Surgical treatment of esophageal leiomyoma larger than 5 cm in diameter: A case report and review of the literature
Department of Thoracic Surgery, Qilu Hospital, Shandong University, Jinan, China
Case Report
Surgical treatment of esophageal leiomyoma larger than 5 cm in diameter: A case report and review of the literature
Department of Thoracic Surgery, Qilu Hospital, Shandong University, Jinan, China
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Abstract
Although leiomyoma is the most common benign esophageal neoplasm, it is a rare condition. Resection of the tumor is recommended in symptomatic patients, and observation is recommended in asymptomatic patients with small lesions. We discussed here a patient admitted to our hospital for dyspepsia in whom a calcified mediastinal neoplasm was diagnosised preoperatively and esophageal leiomyoma was diagnosised postoperatively. Enucleation of a leiomyoma of the esophagus is recommended and the optimal approaches should be tailored based on the location and size of the tumor.
Key words
Benign esophageal tumor; enucleation; esophageal leiomyoma
J Thorac Dis 2012;4(3):323-326. DOI: 10.3978/j.issn.2072-1439.2011.11.02
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Introduction
Benign tumors of the esophagus are rare and leiomyoma is the most common benign tumor of the esophagus (1). It is reported that most leiomyomas originate the inner circular muscle layer of the distal and midthoracic esophagus, particularly at the esophagogastric junction (2,3). Middle-aged men are most frequently affected (4,5). The main symptoms usually are dysphagia and epigastric pain, but they are not specific for the disease. The size of the esophageal leiomyoma may change, a size of 1 to 29 cm has been defined in the literature (1,6,7). But most of them was samller than 5 cm in diameter. Tumor that size lager than 5cm are rare. It was easily misdiagnosised as mediastinal mass, esophageal cancer (8) and esophageal stromal tumor. Its clinical feature and management are differente with other smaller esophageal leiomyoma. Here, a patient with large calcified esophageal leiomyoma who was treated in our institute is presented, initial discuss its diagnosis and management against the background of previously published cases and series.
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Case report
A 42-year-old woman was admitted to our hospital with dyspepsia and esophageal reflux. There was no nausea, vomiting, or weight loss. Results of a physical examination and standard laboratory tests were normal. A chest radiograph showed a mass in the right low mediastum, and a filling defect was apparent on esophagography. Computerized tomography (CT) scanning of the chest revealed a completely calcified 70×60 mm mass at the right lower posterior mediastinum narrowing the esophagus lumen and the cacified benign mediastinal neoplasm was diagnosis (Figure 1A and 1B). Growth over the organ borders or infiltration in neighboring structures was not detected. There were no enlarged lymph nodes, and there was no evidence for distant metastases. A right thoracotomy was performed. During surgical exploration, a unregular cacified mass in the distal esophagus (Figure 1C). The mass was enucleated completely. Histopathologic examination revealed a tumor of 70×60×50 mm and cacified spindle cell fascicles without mitosis or atypia was observed (Figure 1D).
Figure 1. (A/B): Computerized tomography (CT) scanning of the chest revealed a completely calcified 70×60 mm mass at the right lower post mediastinum; (C): A solitary esophageal leiomyoma with a ginger-like shape after enucleation; (D): Histopathologic examination revealed
cacified spindle cell fascicles without mitosis or atypia (HE×100) course was uneventful, and the patient was discharged subjectively free of
complaints on the 9th postoperative day.
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Discussion
Leiomyomas are benign tumors descending from smooth muscle cells of the esophagus. They are the most common benign tumors of the esophagus and they may occur in all parts of the esophagus, but 60% occur in the distal third, 30% in the middle, and 10% in the proximal esophagus (4). Although gastrointestinal bleeding is a common finding in gastric leiomyomas, esophageal leiomyomas rarely bleed, which may be because they do not ulcerate (9). Leiomyomas grow slowly, and half of the patients are asymptomatic and the symptoms are not specific. Dysphagia with concomitant epigastric pain or retrosternal burning usually appears when the tumor's diameter becomes larger than the critical point of 4.5-5 cm (10). It seemed that the size of tumor correlates with the severity of the symptoms, but larger leiomyoma usually grow toward outside of esophageal lumen. So dysphagia don't always proportionate to the size of tumor in larger leiomyoma. In our report, the symptoms were dyspepsia and esophageal reflux.
The diagnosis of esophageal leiomyoma is mostly not clear
preoperatively. It may present as a mediastinal mass on chest radiograph
and may be seen as an incidental radiologic finding. Differential
diagnoses include foreign oppressed desease of esophagus, malignant
esophageal tumors such as squamous or adenomatous carcinomas or
leiomyosarcoma and other benign tumors. Barium swallow is the most
commonly used radiologic test for esophageal lesions (4). The finding on
barium swallow is a smooth filling defect in esophageal lumen without a
mucosal abnormality. More esophagus was involved in large esophageal
leiomyoma, the mucosa of diseased region become thinnings, and
show hyperaemia. So it can be regarded as mucosa destroy when
barium swallow examination, that misdiagnosis as esophageal cancer
(8) or esophageal stromal tumor. Computed Tomography scans of the
chest show in most cases a mass originating from esophagus without
mediastinal lymphadenopathy, but in giant leiomyoma, tumor usually
grow toward outside of esophageal lumen. To form soft tissue shadow
in mediastinum, it can be misdiagnosised as mediastinal mass. The
diagnosis is difficault to do and may cause diagnostic confusion. So to
posterior mediastinum mass that close neighbor esophagus, it maybe a
esophageal leiomyoma, this is worthy think highly (11). Esophagoscopy
is also used for the diagnosis of esophageal leiomyoma, but it only shows
submucosal lesions and will not lead to an accurate diagnosis (4,12).
