Multicenter Evaluation with Eckardt Score of Laparoscopic Management with Heller Myotomy and Dor Fundoplication for Esophageal Achalasia in a Pediatric Population in Colombia

Abstract

Background:

Esophageal achalasia is a rare, chronic, and progressive neurodegenerative motility disorder that is characterized by a lack of relaxation of the lower esophageal sphincter. Laparoscopic Heller myotomy (LHM) is the ideal in our population. Multiple surgical and medical treatments have been raised. However, there has been a need to expand studies and generate a clear algorithm for an ideal therapeutic algorithm.

Methods:

Clinical record was retrospectively analyzed of patients who underwent LHM and Dor fundoplication evaluated with Eckardt score, at four Colombian medical centers between February 2008 and December 2018.

Results:

There were a total of 21 patients (12 males and 9 females, ages 8 months to 16 years). The time from onset of symptoms to surgery was between 5 months and 14 years. One patient had esophageal mucosa perforation, 2 patients were converted to open surgery, and 1 patient had a postoperative fistula.

All patients were discharged 3 to 9 days postoperatively, at which time they tolerated normal oral feeding. During follow-up, all the patients had an improvement in nutritional status and a greater functional recovery; 4 had reflux and 1 had reflux-like symptoms.

Conclusion:

LHM with Dor-type fundoplication maintains the effectiveness of open surgery with low postoperative morbidity and mortality and good functional results according to Eckardt score evaluation.

Introduction

Esophageal achalasia is a primary neurodegenerative disorder that is described as impaired motility of the distal esophagus with incompetence of the lower esophageal sphincter, due to degeneration of myenteric plexus, which results in partial or complete functional obstruction.^1–5

Esophageal achalasia incidence is 0.11 to 0.5 per 100,000 cases. Only 5% is present in the pediatric population.^6–9 It is more frequent in males, and <5% of patients who present symptoms are <15 years. The most common symptom is dysphagia.⁶

This entity can be associated with adrenal insufficiency and alacrimia, known as triple-A or Allgrove syndrome, <100 cases have been reported since its first known case in 1978.⁷

Given the low incidence of esophageal achalasia in the pediatric population, management aims to relieve symptoms.⁸ Nonoperative treatment includes botulinum toxin injection (BTI), pneumatic dilatation (PD), and oral calcium channel blockers.^6,8–10

Surgical treatment includes esophageal myotomy (Heller myotomy) with or without antireflux procedure and, most recently, per oral endoscopic myotomy (POEM).⁸ This study evaluates Heller myotomy and antireflux procedure for the management of esophageal achalasia in the pediatric population with the Eckardt score.

Methods

A retrospective analysis was performed on patients who underwent laparoscopic Heller myotomy (LHM) with Dor fundoplication for esophageal achalasia at four Colombian medical centers between February 2008 and June 2018.

All patients provided informed consent before the procedure. The study protocol was approved by the institution's institutional review board committee of each hospital. The protocol was implemented in accordance with the provisions of the Declaration of Helsinki and Good Clinical Practice guidelines.

Relevant data were collected from the patient's medical records. Information reviewed included age, gender, duration of symptoms, comorbidities, and diagnostic methods. Intra and postoperative data included surgical time, procedure type, complications, and hospital stay. Patients were clinically followed for a minimum of 1 year. All patients underwent a presurgical multidisciplinary assessment by the pediatric and pediatric surgery departments.

To ensure strict follow-up and avoid a gap in the continuity of clinical surveillance, all patients were provided with surgeon's personal email.

Preoperative patient assessment

In the first clinical evaluation, the Eckardt score was applied to objectify the symptoms of the patients, it allows to compare the effectiveness of the treatment.

The Eckardt score evaluates symptoms (chest pain, dysphagia, weight loss, and regurgitation) versus severity. The score is given as 0 for absence of symptoms, 1 for occasional and loss of <5 kg, 2 for daily and weight loss of 5 to 10 kg, and 3 points when the symptom is with every meal, chest pain several times a day, and weight loss >10 kg.

The result of the Eckardt score ranges from 0 to 12 points. Scores of 0 to 1 correspond to clinical stage 0; scores of 2–3 points correspond to stage I; scores of 4–6 points correspond to stage II; and scores of >6 points correspond to stage III.^11,12

A higher Eckardt score reflects more severe symptomatology, whereas a lower score after surgery indicates an improvement in the clinical status (Table 1).

Table 1.

Comparative Results with Eckardt Score Before and After Laparoscopic Heller Myotomy and Dor Fundoplication

n = 20	Eckardt score
n = 20	0–1	2–3	4–6	>7
Before LHM and Dor fundoplication	—	—	13	7
6 months PO	13	5	2	0
1 year PO	19	1	0	0

LHM, laparoscopic Heller myotomy; PO, postoperation.

