ATS
HOME HELP FEEDBACK SUBSCRIPTIONS ARCHIVE SEARCH TABLE OF CONTENTS
 QUICK SEARCH:   [advanced]


     


This Article
Right arrow Abstract Freely available
Right arrow Full Text (PDF)
Right arrow Alert me when this article is cited
Right arrow Alert me if a correction is posted
Services
Right arrow Email this article to a friend
Right arrow Similar articles in this journal
Right arrow Similar articles in PubMed
Right arrow Alert me to new issues of the journal
Right arrow Add to Personal Folders
Right arrow Download to citation manager
Right arrow Author home page(s):
Dean M. Donahue
John C. Wain
Cameron D. Wright
Henning A. Gaissert
Hermes C. Grillo
Douglas J. Mathisen
James S. Allan
Right arrow Permission Requests
Citing Articles
Right arrow Citing Articles via HighWire
Right arrow Citing Articles via Google Scholar
Google Scholar
Right arrow Articles by Mutrie, C. J.
Right arrow Articles by Allan, J. S.
Right arrow Search for Related Content
PubMed
Right arrow PubMed Citation
Right arrow Articles by Mutrie, C. J.
Right arrow Articles by Allan, J. S.
Related Collections
Right arrow Esophagus - other

Ann Thorac Surg 2005;79:1122-1125
© 2005 The Society of Thoracic Surgeons


Original articles: General thoracic

Esophageal Leiomyoma: A 40-Year Experience

Christopher J. Mutrie, MD, Dean M. Donahue, MD, John C. Wain, MD, Cameron D. Wright, MD, Henning A. Gaissert, MD, Hermes C. Grillo, MD, Douglas J. Mathisen, MD, James S. Allan, MD*

Division of Thoracic Surgery, Department of Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts

Accepted for publication August 13, 2004.

* Address reprint requests to Dr Allan, Massachusetts General Hospital, Blake 1570, 55 Fruit St, Boston, MA02114 (E-mail: jallan{at}partners.org).


    Abstract
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: Esophageal leiomyomas, although infrequent, are the most common benign intramural tumors of the esophagus. They represent 10% of all gastrointestinal leiomyomas and frequently cause symptoms, necessitating resection.

METHODS: The Massachusetts General Hospital Pathologic Database was reviewed over a 40-year period for patients who underwent surgical resection of esophageal leiomyomas. Data analyzed included demographic information, presenting symptoms, tumor location, tumor characteristics and histology, diagnostic procedures, and treatment modalities/outcomes. Fifty-three patients were identified; 31 patients were symptomatic from their leiomyomas.

RESULTS: Symptomatic patients presented at a mean age of 44 years old and exhibited a twofold male predominance. Mean tumor diameter among symptomatic patients was 5.3 cm, as compared to 1.5 cm in asymptomatic patients (p < 0.0001). Thirty of the symptomatic patients had solitary leiomyomas, and 1 patient had five separate leiomyomas. Eighty-four percent of the lesions in symptomatic patients occurred in the lower two-thirds of the esophagus, with epigastric discomfort being the most common presenting symptom. Among patients operated on solely for leiomyoma, 97% were enucleated without an esophageal resection. None of the leiomyomas showed malignant transformation or recurrence. All symptomatic patients had relief of symptoms, with no perioperative morbidity or mortality.

CONCLUSIONS: In a large pathologic series, over half of all patients with esophageal leiomyomas were symptomatic. Larger tumors were significantly more likely to be symptomatic. Local enucleation by a variety of surgical approaches was accomplished in most patients. All symptomatic patients had relief of symptoms, with no perioperative morbidity or mortality. There was no observed tendency for malignant transformation or recurrence.


    Introduction
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Esophageal leiomyomas are the most common benign intramural tumors of the esophagus. Still, these tumors are relatively rare, with autopsy series data showing an incidence of 0.006 to 0.1% [1]. By comparison, esophageal carcinoma is approximately 50 times more common than leiomyoma [2]. Esophageal leiomyomas are typically found in patients between 20 and 50 years old, and represent 10% of all gastrointestinal leiomyomas. They are multiple in 3% to 10% of patients and are found more often in men by a ratio of 2:1 [3–5]. Eighty percent occur in the middle to lower third of the esophagus [6]. Historically, dysphagia and odynophagia are the most common complaints of those patients who are symptomatic [7].

