Research Article
Laparoscopic Fundoplication after Esophageal Atresia Repair
Scarpa MG*, Codrich D and Schleef J
Department of Pediatric Surgery, Institute for Maternal and Child Health - IRCCS “Burlo Garofolo”, Italy
*Corresponding author: Maria-Grazia Scarpa, Department of Pediatric Surgery, Institute for Maternal and Child Health - IRCCS “Burlo Garofolo”, Via dell’Istria 65/1, 34137 Trieste, Italy
Published: 01 Nov, 2018
Cite this article as: Scarpa MG, Codrich D, Schleef J.
Laparoscopic Fundoplication after
Esophageal Atresia Repair. Clin Surg.
2018; 3: 2195.
Abstract
To evaluate our experience with Gastro-Esophageal Reflux (GER) treatment after Esophageal
Atresia (EA) repair. Esophageal Atresia (EA) is a rare congenital malformation. A high incidence of
GER unresponsive to medical management is noted with EA. Literature suggests that complications
from GER can persist in adulthood.
In pediatric age, laparoscopic treatment is a valid option even if recurrence rate is not negligible. We
retrospectively analyzed 29 consecutive patients treated for EA at birth and studied for GER at our
Institute in a period of 11 years. 24/29 (82.7%) cases had symptoms of reflux, 17/29 (58.6%) cases
were treated with Laparoscopic Fundoplication (LF). Three infants were younger than 6 months
and had Apparent Life Threatening Events (ALTE) condition as principal indication for surgery.
No intra-operative complications occurred. 3/17 LF had opens surgical conversion due to technical
problems. 2/17 cases required a second operation. At the last follow-up: - 6/17 (35.3%) of patients
healed after the last operation!
Keywords: Esophageal atresia; Gastroesophageal reflux; Laparoscopic treatment
Abbreviations
EA: Esophageal Atresia; GER (D): Gastro-Esophageal Reflux (Disease); LES: Low Esophageal Sphincter; LF: Laparoscopic Fundoplication; ALTE: Apparent Life Threatening Events; G: Gastrostomy; J: Jejunostomy; CCAM: Congenital Cystic Adenomatoid Malformation; ARM: AnoRectal Malformation; TEF: Tracheo-Esophageal Fistula
Introduction
Esophageal Atresia (EA) is a rare congenital malformation (1: 3500 live births). Mortality rate after EA repair is low and depends on birth weight and other associated malformations, while long term morbidity is not to be neglected. Gastro-Esophageal Reflux (GER) is the most common long-term complication. The poor esophageal motility, the inadequate clearance of acid from the esophagus, the shortening of the esophagus and the abnormal Low Esophageal Sphincter (LES) pressure predispose patients to develop GER [1]. In literature, a clinical improvement after school age and adolescence is reported [2]. Due to many bias in the epidemiological studies, the actual prevalence of GER is not clear, it ranges from 25% to 75% of the cases [3,4] depending on anatomical changes of the gastroesophageal region after EA repair and on pathophysiological mechanisms like reduced esophageal clearance. Medical treatment with H2-antagonist, prokinetics and proton pump inhibitors can be efficient, but if no improvement occurs a surgical approach is suggested. Children with esophageal atresia have high incidence of symptomatic reflux that is not responsive to medical therapy [5]. According to Tovar more than 40% of these cases require surgical correction [6]. Among this group of patients, GER late recurrence rate after fundoplication procedures is high [7].
Materials and Methods
From January 2004 to September 2015, 29 consecutive patients treated for EA were studied
for GER at our Institute and were available for follow-up. GER study included: contrast X-ray,
endoscopy, 24 hours impendence-pH monitoring. The standard follow-up consisted on clinical
evaluations 1 and 3 months after AE repair, contrast X-ray studies after 6 and 18 months and,
endoscopic procedures after 12 and 24 months associated with esophageal 24 hour impedance-pH
monitoring when possible, clinical evaluations and/or repetition of the examinations once a year
in persistent GER cases. The medical records of all patients were reviewed and the demographic
and surgical data have been analyzed. Standard surgical treatment
consisted in a complete or partial Laparoscopic Fundoplication (LF).
Our aim was to evaluate the outcome of EA patients who received
GER surgical treatment.
A visit or a telephone call represented the last follow-up. We
considered:
• Resolution of GER
• Improvement of GER symptoms out of medical therapy
• Improvement of GER symptoms with medical therapy
• Recurrent GER requiring redo-operation.
We describe: EA type, associated conditions, age at surgery,
presence of gastrostomy or jejunostomy, indications and type of
surgical treatment.
Figure 1
Figure 1
Graphic representation of GER evolution among EA patients
surgically treated. The values are expressed in percentages.