For 1 patient under the age of 1 year, the follow-up was clinical, because it was not possible to objectify the symptoms, but only the weight loss. Preoperative tests included manometry, endoscopy, and barium swallow examination when it is possible and available.

High-resolution manometry was ordered for all patients; however, because this modality was not available at all medical centers, management was carried out based on the severity of the symptoms and imaging/endoscopic findings. Patients with Eckardt score <4 initially underwent endoscopic management with pneumatic dilatations, and those with worsened or persistent symptoms, laparoscopic management was carried out.

Surgical Procedure

Equipment and room set-up

Under general anesthesia, the patient was set in reverse Trendelenburg position, then the patient was securely strapped to the surgical table to facilitate maximum tilting and lateral rotation. The surgeon positioned himself between the patient's legs with the monitor above the patient's shoulder. The first surgical assistant positioned himself to the right of the surgeon, and the second assistant stood to the left. The scrub nurse stood to the right of the first surgical assistant.

LHM with Dor fundoplication

Using umbilical approach, the optical trocar was inserted 2 − 4 cm above the umbilicus, depending on the patient's size. Creation of pneumoperitoneum with CO₂ insufflation was performed with intraabdominal pressure of 8–12 mm Hg. A 30° laparoscope was used. The trocars were placed at the right and left flanks. Epigastric percutaneous retraction of the liver left lobe was performed using a polypropylene suture.

Dissection was minimal. Coagulation devices were used at a low energy setting to limit the risk of esophageal mucosa and branches of the pneumogastric nerve thermal injury. The surgeon incised the anterior aspect of the phrenoesophageal membrane, gaining direct access to the anterior aspect of the esophagus. The pericardium was pushed upward during the proximal esophageal dissection to the pulmonary veins level with no lateral extension to avoid pleural injury.¹³

The myotomy was performed with a cautery hook and was at least 5 cm long. Mobilization of the anterior aspect of the gastric fundus was simple and did not require division of the short gastric vessels. The right edge of the gastric fundus wrap was attached to the right edge of the abdominal esophagus with four nonabsorbable interrupted sutures. In the absence of esophageal mucosal perforation, drain was not used. Liquid diet was started the next day after the procedure, followed by a soft diet for the next 15 days.

Follow-up and statistical analysis

All patients were scheduled for follow-up at 2, 4, 6, and 12 months, and annually thereafter. Treatment success was defined as symptom relief, based on an Eckardt score (stage 0–1).

Collected data were analyzed by descriptive statistics (median and range), using a statistical software Microsoft Excel databases and version 22.0 SPSS1 (Statistical Package for the Social Sciences). Continuous variables were treated as means (range). Variables were summarized using median, minimum, and maximum values and percentages.

Results

Patients' demographics

Twenty-one patients were part of the study, 9 females and 12 males, with a mean age of 9.5 years. The median preoperative duration of symptoms was 9.5 (range 0.67–16) months. Of the total number of patients, 8 did not receive any prior therapy for treatment of their esophageal disorder, whereas 13 had prior pneumatic dilatation (Table 2).

Table 2.

Patients' Demographics Characteristics

Age, year, mean (max-min)
Age at the time of onset of symptom (range years)	9.5 (0.67–16)
Age at the time of surgery (range years)	7.06 (0.4–14)
Symptoms duration (years)	2.5 (0–5)
Gender (%)
Male	12 (57.14)
Female	9 (42.8)
Comorbidity (n)
Allgrove syndrome	5 (23%)
Previous intervention, n (%)
Botulinum toxin injection therapy	—
Pneumatic dilatation	13 (61%)
Heller myotomy	—

Among the studies performed, all the patients had an esophagram that showed a dilated esophagus with “bird's beak” sign (Fig. 1); the endoscopies typically showed esophageal dilatation with food debris and a tight cardia that allowed the passage of the endoscope in all cases.

FIG. 1.

Esophagogram. Dilatation of the esophagus and termination in a “bird's beak.”

In addition, 14 (66.6%) patients had preoperative manometry that showed aperistalsis and absence of relaxation of the lower esophageal sphincter; the remaining 7 (33.33%) patients did not have a manometric study.

LHM with Dor fundoplication outcomes

The efficacy of LHM with Dor fundoplication was 90% within 6 months after surgery and was 100% 1 year postoperatively (Table 3). LHM with Dor fundoplication was successfully completed (technical success) in 19 patients (90.4%) but 2 patients required conversion to open surgery due to technical difficulties (Table 3).

Table 3.

Symptoms Evaluation with Eckardt Score

n = 20	Before LHM and Dor fundoplication	6 months PO	1 year PO
Average Eckardt score (min -max)	6 (6–8)	1.6 (1–4)	1 (1–2)
Eckardt ≤3	—	90%	100%

LHM, laparoscopic Heller myotomy; PO, postoperation.

