Surgical treatment of esophageal leiomyoma: an analysis of our experience

Tratamiento quirúrgico de leiomioma esofágico: análisis de nuestra experiencia

P. Priego, E. Lobo, G. Rodríguez, N. Alonso, M. A. Gil Olarte, J. Pérez de Oteyza and V. Fresneda

Department of General Digestive Surgery. Hospital Ramón y Cajal. Madrid, Spain

Correspondence

ABSTRACT

Introduction: leiomyoma is the most common benign esophageal neoplasm. Surgical treatment (enucleation) has traditionally been the therapy of choice. The advent of minimally invasive techniques has produced an increase in endoscopic approaches to the detriment of open surgery.
Objective: the aim of this study was to compare the results obtained with open surgery and with laparoscopic surgery in this kind of pathology.
Material and methods: we performed a retrospective study of all leiomyomas operated for in our center between 1986 and 2004, and obtained 9 cases of esophageal leiomyoma. Four were women and five men, between the ages of 40 and 70, with a mean age of 53.5 years. The most frequent symptoms were heartburn (5 cases), dysphagia (3 cases), and retrosternal pain (3 cases). Surgery was in all the cases an enucleation. An open approach was performed in 5 cases (3 thoracotomies and 2 laparotomies), and an endoscopic approach in 4 (2 thoracoscopies and 2 laparoscopies).
Results: the mean postoperative hospital stay was 5.12 days (range 2-8 days). This was shorter for endoscopic approaches versus open surgery (3.25 vs. 7 days). There was no case of esophageal mucosal perforation or reconversion. No death, intraoperative complication, or tumor relapse was described. Only 2 patients had complications: post-surgical thoracic pain, and intestinal obstruction by adhesions 8 years after surgery.
Conclusion: enucleation is an easier procedure and constitutes the therapy of choice for esophageal leiomyoma. This approach has to be laparoscopic. We think that muscle borders should be closed after enucleation, and that biopsy is not indicated preoperatively.

Key words: Esophageal leiomyoma. Enucleation. Thoracoscopy. Laparoscopy. Thoracotomy. Benign tumor.

RESUMEN

Introducción: el leiomioma es el tumor benigno más frecuente del esófago. Tradicionalmente el tratamiento quirúrgico de estos pacientes consistía en la enucleación. El avance en las técnicas mínimamente invasivas ha provocado un aumento del abordaje endoscópico en detrimento de la cirugía abierta. ]]> Objetivo: el objetivo del trabajo fue comparar los resultados obtenidos por vía abierta y por vía laparoscópica en este tipo de patología.
Material y métodos: realizamos un estudio retrospectivo de los leiomiomas intervenidos en nuestro centro entre 1986-2004, obteniéndose 9 leiomiomas esofágicos. Cuatro eran mujeres y cinco varones, con unas edades comprendidas entre los 40-70 años, siendo la edad media de 53,5 años. Los síntomas más frecuentes eran pirosis (5 casos), disfagia (3 casos) y dolor retroesternal (3 casos). El tratamiento quirúrgico fue en todos los casos la enucleación. En cinco pacientes se realizó un abordaje abierto (3 toracotomías y 2 laparotomías) y en 4 casos se realizó una laparoscopia (2 toracoscopias y 2 laparoscopias).
Resultados: la estancia hospitalaria postoperatoria oscila entre los 2-8 días, con una media de 5,12 días, siendo esta menor en el grupo en el que se realizó cirugía laparoscópica en comparación con el grupo de cirugía abierta (3,25-7 días). No existe ningún caso de perforación de la mucosa esofágica ni de reconversión. Tampoco se describe ningún caso de mortalidad, complicación intraoperatoria ni de recidiva tumoral. Dos pacientes presentan algún tipo de complicaciones: dolor torácico post cirugía, obstrucción intestinal por bridas 8 años después de la cirugía.
Conclusión: la enucleación es un procedimiento fácilmente realizable y constituye el tratamiento de elección del leiomioma esofágico. Actualmente el abordaje debe ser laparoscópico. Somos partidarios del cierre de la muscular y bajo nuestro punto de vista la biopsia preoperatoria no está indicada.

Palabras clave: Leiomioma esofágico. Enucleacion. Toracoscopia. Laparoscopia. Toracotomía. Tumor benigno.

Introduction

Leiomyoma is the most common benign esophageal neoplasm. Incidence is variable, and in autopsy series ranges from 0.005 to 5.1%. Leiomyoma is relatively rare when compared with esophageal carcinoma, which occurs 50 times more frequently.

Morgagni first described leiomyoma in 1761, but Munro, in 1797, was the first to report a localized intramural leiomyoma of the esophagus.

Leiomyoma has traditionally been classified within the term of gastrointestinal stromal tumor (GIST); however, recent advances in immunohistochemical and molecular biology have suggested that these two conditions are different.