Results
From January 2004 to September 2015, 29 EA patients were
studied for GER. 28/29 newborns had type III (Gross type C) three
of which were long gap form, 1/29 had type I (Gross type A) long gap
atresia. Both X-ray contrast and endoscopic studies were performed
in 25/29 cases; X-ray contrast studies, endoscopic procedures and
esophageal 24 hr impedance–pH monitoring studies were performed
in 14/29 cases. 18/29 children presented associated conditions like
prematurity, other malformations, presence of ventricular-peritoneal
derivation. 25/29 newborns belonged to group I according Spitz
risk classification: patients’ birth weight was >1500 gr without
major cardiac defect; 4/29 newborns belonged to group II having
a birth weight <1500 gr. 12/29 had a gastrostomy (ten cases) or
a jejunostomy (two cases). 24/29 patients (12 males, 12 females)
presented symptomatic GER (82.7%) and 17/29 cases (5 males and
12 females), were surgically treated (58.6%). All long gap EA needed
surgical therapy except for one (Gross C, III type). LF was the initial
approach in all patients and skilled surgeons did it at a median age of
37.3 months (range 4-204 months). 3/29 infants were younger than 6
months (weight between 4 and 8 kg) with Apparent Life Threatening
Events (ALTE) condition as principal indication for surgery. In all
cases, an evaluation to rule out other causes of respiratory difficulties
that could assume to be gastro-esophageal reflux was done. Surgical
treatment of GER consisted in 6/17 complete fundoplication (Nissen),
and 11/17 partial wrap (Thal or Toupet). 3/17 (17.6%) laparoscopic
procedures had open surgical conversion due to technical problems:
two of them were low weight infants, all three patients had with
gastrostomy or jejunostomy. In one case an important hepatomegaly
was the reason for conversion, in the second one anaesthesiologic
difficulties with poor compliance to pneumoperitoneum, in the third
the presence of gastrostomy and a small stomach. No intra-operative
complications occurred. All patients were fed after 6 hrs from the
surgical procedure and they were discharged from the hospital
between the 3rd and 12th day after the operation: the longest hospital
staying was not related to surgery.
2/17 (11.8%) patients with associated malformations required a
second fundoplication: one of them was performed in a low weight
infant (age <6 months) with ALTE, and presence of jejunostomy at
time of surgery.
At the last follow-up (visit or call phone):
• 6/17 (35.3%) of patients healed after the last operation
• 4/17 (23.5%) have GER improvement out of medical
therapy
• 4/17 (23.5%) have GER improvement still in medical
therapy
• 2/17 (11.8%) have recurrent GER
• 1/17 (5.9%) died for causes not related to antireflux surgery.
In summary 82.3% of patients had a good outcome after surgical
treatment. The average follow-up was 80 months (range 1 to 140
months). In Figure 1 and Figure 2 we resume our results.
Figure 2
Figure 2
Graphic representation of GER outcome among EA surgical group:
success rate (healing and improvement) and the failure rate (recurrence and
death) are highlighted. The values are expressed in percentages.
Discussion
Recent studies report a high percentage of GER complications
in the adult age causing GER Disease (GERD) in patients operated
of EA. In a Finnish adult study published in 2010, the occurrence of
symptomatic GER is 34% and the occurrence of dysphagia is 85%
compared with 8% and 2% respectively among general population
[8]. According to Rintala [9], in adulthood approximately one fifth of
EA patients develop epithelial metaplastic changes; one-third of these
have intestinal metaplasia (Barrett’s esophagus). It generally occurs
at a much younger age than general population [10]. In the long run,
these patients have 50-fold higher risk of carcinoma than the control
population [11].
According to Tovar [6] and to our experience more than 40%
of EA patients are refractory to medication and require surgical
correction. In a retrospective study published in 2010 by the Great
Ormond Street Hospital group, the laparoscopic GER operation
is considered an appropriate treatment for EA/GER. It is feasible
and effective even in children under one year old if performed by
skilled laparoscopic surgeons [12]. Anyway, other Authors report
increased fundoplication failure rates in small children [13]. In our
experience 3/17 cases (17.6%) were converted to open surgery: in all
3 cases a gastrostomy or a jejunostomy was present at the time of
surgery. 2/17 cases (11.7%) had a redo-operation: both were patients
with associated malformations; one of them was a small infant with
jejunostomy at the time of operation Kubiak et al. Observed a high
incidence of failure and redo fundoplication in infants with associated
anomalies, particularly those with EA: almost half of these patients
showed no improvement of their symptoms after fundoplication [14].
Tovar reports surgery failure rate in 30% of cases [11]. As far as the
type of LF is concerned, Snyder reports that a complete wrap (Nissen)
is inappropriate for GER associated with EA. Severe esophageal
dysmotility may contribute to poor passage of food through the
gastro-esophageal junction, especially against the increased resistance
provided by a complete wrap, so partial fundoplication could be
theoretically more attractive with a minor risk of prolonged dysphagia
[1]. In literature, a clinical GER improvement after school age and
adolescence is reported [2], anyway serious respiratory disorders
related to GER are a strong indication for surgery even in very small
babies [15]. Sload et al. in a systematic review in 2014 suggest that
anti-reflux surgery is an effective and safe treatment for severe reflux
related airway disease [16].