Leiomyomas are most commonly diagnosed on contrast esophagography, where they appear as a smooth concave filling defect within the esophageal lumen [1]. They can also be seen on chest radiography, computed tomography, and magnetic resonance imaging.

Pathologically, leiomyomas consist of interlacing bundles of smooth muscle cells and occasionally can grow as large as 20 cm in diameter. Generally, tumors less than 5 cm in diameter are asymptomatic [1]. In a comprehensive review of more than 800 cases reported in the world literature, only two (0.2%) were documented to show malignant transformation from leiomyoma to leiomyosarcoma [2].


    Patients and Methods
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
A search of the Massachusetts General Hospital Pathologic Database was performed to identify all cases of esophageal leiomyoma that were surgically resected between January 1962 and January 2002. Office records, hospital records, or both from all identified patients were also reviewed. The data extracted from the medical records included demographic information, the presenting symptoms, the location of the tumor in the esophagus, the size and shape of the tumor, the diagnostic procedure, the treatment modality, the incidence of recurrence, surgical complications and deaths. Information was cataloged and analyzed.

Fifty-three patients were identified. Of these, 31 patients (58%) underwent surgery solely for the resection of symptomatic leiomyomas. One patient (2%) underwent resection of an asymptomatic leiomyoma. The remaining 21 patients (41%) had thoracic procedures for other indications (eg, lung cancer, esophageal cancer, hiatal hernias, etc.), and resection of an incidentally discovered leiomyoma was concurrently performed. Follow-up data that adequately described the patient's health status was available on 27 of 53 patients in this series, with a mean follow-up interval of 6.3 years.


    Results
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Gender
Sixty-four percent (n = 34) of all patients studied were male; 36% (n = 19) were female, yielding a male predominance of 1.8:1. Among symptomatic patients operated on solely for leiomyoma (n = 31), 66% (n = 21) were male, and 34% (n = 10) were female, yielding a male predominance of 2.1:1.

Age
The mean age of all patients studied was 51.7 years old (range, 19 to 81 years old). Among symptomatic patients operated on solely for leiomyoma (n = 31), the mean age was 43.8 years old (range, 19 to 76 years old).

Size
The mean tumor diameter of all patients studied was 3.7 cm (range, 0.2 to 17.0 cm). Among symptomatic patients operated on solely for leiomyoma (n = 31), the mean tumor diameter was 5.3 cm (range, 0.8 to 17.0 cm). Asymptomatic patients had a significantly lower mean tumor diameter (1.5 cm [range, 0.2 to 6.0 cm]; p < 0.00001, Student's t test).

Location
Among all patients studied, the majority of the tumors arose in the lower (53%) and middle (43%) thirds of the esophagus (Fig 1). Only 4% were in the upper third of the esophagus. Among symptomatic patients operated on solely for leiomyoma (n = 31), the tumors were distributed as follows: lower third (39%), middle third (55%), and upper third (6%).



View larger version (46K):
[in this window]
[in a new window]
 
Fig 1. Anatomical location of leiomyoma. {blacksquare} = upper third; {cjs2113} = middle third; {cjs2110} = lower third.

 
Tumor Characteristics
Among all patients studied, 48 patients (91%) had solitary tumors and 5 (9%) had multiple tumors. Three leiomyomas (6%) were calcified. Fourteen (26%) patients had lobulated masses. Three other patients (6%) had serpigenous leiomyomas. One leiomyoma (2%) was horseshoe-shaped (Fig 2). Among symptomatic patients operated on solely for leiomyoma (n = 31), 30 patients (97%) had solitary tumors and 1 patient (3%) had multiple tumors. Three leiomyomas (9%) were calcified. Thirteen patients (41%) had lobulated masses. Three other patients (9%) had serpigenous leiomyomas. One leiomyoma (3%) was horseshoe-shaped. None of the lesions showed any foci of leiomyosarcoma, suggestive of malignant degeneration.



View larger version (101K):
[in this window]
[in a new window]
 
Fig 2. Horseshoe leiomyoma.

 
Symptoms and Associated Disorders
Among symptomatic patients operated on solely for leiomyoma (n = 31), 21 patients (68%) reported epigastric distress (including heartburn and other epigastric symptoms commonly attributed to gastroesophageal reflux). Sixteen patients (52%) reported dysphagia. Other symptoms were reported less frequently (see Table 1).


