shows esophageal rupture (arrow) after FBD in a 4-year-old boy with recurrent anastomotic stricture after repair of EA.
of our patients was 1.9 years (range, 0.5–11 years).
Discussion Esophageal stricture is a common complication after surgical repair of EA (2,4). The symptoms are mainly 600
obstructive, with dysphagia and inability to feed, which results in a failure to thrive. Dilation of the stricture reestablishes an adequate lumen and facilitates normal enteral feeding. Historically, dilation of the strictures was performed with endoscopic visualization, by using bougies. The use of FBD, however, has been increasing. The main perceived advantage of FBD over bouginage is the decreased risk of perforation, as FBD involves the use of a balloon device that applies radial pressure to dilate the stricture. Another advantage is the ability to observe full expansion of the balloon. We have shown FBD to be successful in 90% of our patients, with a median of two dilations needed per patient. Lang et al (8) showed superior results of balloon dilation over bouginage in terms of the median number of dilation sessions (two [range, 1–7] balloon dilations vs nine [range, 1–60] bouginages per patient) and fewer complications. In addition, our study shows that half of the patients only required a single dilation session. Esophageal perforation is the most serious common complication after balloon dilation. Two (2%) of our patients developed perforations after FBD, with a perforation rate per procedure of 1.0%. While most of our perforations were amenable to conservative
treatment, surgery was still required in one patient. Our perforation rate is similar to that of Said et al (12), who reported two perforations from 115 FBD sessions (1.7%) among 25 children (12). Ko et al (11), however, showed a higher perforation rate in their review of 29 patients: Three children (10%) had transmural perforations from 44 balloon dilation sessions (perforation rate of 6.8%). The incidence of anastomotic strictures undergoing surgical repair of EA with or without tracheoesophageal fistula has been reported to be 37% (74 of 199 patients), with the most significant predisposing factors being the use of braided silk sutures, the presence of GER, and leakage at the anastomosis (2). Most patients (70 of 74, 94%) with anastomotic strictures responded to dilation alone, and antireflux surgery was performed in 19 (26%) of the 74 patients. Said et al (12) evaluated 25 patients undergoing FBD and found 15 (60%) to have GER, with antireflux surgery required in 11 (44%) of the children (12). Our study, however, shows that only 17% of patients undergoing FBD for anastomotic strictures had symptomatic GER refractory to medical therapy, and they required antireflux surgery. GER can contribute to worsening of the anastomotic stricture and/ or development of further strictures.
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Indeed, as reported previously by Chittmittrapap et al (2), most of the strictures resolve with fewer dilations after antireflux surgery. In our series, a single FBD session achieved success in 43% of patients. It was possible to treat the stricture successfully in most patients who needed more than one dilation session. Only 10% were resistant to FBD and required further interventions, such as stent placement and/or esophageal replacement therapy. Best et al (16) reported the use of retrievable, covered stents in seven children (aged 0.5 to 7 years) with benign strictures, in whom six showed apparent benefit from the stent, and one underwent emergency retrieval of the stent as a result of respiratory distress. We placed a stent in three (3%) of our patients, of whom two required reconstructive surgery. Indications for stent placement in EA may include symptom control before surgical revision of the anastomosis and treatment of esophageal perforations. It is important to choose the size of the stent carefully in children with EA, because of the risk of compressing the typically malacic trachea. We practice selective dilation for symptomatic esophageal strictures, as advocated by Koivusalo et al (17), because this method has been shown to be superior in reducing the number and frequency of dilations. Subsequent dilations are based on clinical response and need. Our report constitutes the largest series of FBD in patients with symptomatic esophageal anastomotic strictures after surgical repair of EA. The most important limitation of our study is its retrospective design. Although we have no comparative group (for example, patients with strictures treated with endoscopic dilation), it seems unlikely that a randomized controlled trial of these techniques will now be performed.