Seven days after surgery, 1 patient had an episode of vomit with esophageal mucosa perforation, which developed an esophagocutaneous fistula. We did not find any correlation between age and success rate or complications after surgery.

All patients underwent a barium esophagram at 12 months postoperatively. The criteria of clinical improvement were established according to the Eckardt score for patients undergoing LHM and Dor fundoplication. Ninety percent of patients had a decrease in symptoms after 6 months, and all had an Eckardt score <3 points (Table 3). One patient had persistent dysphagia. Four patients had persistent gastroesophageal reflux. Only 1 patient required esophagectomy and gastric tube reconstruction.

Adverse events

Adverse events were classified using the Clavien–Dindo classification. Grade ≤ IIIb occurred in 8 patients (4.7%): 2 patients (9.5%) experienced esophageal mucosal perforation, 1 patient (4.7%) experienced persistent dysphagia, and 5 patients (23.8%) experienced reflux, which was managed endoscopically and pharmaceutically.

There were no Clavien–Dindo classification grade ≥ IIIb adverse events. No predisposing variables were found for the occurrence of complications or therapeutic failure because both patients were of average age and their symptoms were not severe.

Discussion

Achalasia is a low prevalence condition; therefore, literature is limited, which is reflected in the absence of protocols for the pediatric population. Most of the surgical recommendations are based on adult studies, then it is important to clarify the relationship between pediatric and adult management.⁸

According to the findings of our study, the average age of onset of symptoms was 6 years with a minimum at 5 months and a maximum at 10 years; however, Hallal et al.¹⁴ present an average age at 7 years, indicating that symptoms appeared at an earlier age in our population. The average age of surgical intervention reported by Franklin et al.¹⁵ was 12 years, whereas in our study it was 8 years due to an earlier diagnosis. Allgrove syndrome was present in 31% of our patients, whereas other series reported an association with autoimmune diseases, herpes zoster infections, Chagas disease, amyloidosis, and sarcoidosis.^9,14,16,17

The diagnosis approach requires functional and anatomical studies. It is recommended that in patients older than 10–17 years old, they should undergo endoscopy, manometry, and a standard or timed barium swallow study. Recent studies have demonstrated that high-resolution manometry has a higher sensitivity than conventional manometry. Obtaining some of these studies in the pediatric population may be difficult owing to size mismatch and compliance, as we show it in our case series. There are no systematic studies defining the optimal diagnostic regimen in children; hence, the pediatric population with suspected achalasia should follow the same diagnostic pathway as adult patients. Meyer et al.¹⁸ with a total population of 42 patients showed that all patients undergo endoscopy and barium studies and 90% of the sample had manometry.

The Chicago Classification (CC) of esophageal motility disorders has not been validated in the pediatric population. In the literature, there is no scale of the pediatric population that evidence clinical prognosis, surgical procedure indication, or outcomes. For this reason, the standardization of scores that allow objectifying the symptomatic improvement of the treatment is of utmost importance, as proposed in this study with Eckardt score.

Owing to the limited resources in our population, only regular nonhigh-resolution manometries were performed without being able to classify the patients according to the CC, without change of the surgical treatment or the outcomes of the patients. All treatments for esophageal achalasia have been shown to be safe and effective in the pediatric population.

BTI has good short-term results but a high rate of recurrence in the long term, making it is unfavorable in the pediatric population. The only application of this therapy is for unfit patients with high risk for surgical intervention. In our study, all patients were fit for surgery.³

Pneumatic dilatation (PD) is an endoscopic technique recommended for patients >8 years old. This technique has described benefits such as shorter hospital stay, faster recovery time, and reduced cost.¹⁶ Likewise, it has a 53% recurrence rate for dysphagia, requiring new dilatations between 30% and 70%.⁹ The most severe complication is esophageal perforation that occurs in 2%. The patients can develop gastroesophageal reflux in 15%–35%.¹⁹

Di Nardo et al.²⁰ reported 87% success rate of PD in pediatric patients with 6 years of follow-up, with an average of three PD sessions. However, technical factors such as instruments and anatomical characteristics of patients play a fundamental role, as seen in other types of esophageal surgical procedures, such as that presented by Esposito et al.²¹ Balloon size mismatch for the younger pediatric patients limits the PD application.^18,20 However, young age should not be a contraindication for PD.

Smits et al.²² described the Netherlands experience with an 88% of symptoms recurrence in PD-treated patients that required a new treatment therapy. In LHM-treated patients, the need of a new treatment therapy was only 22%. The common aspect in all pediatric achalasia treatment series is that the treatment immediately improves patients' quality of life and reverses weight loss. Most patients will need repeat treatments over time, particularly patients having BTI or PD as an initial treatment. The long-term outcome of achalasia treatment in the pediatric population should be assessed by symptoms, function, and physical growth.