Sauerbruch reported in 1932 the first successful surgical treatment of leiomyoma with esophageal resection, and one year later Oshawa performed the first successful surgical enucleation of this type of tumor.

Surgical treatment has been performed by laparotomy or thoracotomy. However, the advent of minimally invasive techniques has produced an increase in endoscopic approaches to the detriment of open surgery.

The aim of this study is a retrospective analysis of hospital experience with this kind of pathology, and compare the results obtained for open an laparoscopic surgery.

Material and methods (Table I)

Between 1986 and 2004 we treated 9 cases of esophageal leiomyoma at the "Hospital Ramón y Cajal'. Four were women and five were men, between the ages of 40 and 70 years, with a mean age of 53.5 years.

Five of these tumors were found in the distal third of the esophagus (56%), three in the middle third (33%), and one in the proximal third (11%). Size of the tumors was 1-8 cm, with a mean size of 3.4 cm. There was no case of multiple leiomyoma, and all had an intramural localization.

The definitive anatomopathological diagnosis was in all cases esophageal leiomyoma except for one case of esophageal duplication cyst. We observed the typical aspect of leiomyomas with their firm consistency, white color, well encapsulated nature, and smooth or nodular surface. Histologically we saw smooth-muscle cells with hypovascularity and absent mitoses.

The most common symptoms included heartburn in 5 cases (56%), dysphagia in 3 (33%), and epigastric-retrosternal pain in 3 cases (33%). Two patients had symptoms of upper GI blood (melena and duodenal ulcus); one reported regurgitation, and one rectorrhagia. There were only two asymptomatic patients (duodenal ulcus and rectorrhagia), which represents approximately 22.2%, and one case was intraoperatively diagnosed during antireflux surgery.

A biopsy was performed in 3 patients before surgery, and no tumor was found in any of them. In two cases it was performed by endoscopic ultrasonography, and in one case by CT.

A hiatal hernia was found in four patients (44.4%).

We performed 5 open surgeries (3 thoracotomies and 2 laparotomies), and 4 laparoscopic surgeries (2 thoracoscopic and 2 laparoscopic). If we analyze results after 2000, we see a greater use of minimal invasive surgery, with 4 endoscopic approaches and one thoracotomy.

For thoracic approaches intubation was performed using a double-lumen endobronchial tube; the patient was placed in a left lateral decubitus position, and we usually entered the pleural cavity with 4 trocars.

For the abdominal laparoscopic approach we applied the same technical principles as for esophageal hiatal surgery.

In two cases -thoracoscopic approaches- we performed an intraoperative endoscopy to check tumor localization.

In all cases the esophageal mucosa was complete after checking in one case with methylene blue and in one case with an air injection through a nasogastric tube.

We sutured muscular defects in all cases.

In 4 cases we associated an antireflux technique (Dor's in 3 cases and Nissen's in one case). In contrast, we performed a simultaneous cholecistectomy in one case.

Results

The mean operating time was 182.14 minutes (70-270 min). For open surgery it was 146.25 minutes (70-205 min), and 230 minutes for laparoscopic surgery (165-270 min).

The mean postoperative hospital stay was 5.12 days (range 2-8 days). When comparing postoperative hospital stay between the laparoscopic approach and open surgery, a reduced stay (3.25 vs. 7 days) was seen in the group with laparoscopic approach.

There was no case of mortality or intraoperative complications. There was no case of conversion to open surgery either.

There was no case of recurrent leiomyoma.

Regarding postoperative complications: we have a patient with severe pain in the right hemithorax (operated by thoracoscopy) who requires periodic control at the Pain Unit, and another case of intestinal obstruction by adhesions eight years after surgery (laparotomy).

Two patients were re-operated on for reflux symptoms (both had previous surgery), and underwent a Nissen's procedure. Case number 5 was operated on 11 months after first surgery, and case number 9 at 8 months. In both cases the approach was laparoscopy.

Discussion

Leiomyoma is a smooth muscle cell tumor that develops predominantly intramurally and in the lower third of the esophagus (56%) (1-3). Our results confirm these affirmations, because all of them were intramural, most were in the lower third (56%) and middle third (33%), and mean size was smaller than 5 cm (approximately 3.4 cm).

Clinically, they are slow-growth tumors, most of them asymptomatic (15-50%), and their diagnosis is followed during a long period. Interestingly there appears to be no direct correlation between tumor size and symptoms (1-3). In our series, not surprisingly most patients had gastroesophageal reflux symptoms (56%), as these tumors are associated with esophageal disorders (hiatal hernia, gastroesophageal reflux, achalasia, epiphrenic diverticulum), and primarily with hiatal hernia in 4.5-23% of cases. We found an associated hiatal hernia in a 44% of cases. Other symptoms are dysphagia (33%) and retrosternal pain (33%), but no case of weight loss was seen.