In conclusion, FBD for anastomotic strictures after EA repair is feasible and acceptably safe and provides relief of symptoms in most patients (90%); however, about half require more than one dilation, and surgery is best predicted if more than 10 dilations are required. Disclosures of Conflicts of Interest: M.T. No relevant conflicts of interest to disclose. A.B. No relevant conflicts of interest to disclose. S.C. No relevant conflicts of interest to disclose. S.E. No relevant conflicts of interest to disclose. D.P.D. No relevant conflicts of interest to disclose. K.M.K.C. No relevant conflicts of interest to disclose. P.D.C. No relevant conflicts of interest to disclose. E.M.K. No relevant conflicts of interest to disclose. A.P. No relevant conflicts of interest to disclose. J.I.C. No relevant conflicts of interest to disclose. D.J.R. No relevant conflicts of interest to disclose.
References 1. Spitz L. Esophageal atresia: lessons I have learned in a 40-year experience. J Pediatr Surg 2006;41(10):1635–1640. 2. Chittmittrapap S, Spitz L, Kiely EM, Brereton RJ. Anastomotic stricture following repair of esophageal atresia. J Pediatr Surg 1990;25(5):508–511. 3. Chittmittrapap S, Spitz L, Kiely EM, Brereton RJ. Anastomotic leakage following surgery for esophageal atresia. J Pediatr Surg 1992;27(1):29–32. 4. Spitz L. Oesophageal atresia. Orphanet J Rare Dis 2007;2:24. 5. Serhal L, Gottrand F, Sfeir R, et al. Anastomotic stricture after surgical repair of esophageal atresia: frequency, risk factors, and efficacy of esophageal bougie dilatations. J Pediatr Surg 2010;45(7):1459–1462. 6. London RL, Trotman BW, DiMarino AJ Jr, et al. Dilatation of severe esophageal strictures by an inflatable balloon catheter. Gastroenterol 1981;80:173–175. 7. Ball WS, Strife JL, Rosenkrantz J, Towbin RB, Noseworthy J. Esophageal strictures in children: treatment by balloon dilatation. Radiology 1984;150:263–264.
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8. Lang T, Hümmer HP, Behrens R. Balloon dilation is preferable to bougienage in children with esophageal atresia. Endoscopy 2001;33(4):329–335. 9. Antoniou D, Soutis M, Christopoulos-Geroulanos G. Anastomotic strictures following esophageal atresia repair: a 20-year experience with endoscopic balloon dilatation. J Pediatr Gastroenterol Nutr 2010;51(4):464– 467. 10. Johnsen A, Jensen LI, Mauritzen K. Balloon-dilatation of esophageal strictures in children. Pediatr Radiol 1986;16(5):388– 391. 11. Ko HK, Shin JH, Song HY, et al. Balloon dilation of anastomotic strictures secondary to surgical repair of esophageal atresia in a pediatric population: long-term results. J Vasc Interv Radiol 2006;17(8):1327–1333. 12. Said M, Mekki M, Golli M, et al. Balloon dilatation of anastomotic strictures secondary to surgical repair of oesophageal atresia. Br J Radiol 2003;76(901):26–31. 13. Lan LC, Wong KK, Lin SC, et al. Endo scopic balloon dilatation of esophageal strictures in infants and children: 17 years’ experience and a literature review. J Pediatr Surg 2003;38(12):1712–1715. 14. Saklad M. Grading of patients for surgical procedures. Anesthesiology 1941;2(3):281– 284. 15. Ade-Ajayi N, Roebuck D, Spitz L. Esophageal dilatation. In: Najmaldin AR, Rothenberg S, Crabbe D, Beasley S, eds. Operative endoscopy and endoscopic surgery in infants and children. London, England: Hodder Arnold, 2005; 127–133. 16. Best C, Sudel B, Foker JE, Krosch TC, Dietz C, Khan KM. Esophageal stenting in children: indications, application, effectiveness, and complications. Gastrointest Endosc 2009;70(6):1248–1253. 17. Koivusalo A, Pakarinen MP, Rintala RJ. Anastomotic dilatation after repair of esophageal atresia with distal fistula: comparison of results after routine versus selective dilatation. Dis Esophagus 2009;22(2):190–194.
