Published online Sep 19, 2020. doi: 10.5409/wjcp.v9.i2.29
Peer-review started: May 28, 2020
First decision: June 15, 2020
Revised: June 28, 2020
Accepted: September 1, 2020
Article in press: September 1, 2020
Published online: September 19, 2020
Esophageal atresia (EA) is the most common congenital anomaly of the gastrointestinal tract. Esophageal dysmotility and gastroesophageal reflux disease (GERD) are frequent and lifelong problems after repair of EA, even after successful surgical repair of the esophagus anatomy. It is important to diagnose and manage GERD to reduce subsequent related respiratory and gastrointestinal problems and their associated short-term and long-term complications. GERD can be asymptomatic and several studies have shown the absence of correlation between symptoms and esophagitis in this population. All EA patients (including asymptomatic patients) should undergo monitoring of GER (impedance/pH-metry and/or endoscopy) at time of discontinuation of anti-acid treatment and during long-term follow-up.
In Belarus, a national follow-up program for EA patients has not yet been developed. So, these patients come to our clinic for examination when they have symptoms. Some of them did not experience any symptoms during pH-impedance monitoring. Before pH-impedance testing their parents reported symptoms spontaneously.
This study was designed to assess clinical symptoms and pH-impedance data in children after EA open surgical repair, and to compare with a control group of children with proven GERD in order to find specific GERD features in these patients and to provide data that will support development of a national program for the follow-up of EA patients. This was accomplished via a retrospective chart review of EA open surgical repair patients with GERD-related symptoms in our clinic from November 2017 to February 2020 using pH-impedance data, upper endoscopy data, medical records and clinic letters.
The main objectives of this study were to assess clinical symptoms and pH-impedance data in children with EA open surgical repair and to compare with a control group of children with proven GERD in order to identify specific features of reflux disease in these groups of patients. According to the results, we hope to develop a national program for the follow-up of EA patients and to personalize their treatment.
Patients with EA who received open surgical repair and combined impedance-pH testing while off proton pump inhibitor therapy and who underwent upper gastrointestinal endoscopy with histological study of mucosa biopsy samples were involved in the study. Data on patient symptoms were collected via a specially-prepared questionnaire for our study patients with GERD-related symptoms. We asked the parents of children (usually younger than 8 years) to fill out this questionnaire so that we could see what worries parents of children who cannot explain the symptoms that bother them. We used the index of proximal events (IPE), calculated as the ratio of the number of proximal refluxes to the total number of refluxes per day. We also determined distal mean nocturnal baseline impedance in all patients at the same distance depending on age (1 year to 10 years: 3 cm above the lower esophageal sphincter; older than 10 years: 5 cm above the lower esophageal sphincter).
We found a strong correlation with IPE and total proximal event in each EA group (EA with GERD: 0.96, P < 0.001; EA without GERD: 0.97, P < 0.001). The level of IPE in both EA groups was significantly lower than in GERD patients without any surgical treatment of esophagus (Kruskal-Wallis test, P < 0.001). Data on distal mean nocturnal baseline impedance in comparison of EA with GERD, EA without GERD, nonerosive reflux disease (commonly referred to as NERD) and reflux esophagitis (commonly referred to as RE) groups showed significant difference between EA with GERD (Kruskal-Wallis test, P < 0.001; one-way analysis of variance: F-ratio 6.69, P < 0.005) and the other two control groups but an absence of difference between EA without GERD, NERD and RE groups. We also found strong correlation with the IPE and total proximal events in each of the EA groups, and our data showed that the IPE in both EA groups was significantly lower than in GERD patients with non-operated esophagus.
Distal mean nocturnal baseline impedance has good diagnostic value for GERD in children with EA after open surgical repair, with cut-off of < 1.69 kOhm, and can be used as an indicator to design a personalized follow-up program for EA patients. The IPE might be an additional parameter of pH-impedance monitoring.
Not all patients were included in this study but only those who were treated for troublesome symptoms (after applying the exclusion criteria) and who had contacted our clinic over the past 3 years. In Belarus, a national follow-up program for EA patients has not yet been developed.
Our results confirm the importance of pH-impedance testing in EA patients in order to evaluate GERD and to individualize the treatment strategy for each patient. This finding has very important implications for the evaluation of GERD in symptomatic EA patients before prescribing antireflux medication and especially in the consideration of proceeding to fundoplication.
Although it is generally known that esophagus after atresia open surgical repair is restored anatomically, whether it is restored functionally remains unknown. Another important unknown for focus of future study is whether these motility disturbances will disappear with age? For such, correct and comprehensive follow-up of surgically-repaired EA patients (such as that designed upon the results of our study presented herein) is needed.