View this table:
[in this window]
[in a new window]
 
Table 1. Presenting Complaint(s) Among Symptomatic Patients (n = 31)
 
Diagnostic Evaluation
All symptomatic patients underwent barium esophagography. The majority of patients treated within the last decade also received chest computed tomographic scan. Esophageal ultrasounds and endoscopic biopsies were used occasionally by referring physicians, although the ultimate management of those patients so examined was not significantly impacted. We do not routinely perform endoscopic biopsies ourselves unless there is some diagnostic uncertainty that would make resection unwarranted. There is also some anecdotal experience suggesting that a preoperative biopsy (and consequent mucosal adhesions) may impede easy enucleation of the tumor.

Surgical Approach and Outcomes
Among symptomatic patients operated on solely for leiomyoma (n = 31), 21 patients (66%) were approached through a right thoracotomy (limited or posterolateral). The remaining 10 patients (34%) were approached in accordance with Table 2. In all patients except one, the leiomyoma was enucleated by dissection within the muscular wall of the esophagus. In all patient the muscular layer was reapproximated with interrupted silk sutures. One patient sustained a full-thickness injury to the esophageal wall during enucleation, which was directly repaired and buttressed with a pedicled intercostal muscle flap at the time of surgery without sequelae. One patient required an esophagogastrectomy for removal of a giant (> 10 cm) leiomyoma. All symptomatic patients had relief of symptoms. There were no postoperative complications or perioperative deaths in this group. No patients represented to our care with recurrence.


View this table:
[in this window]
[in a new window]
 
Table 2. Surgical Approach in Patients Operated on Solely for Leiomyoma (n = 32)
 

    Comment
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
We report here a large single-institution case series of esophageal leiomyoma. Our data first point to the rarity of this entity. Despite being a tertiary referral center for surgical esophageal disease, our hospital performed less than one operation per year solely for the treatment of a symptomatic esophageal leiomyoma. These data are in concordance with reported autopsy series suggesting an overall incidence of 0.006% to 0.1% for esophageal leiomyoma [1]. Our data also show that leiomyoma have a twofold male sex predominance. This finding is in agreement with the 1.9:1 ratio found in a compilation of 838 patients from the world literature, reported by Seremetis and colleagues in 1971 [5]. Our data also show that leiomyoma are preferentially found in the lower two-thirds of the esophagus, which is consistent with the normal anatomical distribution of smooth muscle within the esophageal wall. These benign tumors tend to present as solitary round masses without calcification.

A slight majority of our patients were symptomatic and presented with complaints of epigastric distress, dysphagia, or both. Seremetis and coworkers [5] reported similar symptomology in their review, noting that dysphagia and pyrosis (heartburn) were the most common presenting symptoms. However, Seremetis noted in his own series of 19 patients that only those with concomitant hiatal hernias presented with pyrosis. Our data also show the intuitive finding that large leiomyomas are significantly more likely to be symptomatic. The mean diameter of tumors in symptomatic patients was 5.3 cm, as opposed to 1.5 cm for patients with incidentally found leiomyomas (p < 0.001; Student's t test).

The majority of leiomyomas in this series were resected by open thoracotomy by enucleating the lesion from within the muscular wall of the esophagus, without entering the mucosal lumen. It has been our practice to routinely reapproximate the muscular layer of the esophagus with interrupted silk suture following enucleation, which may be significant in the prevention of postoperative complications such as diverticula. We have found this approach to be both safe and efficacious, and report no morbidity or mortality using these techniques. In particular, we have not observed an increased incidence of heartburn or epigastric pain following resection. This is most likely due to our efforts to minimize hiatal dissection and mobilization during the operation.

In this series, we are reporting only 1 patient whose leiomyoma was removed by a video thoracoscopic approach. We recognize that thoracoscopic removal of these benign tumors is currently an excellent approach to the management of most simple leiomyomas. The low frequency of minimally invasive resections in this series is due both to its historical nature and due to individual surgeon preferences. Bardini and colleagues [8] first described video thoracoscopic enucleation of esophageal leiomyomas in 1992, and several investigators have reported favorable results using combined thoracoscopic and endoscopic approaches [9, 10]. We believe that minimally invasive procedures are certainly acceptable, particularly for smaller rounded solitary lesions, where the submucosal plane can be easily identified and developed.