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and Interventional Radiology
Note: This copy is for your personal non-commercial use only. To order presentation-ready copies for distribution to your colleagues or clients, contact us at www.rsna.org/rsnarights.
Fluoroscopic Balloon Dilation of Esophageal Atresia Anastomotic Strictures in Children and Young Adults: Single-Center Study of 103 Consecutive Patients from 1999 to 20111 Mandela Thyoka, MB, BS, MCS (ECSA), MRCS Ed Alex Barnacle, BM, MRCP, FRCR Samantha Chippington, BM, BS, BMedSci, MRCP, FRCR Simon Eaton, PhD David P. Drake, MA, MB, BChir, FRCS, DCH Kate M. K. Cross, FRACS Paolo De Coppi, MD, PhD Edward M. Kiely, FRCS Agostino Pierro, MD, FRCS Joseph I. Curry, FRCS Derek J. Roebuck, BMedSc, MB, BS, DMRD, FRCR, FRANZCR, MRCPH
Purpose:
To determine whether fluoroscopic balloon dilation (FBD) is a safe and effective method of treating esophageal anastomotic stricture after surgical repair in an unselected patient population.
Materials and Methods:
With ethics committee approval, records for 103 consecutive patients who underwent FBD with our interventional radiology service (1999–2011) were reviewed retrospectively. Patients underwent diagnostic contrast material– enhanced study prior to the first dilation. Dilations were performed by using general anesthesia. Outcomes were number and/or frequency of dilations, clinical effectiveness and response to dilations, esophageal perforation, requirement for surgery, and mortality. Data were expressed as mean 6 standard deviation (with range). Comparisons were conducted by using the Fisher exact test and log-rank test. The significance level was set at P , .05.
Results:
One hundred three patients (61 male patients, 59%) underwent 378 FBD sessions (median, two dilations per patient; range, 1–40 dilations). The median age at first FBD was 2.2 years (range, 0.1–19.5 years). The balloon catheter diameters ranged from 4 to 20 mm. FBD was successful in 93 patients (90%): 44 (47%) after single dilation and 49 (53%) after multiple dilations. There was no difference in the proportion of patients who required one dilation and were younger than 1 year versus those who were 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]). Ten patients (10%) required further procedures: Three underwent stent placement, three underwent esophageal stricture resection, and four underwent esophageal reconstruction. Four esophageal perforations (1%) developed after FBD. Antireflux surgery was performed in 18 patients (17%). There were no deaths.
Conclusion:
FBD for anastomotic strictures after esophageal atresia repair is feasible and acceptably safe and provides relief of symptoms in most patients (90%); however, about half require more than one dilation, and surgery is best predicted if more than 10 dilations are required.