Long-term results have shown the need for multiple reinterventions, due to a high incidence of symptomatic recurrence in PD. In the Dutch longitudinal study, with a 10-year follow-up, Eckardt scores of >3 were seen in 45% of patients (no differences between PD and LHM). Gastroesophageal reflux disease symptoms were also common at long-term follow-up with incidence of 76% in LHM patients and 33% PD patients, as in our study wherein we followed up patients with imaging and clinical symptoms.²³

Surgical management with LHM has a success rate of 60%–95%.²⁴ Currently, this procedure is performed with an antireflux surgery.^24,25 In our study, all patients underwent LHM, and 2 required conversion to open surgery; no mortality occurred. The antireflux technique used was the Dor-type fundoplication. Complications occurred in 15% of the patients, and recurrence of dysphagia was reported in 20%.

Franklin et al. reported that the success rate of this procedure was 60%–95% with an estimated mortality rate of 0.1%.¹⁶ The main complications according to Stavropoulos et al. were esophageal perforation of 15% and recurrence of dysphagia of 8%.^16,19,26,27 One patient in our study required a new LHM after 1 year of follow-up, whereas Pandian et al.²⁸ reported that reoperations were performed in 48% of the cases. Complications can happen even if the operation is performed by expert laparoscopic surgeons.²⁹

LHM or POEM is the ideal treatment for pediatric patients with esophageal achalasia. LHM is often considered the first-line treatment for pediatric achalasia, accompanied by a partial fundoplication. Moreover, the surgeon, equipment, and the correct indication for laparoscopic surgery are important to reduce the incidence of complications,³⁰ although there is no conclusion regarding the superiority of a Dor or Toupet fundoplication in the pediatric population. In our patients, we performed a partial Dor fundoplication. LHM is now the most minimal invasive accepted method.

The thoracic approach has been ruled out due to poor outcomes and the inability to perform a partial fundoplication. Although the open Heller myotomy approach is safe, most centers prefer minimally invasive surgery, as in our study population.

Pastor et al.,³¹ in a retrospective study, documented a need for a new treatment therapy in 83% of PD-treated patients compared with 30% of LHM-treated patients. They also showed that open versus laparoscopic approach for Heller myotomy had no differences in short- and long-term outcomes. Lee et al.³² presented a retrospective comparison between LHM and PD. They concluded that, in the pediatric population, LHM with partial fundoplication was the best treatment for achalasia, similar to our results.

Another recent approach is POEM, which has already been shown to be feasible and safe. Data have shown that POEM is equivalent to PD or LHM in the relief of dysphagia; however, in our country, experience on POEM in the pediatric population is poor.

Nabi et al.³³ reported a total of 15 pediatric patients who underwent POEM. Only 10 patients (10/15) completed 1 year of follow-up. The mean Eckardt score before and after POEM was 7.32 ± 1.42 and 1.74 ± 0.67, respectively (P = .001). All the pediatric patients had complete resolution of their symptoms at 1 year. Although some investigators have suggested that reflux prevention may be less essential in the pediatric population. Future studies will define whether POEM could be the ultimate preferred initial strategy in the pediatric population.

However, it should be kept in mind that abnormal reflux after POEM has the potential to lead to dysplasia or adenocarcinoma in the esophagus later in life. Corda et al.³³ reported 20 patients (13 males and 7 females) who underwent LHM without antireflux procedure with a median length of follow-up of 60 months (8–114 months). None of the patients had evidence of postoperative gastroesophageal reflux. There were concerns that the size of the therapeutic endoscopic instrumentation might be too large for small infants; however, the youngest patients in case series are 5 years old, and in anecdotal reports, patients of 2 years have been treated.^34,35

The main limitation of this study is that it is an observational retrospective study without randomization and no control group. As well as lack of long-term outcomes as our data were based on the short-term results (1-year), so the long-term results should be clarified in other studies.

Further studies will be necessary to compare the use of laparoscopic approach versus POEM, to evaluate which has more benefits in the pediatric population. Also it is important to take into account that POEM is not a technology that is found throughout the world, so the laparoscopic approach assures a useful approach in the management of this entity, in addition we recommend for future prospective studies to evaluate the efficacy of the procedure with Eckardt score pre- and postprocedure.

Conclusions

LHM with Dor fundoplication according to Eckardt score pre- and postoperative evaluation results in a feasible procedure with good clinical result in the pediatric population.

Footnotes

Acknowledgments

We thank the Department of Pediatric Surgery at Hospital San Vicente Fundación, Hospital Erasmo Meoz, and Hospital de la misericordia from Colombia.

Disclosure Statement

No competing financial interests exist.

Funding Information

No funding was received for this article.

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