The definitive diagnosis of these tumors can be made only by histologic examination. Biopsy before surgery, from our standpoint, would be contraindicated (3,4). This procedure involves risks such as secondary infection, bleeding, and esophageal mucosal perforation. In our series three biopsies were obtained (33%), and none of them was important for the diagnosis, since histology showed a normal esophageal mucosa.

One study found that mucosal tears occurred in 50% of patients undergoing a preoperative biopsy (1,3,5), and in only 8% of those who did not.

Multiple studies are available to diagnose this kind of tumors (1,3); however, the two studies to be performed in all patients are, in our view, barium swallow and oral endoscopy. Thoracoabdominal CT and endoscopic ultrasonography are also interesting, as they may depict the anatomical relations of the tumor, and differentiate between intramural and extrinsec lesions.

The treatment of these tumors is controversial. There is a general consensus in the literature that esophageal leiomyoma should be surgically removed in symptomatic patients. The main problem arises when the diagnosis is made in an asymptomatic patient, which occurred in 50% of our cases.

Overall we agree with most authors on the surgical indications of these tumors (1,5): unremitting symptoms, increased tumor size, mucosal ulceration, histopathologic diagnosis, and facilitation of other surgical procedures.

Glaz and Grunebaum (6) recommended that asymptomatic and mildly symptomatic patients should not undergo surgery but be regularly monitored using radiography and endoscopy every 1-2 years instead.

The rationale for non-surgical therapy is: the slow growth of these tumors, rare malignant transformation, and the trauma of surgical treatment may be more harmful to the patient than no treatment in any case (1).

The surgical approach has classically been thoracotomy. With this procedure we obtained a highly successful outcome and a low complications rate; however, we think that with the advent of laparoscopic techniques, the current surgical approach should be laparoscopic, as we obtained: a) short postoperative hospital stay (in our series, 3.25 vs. 7 days); b) decreased postoperative pain (however, in our experience, one patient with postoperative complications had undergone a thoracoscopic approach and has trocar-induced intercostal neuralgia, requiring treatment by the Pain Unit); and c) a rapid and full re-expansion of the lung (4,8). Other authors (1,8) recommended that the thoracoscopic approach may best be suited for use in patients with lesions smaller than 5 cm, and when the preoperative workup has excluded malignancy.

Bonavina considers that the thoracoscopic approach would not be indicated for distal tumors if we are going to associate an antireflux technique. In these cases the abdominal laparoscopic approach would be indicated.

There are other forms of minimally invasive treatment, as the combination of endoscopy with balloon dilator assistance. When the balloon is intraluminally inflated, it promoted the expulsion of the tumor from the esophageal wall, thus facilitating thoracoscopic resection; however, there are few cases described in the literature with unreported complications and mortality (11,12).

Another technique performed in Eastern countries is an endoscopic approach alone. This is generally indicated for submucosal tumors. After separating the tumor from the submucosal layer by injection of a hypertonic solution containing 10% glycerol, 5% fructose, and saline, the tumor is suctioned into a cylinder (13-17). However, we have no experience with both treatments.

Esophageal resection would be only indicated for high tumors (> 8 cm), annular morphology, multiple leiomyomatosis, or when accidental extensive damage to the esophageal wall appears to pose a risk for postoperative leakage. We have no case of esophageal resection in our series (1), although we are experienced thereupon.

We used endoscopy intraoperatively in two cases to facilitate tumor localization.

We had no case of mucosal perforation. We do not systematically prove mucosal integrity, but for one case we used methylene blue and for another case insuflated air via a nasogastric tube.

There is some disagreement in the literature about whether myotomy should be sutured after enucleation. Hennessy and Cushieri (18) state that a wide breach can be left open without concern, but most experts (1,3,4,8) emphasize the need to reapproximate the muscular wall to prevent mucosal bulging. We used vicryl or ethibond to suture myotomy. It is thought that an approximation of muscle borders after enucleation may preserve the esophageal propulsive activity, thus preventing such complications and improving long-term surgical treatment outcomes.

Several studies have reported the occurrence of postoperative dysphagia due to pseudodiverticular mucosal bulging at the enucleation site in patients in whom muscle edges were not approximated (3,8).

One criticism may be that two patients had to be operated on a few months after surgery for persistent symptoms of gastroesophageal reflux that had started before enucleation, as we could have associated an antirreflux technique during the first surgery. Probably, the association of an antirreflux technique could be indicated in patients undergoing enucleation to protect the muscular myotomy borders and to treat gastroesophageal reflux, but this point remains highly controversial (3).

Conclusion

-Enucleation is an easy procedure and the treatment of choice for leiomyioma.

-The approach should be currently laparoscopic.

-We think that muscular borders should be closed, and that biopsy is not indicated before surgery.

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Correspondence: ]]> Pablo Priego Jiménez.
C/ Fermín Caballero, 26, 1ºA.
16004 Cuenca.
e-mail: papriego@hotmail.com

Recibido: 29-06-05.
Aceptado: 24-11-05.