1
From the Surgery Unit, University College London Institute of Child Health, 30 Guilford St, London WC1N 1EH, England (M.T., S.E., P.D.C., A.P.); and Departments of Radiology (A.B., S.C., D.J.R.) and Surgery (M.T., D.P.D., K.M.K.C., P.D.C., E.M.K., A.P., J.I.C.), Great Ormond Street Hospital for Children, London, England. Received October 9, 2012; revision requested December 10; revision received August 29, 2013; accepted September 17; final version accepted October 22. Address correspondence to M.T. (e-mail: [email protected]). RSNA, 2013
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radiology.rsna.org n Radiology: Volume 271: Number 2—May 2014
VASCULAR AND INTERVENTIONAL RADIOLOGY: Fluoroscopic Balloon Dilation of Esophageal Anastomotic Stricture
E
sophageal atresia (EA) is a common congenital anomaly of the esophagus, occurring in one in 2500 births (1). Although the treatment for EA has traditionally been primary or secondary surgical repair, postoperative morbidities occur, including anastomotic leak or stricture, recurrent fistula, gastroesophageal reflux (GER), tracheomalacia, and dysmotility (2– 4). In fact, anastomotic strictures are found in up to 50% of patients after EA repair (2). When symptomatic, esophageal strictures demonstrate obstructive symptoms, such as dysphagia and food bolus obstruction, and this may result in failure to thrive. The first treatment of EA strictures is dilation, which has traditionally been performed by using bougies (5). This technique may be associated with a higher rate of mucosal injury, which may contribute to worsening of the stricture. London et al (6) reported the first use of balloon dilation by using Gruentzig catheters in two patients with severe esophageal strictures; however, the first report in children was by Ball et al (7) in 1984. Balloon dilation
Advances in Knowledge nn In patients with anastomotic strictures after previous esophageal atresia repair, fluoroscopic balloon dilation (FBD) is successful in 90%. nn Half of all patients undergoing FBD will require two or more dilations to achieve complete relief. nn There is no difference in the proportion of patients who require one dilation and are younger than 1 year versus those who are 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]).
is considered advantageous over bouginage and is widely accepted as a safer and more effective alternative, as it uses radial forces to dilate the stricture, thus reducing the rate of mucosal injury (8). Balloon dilation can be achieved with endoscopic or fluoroscopic control. Several groups have described their experience with endoscopic balloon dilation. However, only a handful of reports on fluoroscopic balloon dilation (FBD) have been published so far (8–13). Antoniou et al (9) reported the largest endoscopic balloon dilation experience in 59 children with esophageal anastomotic stricture after surgical repair. Experience with FBD in children has been reported in smaller series. We hypothesized that FBD is a safe and effective method of treating esophageal anastomotic stricture after surgical repair in an unselected patient population.
Materials and Methods Ethics committee approval was obtained to undertake a retrospective review of records for 103 consecutive patients who underwent FBD for anastomotic strictures after previous EA repair with our interventional radiology service from 1999 to 2011. Patients with anastomotic strictures underwent diagnostic contrast material–enhanced study prior to the first dilation.
Esophageal Dilations Patients with symptomatic anastomotic strictures after surgical EA repair were referred to the interventional radiology service for FBD. Patients were assessed for anesthetic fitness, per American Society of Anesthesiologists score (14), and were scheduled to undergo an elective procedure. A standard protocol of ingesting nothing by mouth for a period
nn Requirement of more than 10 FBD sessions is a predictor of the need for surgery. nn Perforation rate after FBD is minimal (1.0%), and most of the perforations can be treated conservatively. Radiology: Volume 271: Number 2—May 2014 n radiology.rsna.org
Implications for Patient Care nn Although most patients respond to FBD successfully, several dilations may be needed to achieve compete relief of symptoms. nn Surgical intervention should be considered earlier in those who require multiple dilations.
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of 6 hours before receiving anesthetic was observed, to reduce the risk of regurgitation of stomach contents and subsequent aspiration. Members of the interventional radiology department performed all balloon dilations in an angiography suite or operating room. All dilations were performed with general anesthesia involving endotracheal intubation, by using a standard technique (15). Patients were placed in the supine position, and the esophagus was visualized by using fluoroscopy and water-soluble contrast medium injected through a curved-tip angiographic catheter; a flexible guidewire was used to cross the esophageal stricture, and an angioplasty balloon catheter was inserted into the esophagus over the guidewire and placed across the stricture. Gradual balloon inflation was performed by using fluoroscopy to confirm that the “waist” was successfully overcome (Fig 1). The balloon size was selected by the operator, taking into account the size of the patient and the severity of the stricture, as depicted by luminal narrowing at fluoroscopy. The balloon was then deflated and withdrawn. An immediate postdilation contrast-enhanced study was performed at the discretion of the operator. Indications for this included difficulty crossing the stricture with the guidewire, excessive bleeding on the withdrawn balloon or from the
Published online before print 10.1148/radiol.13122184 Content codes: Radiology 2014; 271:596–601 Abbreviations: EA = esophageal atresia FBD = fluoroscopic balloon dilation GER = gastroesophageal reflux Author contributions: Guarantor of integrity of entire study, M.T.; study concepts/ study design or data acquisition or data analysis/interpretation, all authors; manuscript drafting or manuscript revision for important intellectual content, all authors; approval of final version of submitted manuscript, all authors; literature research, M.T., A.B., S.C., D.P.D., P.D.C., D.J.R.; clinical studies, M.T., A.B., S.C., D.P.D., K.M.K.C., E.M.K., J.I.C., D.J.R.; experimental studies, M.T.; statistical analysis, M.T., S.E., D.P.D.; and manuscript editing, M.T., A.B., S.C., D.P.D., K.M.K.C., P.D.C., E.M.K., A.P., J.I.C., D.J.R. Conflicts of interest are listed at the end of this article.