We have described a pathologic series of esophageal leiomyomas in which slightly over half of the patients were symptomatic. Larger tumors were significantly more likely to be symptomatic. Local enucleation by a variety of surgical approaches was accomplished in the vast majority of patients. One patient with a giant leiomyoma required esophagogastrectomy. None of the leiomyomas showed malignant degeneration or recurred. All symptomatic patients had relief of symptoms, with no perioperative morbidity or mortality.


    References
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Wright CD, Gaissert HA, Puma F, Mathisen DJ. The oesophagus: benign and malignant tumoursIn: Morris PJ, Malt RA, editors. Oxford textbook of surgery. Oxford: Oxford University Press; 1994. pp. 893-904.
  2. Arnorsson T, Aberg C, Aberg T. Benign tumours of the oesophagus and oesophageal cysts Scand J Thorac Surg 1984;18:145-150.
  3. Kabuto T, Taniguchi K, Iwanaga T, Terasawa T, Tateishi R, Taniguchi H. Diffuse leiomyomatosis of the esophagus Dig Dis Sci 1980;25:388-391.[Medline]
  4. Postlethwait RW. Surgery of the esophagusNorwalk, CT: Appleton-Century-Crofts; 1986345–8.
  5. Seremetis MG, Lyons WS, deGuzman VC, Peabody Jr JW. Leiomyomata of the esophagus Cancer 1976;38:2166-2177.[Medline]
  6. Schmidt HW, Clagett T, Harrison EG. Benign tumors and cysts of the esophagus J Thorac Cardiovasc Surg 1961;41:717-732.
  7. Zwischenberger JB, Alpard SK, Orringer MB. Tumors of the esophagusIn: Sabiston DC, editor. Textbook of surgery: the biological basis of modern surgical practice. Philadelphia: W.B. Saunders Company; 1991. pp. 689-700.
  8. Bardini R, Segalin A, Ruol A, Pavanello M, Peracchia A. Videothoracoscopic enucleation of esophageal leiomyoma Ann Thorac Surg 1992;54:576-577.[Abstract]
  9. Bonavina L, Segalin A, Rosati R, Pavanello M, Peracchia A. Surgical therapy of esophageal leiomyoma J Am Coll Surg 1995;181:257-262.[Medline]
  10. Izumi Y, Inoue H, Endo M. Combined endoluminal-intracavitary thoracoscopic enucleation of leiomyoma of the esophagus: a new method Surg Endosc 1996;10:457-458.[Medline]



This article has been cited by other articles:


Home page
GutHome page
A Seicean and R Stan
Dysphagia in a 27-year-old man
Gut, October 1, 2009; 58(10): 1353 - 1353.
[Full Text] [PDF]


Home page
J. Thorac. Cardiovasc. Surg.Home page
M. Kent, T. d'Amato, C. Nordman, M. Schuchert, R. Landreneau, M. Alvelo-Rivera, and J. Luketich
Minimally invasive resection of benign esophageal tumors
J. Thorac. Cardiovasc. Surg., July 1, 2007; 134(1): 176 - 181.
[Abstract] [Full Text] [PDF]


This Article
Right arrow Abstract Freely available
Right arrow Full Text (PDF)
Right arrow Alert me when this article is cited
Right arrow Alert me if a correction is posted
Services
Right arrow Email this article to a friend
Right arrow Similar articles in this journal
Right arrow Similar articles in PubMed
Right arrow Alert me to new issues of the journal
Right arrow Add to Personal Folders
Right arrow Download to citation manager
Right arrow Author home page(s):
Dean M. Donahue
John C. Wain
Cameron D. Wright
Henning A. Gaissert
Hermes C. Grillo
Douglas J. Mathisen
James S. Allan
Right arrow Permission Requests
Citing Articles
Right arrow Citing Articles via HighWire
Right arrow Citing Articles via Google Scholar
Google Scholar
Right arrow Articles by Mutrie, C. J.
Right arrow Articles by Allan, J. S.
Right arrow Search for Related Content
PubMed
Right arrow PubMed Citation
Right arrow Articles by Mutrie, C. J.
Right arrow Articles by Allan, J. S.
Related Collections
Right arrow Esophagus - other


HOME HELP FEEDBACK SUBSCRIPTIONS ARCHIVE SEARCH TABLE OF CONTENTS
ANN THORAC SURG ASIAN CARDIOVASC THORAC ANN EUR J CARDIOTHORAC SURG
J THORAC CARDIOVASC SURG ICVTS ALL CTSNet JOURNALS