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J Thorac Cardiovasc Surg 2007;133:1665-1667
© 2007 The American Association for Thoracic Surgery
Brief Communication |
Department Cardiothoracic Surgery, Weill Medical College of Cornell University, New York, NY.
Received for publication January 16, 2007; accepted for publication February 12, 2007. * Address for reprints: Anthony W. Kim, MD, Rush University Medical Center, 1725 W Harrison St, Suite 774, Chicago, IL 60612. (Email: Anthony_kim{at}rush.edu).
Giant cavernous hemangiomas are rare tumors of the esophagus. In 1976, Feist, Talley, and Hunt1
described an upper esophageal hemangioma as "giant"; it was preoperatively 4 x 7 cm and pathologically 12.0 x 5.0 x 2.5 cm in size. We present the case of an asymptomatic 68-year-old woman who had a giant hemangioma of the distal esophagus. She underwent an esophagectomy with a gastric pull-up via a left thoracotomy. Her recovery was unremarkable and she is doing well at 4 months’ follow-up.
A 68-year-old asymptomatic woman was found to have an abnormality on a chest radiograph before cataract surgery. A follow-up computed tomographic (CT) scan demonstrated a large lobulated distal esophageal mass with multiple small calcifications (Figure 1). Endoscopy was negative for mucosal lesion. A positron emission tomographic (PET) scan revealed that the mass had a standardized uptake value of 2.0. A transthoracic biopsy of the lesion demonstrated only blood and fibrinated material. Magnetic resonance imaging (MRI) reidentified the lobulated soft tissue mass in the distal esophagus and described it as a fusiform lesion involving the esophageal wall, causing narrowing of the lumen. Neither mediastinal invasion nor lymphadenopathy was noted. On the basis of the soft tissue signal and the smooth appearance, the lesion was thought to be a large leiomyoma.
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Generally, patients with cavernous hemangiomas of the esophagus have presented with hematemesis that ranges from self-limiting to life-threatening or with varying degrees of dysphagia.1-5 An asymptomatic presentation, however, is not uncommon.3,4 Endoscopically, hemangiomas usually are described as submucosal lesions that appear anywhere along the esophagus, are bluish, compressible, and protruding.3,4 They can also have atypical appearances including a reddish discoloration, a normal-appearing mucosa, or an ulcerated appearance.1 Barium esophagogram may initially identify a filling or luminal defect, but CT scans may better delineate the lesion itself. The intact mucosa often associated with this submucosal lesion may be missed on endoscopy even in the presence of a large intraesophageal tumor.1 On CT scans, these tumors appear homogeneous with small calcifications or phleboliths.4 Although endoscopic ultrasound was not performed in this patient, early reports have detailed the usefulness of this procedure in further characterizing this lesion. Sonographically, hemangiomas range from anechoic to hypoechoic to isoechoic and involve the mucosal and submucosal layers with an intact muscularis propria.4,5 Endoscopic ultrasound can also confirm the absence of continuity with major blood vessels.4-5
In the patient presented in this report, magnetic resonance imaging and angiography (MRI/MRA) were used preoperatively to help distinguish this lesion. Characteristic MRI findings have included an isointense signal from the mass on T1-weighted images and a high signal on T2-weighted images.3,4 Other reports have described atypical features such as a high-intensity signal from the mass on both T1- and T2-weighted images.4 In this specific patient, the T1-weighted images demonstrated a predominantly soft tissue type signal. The T2-weighted images demonstrated the soft tissue signal type with areas of small hyperintense signals and small areas of hypointense signals. MRA demonstrated the absence of any dominant vessels extending into or toward the mass.
Despite recent advances, arriving at the definitive diagnosis of a hemangioma preoperatively can be elusive when using noninvasive modalities. Endoscopic biopsy of the intraesophageal tumor is commonly used to obtain a preoperative histologic diagnosis. Often a concern of hemorrhage is the reason cited for not performing an endoscopic biopsy, but in reality this is a rare occurrence.5 In lieu of the more frequently described endoscopic biopsy, a transthoracic biopsy was performed in the patient in this current report. Unfortunately, the tissue obtained in this case yielded an inconclusive result. A review of the current literature does not describe the routine use of transthoracic needle biopsy in the diagnosis of this type of tumor.
When preoperative studies are inconclusive, surgical resection is often indicated both for diagnosis and to rule out malignant disease.5 In this case report, the tumor was noted to be large on preoperative imaging studies and locally invasive on initial exploration. These features suggested a malignant process and therefore a resection was indicated. Additionally, the potential risk of bleeding from the vascular tumor, if left unresected, warranted resection.3,5
The modern literature suggests that endoscopic resection is adequate in the treatment of this tumor. Submucosal esophageal cavernous hemangiomas have been successfully resected by the endoscopic polypectomy or mucosal resection techniques.4,5 Alternatively, other ablative techniques, such as laser therapy, also have been described in the treatment of cavernous hemangiomas.3 These modalities, however, are used in the treatment of lesions that are smaller than the one described in the present patient.
Before modern endoscopic instrumentation, limited local resection in the form of a polypectomy or enucleation was the accepted or even favored method of treatment. Approaches to a limited resection have classically been through a thoracotomy.2 Recently, thoracoscopic removal has also been described in the treatment of these lesions with the simultaneous use of a flexible endoscope.2 This obviously is possible only if the lesion is small enough for this type of resection. Esophagectomy has rarely been described in the treatment of this lesion, but this is because, in general, the majority of cavernous hemangiomas of the esophagus reported are on the order of 2 to 3 cm.3 The hemangioma described in this report was giant, and concerns of malignancy and potential hemorrhage warranted an esophagectomy.
References
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