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Figure 1
Figure 1: FBD fluoroscopic images in a 2-year-old girl with an anastomotic stricture after repair of EA. (a) Preliminary contrast-enhanced image shows an anastomotic stricture (arrows) at the usual vertebral level (about T4). (b) The 14-mm balloon catheter has been advanced over the guidewire so that its radiopaque markers (arrows) are equidistant from the stricture, and the balloon catheter has been partially inflated with diluted contrast material by using a 10-mL syringe. (c) Further inflation confirms the presence of a resistant “waist.” (d) Forceful injection by hand (to about 600 kPa [6 atm]) abolishes the waist on the balloon.
effectiveness and response to dilations, esophageal perforation, requirement for surgery, and mortality.
Figure 2 Figure 2: Flowchart of the study population. * = Two patients had stent complications that required resection and anastomosis of the stricture.
patient’s mouth, severe tachycardia, or abrupt complete inflation of the balloon. The major complication was defined as esophageal perforation. All patients were extubated and allowed to recover fully from anesthesia prior to introducing the oral intake of a liquid or soft diet. Technical success was defined as 598
more than 50% increase in diameter at the stricture site after FBD, and clinical success was defined as achievement of a lumen adequate for relief of obstructive symptoms, as well as weight gain, growth, and the absence of procedural complications. Outcomes were number and/or frequency of dilations, clinical
Statistical Analysis The number of dilations required was dichotomized on the basis of age at the time of dilation: those younger than 1 year and those 1 year of age and older. Data were presented as mean 6 standard deviation (with range) as appropriate, and comparisons were made by using the Fisher exact test. Survival curves were constructed by using GraphPad Prism version 4.00 for Windows (GraphPad software, San Diego, Calif; www.graphpad.com). In the survival analysis of our study population, the requirement of the second FBD was defined as the event of interest, and the survival time was defined as the time (in months) from the first to the second FBD. Patients who did not require a second FBD were censored on the basis of their study time (length of time that the subjects were a part of the study). The survival curves between the two groups (patients younger than
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Figure 3
Table 1
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Figure 4
Patient Characteristics Characteristic No. of patients No. of male patients Age at first dilation (y) No. of dilation sessions One dilation More than one dilation Stricture Diameter (mm) Length (mm) Most common vertebral level
Value 103 61 (59) 2.2 (0.1–19.5) 378 44 (43) 59 (57) 5.5 (0–14) 4 (1–65) T4 (n = 53)
Note.—Data are numbers (with percentages in parentheses) or median values (with ranges in parentheses).
Table 2 Number of Strictures (n = 103) Stricture Type Single stricture Multiple strictures Two strictures Three strictures
No. of Strictures (%) 95 (92) 8 (8) 7 1
1 year versus those 1 year of age and older) were compared by using the logrank test. The significance level was set at P , .05.