The use of the EUS can clearly reveal the structure of the esophageal
wall. On EUS, leiomyoma presents as a homogeneous and hypoechoic
lesion with clear margins, surrounded by a hyperechoic area (12), which
can easily be differentiated from a lipoma, cyst, or hemangioma in the
esophageal wall. Preoperative biopsy of the tumor is a debating issue
(13). Our policy is not to recommend it, because the tumor is easily
adhesive to the mucosa and the mucosal damage occurs accidentally
during enucleation. Moreover, in many cases biopsy could not provide
enough material to establish an accurate histopathological diagnosis.
Once the clinical diagnosis of leiomyoma is established, many factors
must be considered for the optimal treatment. Tumor size and location
are important, but also the patient’s symptoms, general condition, and
comorbidities should be taken into account. The surgical indications
of these tumors include unremitting symptoms, increased tumor
size, mucosal ulceration, histopathologic diagnosis, and facilitation of
other surgical procedures (7). Because malignant transformation in
leiomyomas is rare, some authors recommend regular follow-up with
barium swallow and endoscopy for asymptomatic patients with lesions
smaller than 5 cm and when the preoperative workup has excluded
malignancy (7,13). We suggest that a leiomyoma should be removed
when diagnosed even when asymptomatic, because there is always the
possibility, rarely though, of malignant transformation.
For leiomyoma, the location and size of the tumor are important
factors in determining the appropriate surgical approach. Endoscopic
approaches appear possible in case of small pedunculated tumors
of 2–4 cm originating from the muscularis mucosae (14). Usually
to execute endoscopic mucosal resection (EMR) or endoscopic
submucosal dissection (ESD). Symptomatic small leiomyomas <5 cm
can be enucleated either by open surgery (15,16) or by means of videoassisted
thoracoscopy (VATS) (17). Transthoracic extramucosal blunt
enucleation via a left- or right-sided thoracotomy is the most common
procedure for small- to midsized esophageal leiomyoma, which is easier,
faster, and safer compared to resection (1,7). Low tumors and tumors
of the esophago-gastric junction can be approached via upper midline
laparotomy (15). After nucleation, the muscular wall should be closed
to avoid diverticular-like mucosal bulging and for the preservation of
the muscular propulsing activity. The larger the tumor that diameter
>5 cm, associated with muscle atrophy and more muscular defects.
The muscular wall should be repaied with pedunculated pleural film,
diaphragm valve, or omentum, lung, pericardium. Postoperation feeding
time should be delayed to avoid fistula, diverticula. Strengthen the
lower esophageal mucosa, esophageal mucosa even weak are protected,
and can effectively prevent to form postoperative gastric acid reflux and
esophageal diverticulum. For giant esophageal leiomyoma should be
preferred esophageal resection and reconstruction surgery, not the same
as conventional enucleation of tumor. Because: (i) It was technically
difficult to only enucleate giant leiomyoma, and the defect of esophageal
muscle can’t ensure wound healing; (ii) esophageal muscle was
pressed by giant leiomyoma that expansive growth, became thinning
and membranous. So lower esophageal sphincter dysfunction or loss.
Iterature has reported that patients after removal of giant leiomyoma
prone to symptoms of reflux esophagitis (18); (iii) For giant esophageal
leiomyoma, it may have leiomyosarcoma-like transformation or
with small leiomyosarcoma-like lesions (19); (iv) Meanwhile, the
huge tumor endangered the physical health of patients by dysphagia.
Or may be combined esophageal cancer (20), so as soon as possible
surgery. To perform partial or subtotal resection of the esophagus,
and esophagogastric anastomosis, the results are satisfactory. In our
patient, because of the size, the location and the confusion diagnosis
of the tumor, it was extramucosal blunt enucleated using thoracotomic
approach.
IIn conclusion, diagnosis of esophageal leiomyomas requires both
endoscopic and radiologic examinations. Once the clinical diagnosis of
leiomyoma is established, the operation should be performed to remove
the tumor.
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Acknowledgments
This work is supported by Shandong Province Science and Technology
Program (No. 2007GG20002009) and Shandong Province Natural
Science Foundation (No. Y2007C038).
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References
Cite this article as: Sun X, Wang J, Yang G. Surgical treatment of
esophageal leiomyoma larger than 5 cm in diameter: A case report
and review of the literature. J Thorac Dis 2012;4(3):323-326. doi:
10.3978/j.issn.2072-1439.2011.11.02
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