In our experience, one of the six Nissen operations required a
conversion to open surgery and another one failed and required a
redo-procedure: in both cases the patients were small infants with
associated conditions, one of these cases had a very short esophagus
due to a long gap Gross C, III type EA repair. Finally our GER
improvement rate after surgery was acceptable.
Conclusion
Laparoscopic treatment for GER after EA repair is efficient and feasible. According to literature the risk of recurrence after antireflux surgery is higher for low weigh infants compared with general population. Our number of cases is small, but in this retrospective series, low weigh infants younger than 6 months old can be initially treated with LF even if both, the risk of conversion to laparotomy and the rate of failure increase, especially for long gap cases. Associated conditions like malformations, presence of gastrostomy/jejunostomy seems to increase the risk. Anyway, surgery is mandatory for low weight infants with serious respiratory disease related to reflux refractory to medical therapy. While mortality for low risk EA patients is negligible, morbidity is not so low. All individuals who have undergone surgery for EA should be assessed clinically in the adolescence and adult age [17,18]. A careful surveillance and further studies will be necessary to understand the outcome of EA/RGE patients.
References
- Snyder CL, Ramachandran V, Kennedy AP, Gittes GK, Ashcraft KW, Holder TM. Efficacy of partial wrap fundoplication for gastroesophageal reflux after repair of esophageal atresia. J Pediatr Surg. 1997;32(7):1089-92.
- Somppi E, Tammela O, Ruuska T, Rahnasto J, Laitinen J, Turjanmaa V, et al. Outcome of patients operated on for esophageal atresia: 30 years’ experience. J Pediatr Surg. 1998;33(9):1341-6.
- Lima M, Ruggeri G, Domini M, Pelusi G. The gastroesophageal reflux secondary to malformations. Gastroesophageal reflux in infants and children. 2004:27-31.
- Gottrand F, Sfeir R, Coopman S. Atrésie de l’oesophage: devenir des enfants opérés. Arch Pediatr. 2008;15:1837-42.
- Curci M, Dibbins A. Gastroesophageal reflux in children: An underrated disease. Am J Surg. 1982;143(4):413-6.
- Tovar JA, Fragoso AC. Gastroesophageal reflux after repair of esophageal atresia. Eur J Pediatr Surg. 2013;23(3):175-81.
- Lindahl H, Rintala R, Louhimo I. Failure of the Nissen fundoplication to control gastroesophageal reflux in esophageal atresia patients. J Pediatr Surg. 1989; 24(10):985-7.
- Sistonen SJ, Koivusalo A, Nieminen U, Lindahl H, Lohi J, Kero M, et al. Esophageal morbidity and function in adults with repaired esophageal atresia with tracheoesophageal fistula. A population-based long-term follow-up. Ann Surg. 2010;251(6):1167-73.
- Rintala RJ, Pakarinen MP. Long-term outcome of esophageal anastomosis. Eur J Pediatr Surg. 2013:23(3):219-25.
- Vergouwe FW, IJsselstijn H, Wijnen RM, Bruno MJ, Spaander MC. Screening and surveillance in esophageal atresia patients: Current knowlwdge and future perspectives. Eur J Pediatr Surg. 2015; 25(4): 345-52.
- Tovar JA, Fragoso AC. Anti-reflux surgery for patients with esophageal atresia. Dis Esophagus. 2013;26(4):401-4.
- Shariff F, Kiely E, Curry J, Drake D, Pierro A, McHoney M. Outcome after laparoscopic fundoplication in children under 1 year. J Laparoendosc Adv Surg Tech A. 2010;20(7):661-4.
- Ponsky TA, Rothenberg SS. Minimally invasive surgery in infants less than 5 kg: Experience of 649 cases. Surg Endosc. 2008; 22(10):2214-9.
- Kubiak R, Spitz L, Kiely EM, Drake D, Pierro A. Effectiveness of fundoplication in early infancy. J Pediatr Surg. 1999:34(2):295-9.
- Mattioli G, Caffarena PE, Battistini E. Malattia da reflusso gastro-esofageo e patologia respiratoria. Ann Ital Chir. 1995;5:629-35.
- Sload RL, Brigger MT. Surgery for reflux induced airway disease: A systematic review. Int J Otorhinolaryngol. 2014;78(8):1211-5.
- Taylor AC, Breen KJ, Auldist A, Catto-Smith A, Clarnette T, Crameri J, et al. Gastroesophageal reflux and related pathology in adults who were born with esophageal atresia: Long term follow-up study. Clin Gastroenterol Hepatol. 2007;5(6):702-6.
- Connor MJ, Springford LR, Kapetanakis VV, Giuliani S. Esophageal atresia and transitional care-step 1: A systematic review and meta-analysis of the literature to define the prevalence of chronic long-term problems. Am J Surg. 2015;209(4):747-59.