Results One hundred three patients (61 male patients, 59%) were treated consecutively with FBD for anastomotic strictures after surgical repair of EA during the study period (Fig 2). In total, 378 balloon dilation sessions were performed (median, two dilations per patient; range, 1–40 dilations). The median age at first FBD was 2.2 years (range, 0.1–19.5 years). The diameters of the balloon catheters used ranged from 4 to 20 mm (Table 1).
Strictures At fluoroscopy, 95 of 103 patients (92%) had a single stricture, and eight of 103 (8%) had more than one stricture (Table 2). The median stricture
Figure 3: Bar graph shows distribution of dilation sessions.
Figure 4: Bar graph shows number of dilations versus age at first dilation.
measurements were a diameter of 5.5 mm (range, 0–14 mm) and a length of 4 mm (range, 1–65 mm). The commonest vertebral level for a stricture was T4 (n = 53, 52%).
third perforation required surgical intervention for sepsis control. The perforation rate after FBD in our study was 1.0% (four of 378 FBD sessions).
Dilation Success One or more FBD procedures produced clinical success in 93 patients (90%), while 10 patients (10%) required further interventions. In 44 patients (43%), there was complete resolution of symptoms after the first FBD, without the need for further dilations; however, 59 patients (57%) had a recurrence of symptoms that necessitated further dilations (Fig 3). Symptoms recurred at a median time of 24 months. In those requiring more than a single dilation, 15 patients (14%) needed more than five FBD procedures. There was no difference in the proportion of patients who required one dilation and were younger than 1 year versus those who were 1 year of age and older (P . .99; odds ratio, 1.07 [range, 0.43–2.66]) Fig 4) or the effectiveness of the index (first) FBD procedure by using the log-rank test (P = .70; Fig 5). Perforations Two patients (2%) developed up to four esophageal perforations after FBD (Fig 6). One patient had a single perforation, which responded to conservative treatment, and the second patient had three perforations at different dilation sessions. In the latter case, the first two perforations were amenable to conservative therapy; however, the
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GER Disease Eighteen patients (17%) required antireflux surgery for GER disease that was refractory to maximal medical treatment. All of these patients underwent Nissen fundoplication. Esophageal Stent Placement and Surgery Ten patients (10%) required further intervention after FBD (Fig 1). A stent was inserted in three patients for management of the stricture; however, two subsequently required reconstructive surgery owing to stent complications. Esophageal reconstructive surgery was also undertaken in another four patients (six patients underwent esophageal reconstructive surgery). Lastly, three patients underwent resection of the esophageal anastomotic stricture. All patients (n = 10) who underwent esophageal surgery did so after 10 or more dilation sessions. In these patients, the indications for surgical intervention were varied and included stent complications, uncontrolled reflux, recurrent stricture that affected quality of life, complications of FBD (perforation of the esophagus with sepsis), and failure to thrive. Mortality We observed no mortality related directly to FBD or any complication of EA in this cohort. The median follow-up 599
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Figure 5
Figure 5: Kaplan-Meier curves illustrate the long-term effectiveness of the first FBD, as determined by the requirement of a second dilation. (a) Curve generated for all patients. (b) Curves generated for patients younger than 1 year and those 1 year of age and older at first dilation.
Figure 6
Figure 6: Fluoroscopic image shows esophageal rupture (arrow) after FBD in a 4-year-old boy with recurrent anastomotic stricture after repair of EA.
of our patients was 1.9 years (range, 0.5–11 years).
Discussion Esophageal stricture is a common complication after surgical repair of EA (2,4). The symptoms are mainly 600
obstructive, with dysphagia and inability to feed, which results in a failure to thrive. Dilation of the stricture reestablishes an adequate lumen and facilitates normal enteral feeding. Historically, dilation of the strictures was performed with endoscopic visualization, by using bougies. The use of FBD, however, has been increasing. The main perceived advantage of FBD over bouginage is the decreased risk of perforation, as FBD involves the use of a balloon device that applies radial pressure to dilate the stricture. Another advantage is the ability to observe full expansion of the balloon. We have shown FBD to be successful in 90% of our patients, with a median of two dilations needed per patient. Lang et al (8) showed superior results of balloon dilation over bouginage in terms of the median number of dilation sessions (two [range, 1–7] balloon dilations vs nine [range, 1–60] bouginages per patient) and fewer complications. In addition, our study shows that half of the patients only required a single dilation session. Esophageal perforation is the most serious common complication after balloon dilation. Two (2%) of our patients developed perforations after FBD, with a perforation rate per procedure of 1.0%. While most of our perforations were amenable to conservative
treatment, surgery was still required in one patient. Our perforation rate is similar to that of Said et al (12), who reported two perforations from 115 FBD sessions (1.7%) among 25 children (12). Ko et al (11), however, showed a higher perforation rate in their review of 29 patients: Three children (10%) had transmural perforations from 44 balloon dilation sessions (perforation rate of 6.8%). The incidence of anastomotic strictures undergoing surgical repair of EA with or without tracheoesophageal fistula has been reported to be 37% (74 of 199 patients), with the most significant predisposing factors being the use of braided silk sutures, the presence of GER, and leakage at the anastomosis (2). Most patients (70 of 74, 94%) with anastomotic strictures responded to dilation alone, and antireflux surgery was performed in 19 (26%) of the 74 patients. Said et al (12) evaluated 25 patients undergoing FBD and found 15 (60%) to have GER, with antireflux surgery required in 11 (44%) of the children (12). Our study, however, shows that only 17% of patients undergoing FBD for anastomotic strictures had symptomatic GER refractory to medical therapy, and they required antireflux surgery. GER can contribute to worsening of the anastomotic stricture and/ or development of further strictures.
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Indeed, as reported previously by Chittmittrapap et al (2), most of the strictures resolve with fewer dilations after antireflux surgery. In our series, a single FBD session achieved success in 43% of patients. It was possible to treat the stricture successfully in most patients who needed more than one dilation session. Only 10% were resistant to FBD and required further interventions, such as stent placement and/or esophageal replacement therapy. Best et al (16) reported the use of retrievable, covered stents in seven children (aged 0.5 to 7 years) with benign strictures, in whom six showed apparent benefit from the stent, and one underwent emergency retrieval of the stent as a result of respiratory distress. We placed a stent in three (3%) of our patients, of whom two required reconstructive surgery. Indications for stent placement in EA may include symptom control before surgical revision of the anastomosis and treatment of esophageal perforations. It is important to choose the size of the stent carefully in children with EA, because of the risk of compressing the typically malacic trachea. We practice selective dilation for symptomatic esophageal strictures, as advocated by Koivusalo et al (17), because this method has been shown to be superior in reducing the number and frequency of dilations. Subsequent dilations are based on clinical response and need. Our report constitutes the largest series of FBD in patients with symptomatic esophageal anastomotic strictures after surgical repair of EA. The most important limitation of our study is its retrospective design. Although we have no comparative group (for example, patients with strictures treated with endoscopic dilation), it seems unlikely that a randomized controlled trial of these techniques will now be performed.
In conclusion, FBD for anastomotic strictures after EA repair is feasible and acceptably safe and provides relief of symptoms in most patients (90%); however, about half require more than one dilation, and surgery is best predicted if more than 10 dilations are required. Disclosures of Conflicts of Interest: M.T. No relevant conflicts of interest to disclose. A.B. No relevant conflicts of interest to disclose. S.C. No relevant conflicts of interest to disclose. S.E. No relevant conflicts of interest to disclose. D.P.D. No relevant conflicts of interest to disclose. K.M.K.C. No relevant conflicts of interest to disclose. P.D.C. No relevant conflicts of interest to disclose. E.M.K. No relevant conflicts of interest to disclose. A.P. No relevant conflicts of interest to disclose. J.I.C. No relevant conflicts of interest to disclose. D.J.R. No relevant conflicts of interest to disclose.
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