Ann Thorac Surg 1996;61:726-728
© 1996 The Society of Thoracic Surgeons
Case Report
Hemangioma of the Esophagus
Frederick H. Taylor, MD,
Fred C. Fowler, MD,
William L. Betsill, Jr, MD,
Marie-Claire Marroum, MD
Departments of Thoracic Surgery, Gastroenterology, and Pathology, University Memorial Hospital, Charlotte, North Carolina
Accepted for publication August 14, 1995.
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Abstract
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Hemangioma of the esophagus is a rare tumor. Fewer than 100 cases have been found in the literature. The present case report describes a patient who experienced a sudden massive hematemesis and demonstrated unusual changes on the computed tomographic scan.
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Introduction
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Esophageal vascular tumors, albeit rare, can result in life-threatening bleeding or obstruction. To date, fewer than 100 cases have been found in the literature, mostly single case reports. This case, a cavernous hemangioma, presented dramatically, necessitating rapid clinical intervention.
A 46-year-old, healthy businessman suffered massive hematemesis and loss of consciousness. On emergency department arrival, initial blood pressure was 90/60 mm Hg. The hemoglobin level, initially 11 g/dL, quickly fell to 9 g/dL within 30 minutes. Emergency esophagoscopy demonstrated a 2-cm rent in the upper esophagus. The lesion was cauterized successfully, arresting the hemorrhage. Plain film of the chest revealed a mass in the upper right mediastinum. A smooth filling defect was noted on barium swallow (Fig 1
), suggestive of leiomyoma. Computed tomography, however, revealed circular septae associated with the mass, suggestive of a vascular lesion (Fig 2).
After 2 days of a full liquid diet and 2 units of blood, the patient underwent general anesthesia and esophagoscopy. A smooth, submucosal mass was noted at 18 cm. No ulceration was seen, and a right thoracotomy was performed through the fourth intercostal space. A 6-cm, smooth, esophageal intramural mass was localized, 1 cm superior to the azygus vein. During enucleation, the mucosa was entered. The mucosa was closed with interrupted silk and reinforced with a pedicle of pleura due to the absence of available muscularis for closure.
The gross pathology revealed a 7.0 x 6.0 x 6.0-cm apparently encapsulated, ovoid, multinodular, soft, congested tan lesion. The cut surface contained trabecular septae separating vascular spaces ranging from 0.5 to 1.0 cm. Areas of necrosis and thrombosis were present (Fig 3).
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Fig 3. . Gross photograph of 7.0 x 6.0 x 6.0-cm encapsulated multinodular lesion. Cut surface reveals cystic vascular spaces.
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Microscopically the lesion manifested a proliferation of benign vascular spaces of cavernous nature, although capillary-sized channels were present. Vascular proliferation was present at the upper and lower mediastinal margins (Fig 4). There was no evidence of malignancy.
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Fig 4. . Microscopic view of tumor shows proliferation of benign venous structures. (Hematoxylin and eosin; x100 before 54% reduction.)
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The patient's postoperative course was uneventful, and 4 weeks later he was well.
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Comment
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Vascular lesions arising in the esophagus are rare. Gentry and associates [1] reviewed 261 vascular tumors of the gastrointestinal tract; only 11 were associated with the esophagus. Plachta [2] in 1962 reported 99 cases of benign esophageal neoplasms; 3 were hemangiomas. Govoni [3] cataloged 56 esophageal hemangiomas from the literature, mostly single case reports or studies of 2 or 3 cases. Hemorrhage and dysphagia were the most common symptoms. Hemorrhage from an esophageal vascular tumor can be massive or even fatal. Dysphagia is seen with larger lesions, but is usually mild due to esophageal elasticity. Death has been reported from obstruction and subsequent aspiration pneumonitis [4].
Lesions as large as reported herein may be detected by plain chest roentgenography. Barium swallow typically reveals a smooth, lobulated mass, a portion of which seems to be within the esophagus and the other portion outside the esophagus. Intramural esophageal hemangiomas are most often confused with leiomyomas, and less commonly intramural cysts, neurofibromas, and lipomas. The unusual feature in the present case was the septae within the mass on computed tomographic scan. This plus the massive hemorrhage suggested a diagnosis of hemangioma rather than leiomyoma.
Esophagoscopy of intramural hemangiomas typically demonstrates a submucosal mass, which may have bluish coloration and may be compressible. Biopsy of such lesions has anecdotally been associated with surprisingly little bleeding. Notwithstanding, it is never recommended to biopsy submucosal masses of the esophagus. Jasperson and colleagues [5] diagnosed three vascular lesions of the esophagus using Doppler ultrasound studies. Palchick and associates [6] used computed tomography and radionuclide angiography to make a diagnosis in 1 case.
Treatment of vascular esophageal tumors has been reported to include radium/x-ray therapy, endoscopic removal, endoscopic epinephrine injections, fulguration, esophagogastrectomy, and local excision by enucleation [7]. Enucleation is the treatment of choice.
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Footnotes
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Address reprint requests to Dr Taylor, 17 Black Banks Dr, St. Simon's Island, GA 31522-1408.
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References
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- Gentry RW, Dockerty MB, Clagett OT. Vascular malformations and vascular tumors of the gastrointestinal tract. Int Abst Surg 1949;88:281–323.
- Plachta AP. Benign tumors of the esophagus. Review of literature and report of 99 cases. Am J Gastroenterol 1962;38:639–52.
- Govoni AF. Hemangiomas of the esophagus. Gastrointest Radiol 1982;7:113–7.[Medline]
- Hand K, Talley NA, Hunt DR. Hemangioma of the esophagus: an unusual cause of upper gastrointestinal bleeding. Dig Dis Sci 1981;26:257–63.[Medline]
- Jaspersen D, Korner T, Schorr W, Bennenstuhl M, Hammar CH. Extragastric Dieulafoy's disease as unusual source of intestinal bleeding. Dig Dis Sci 1994;39:2558–60.[Medline]
- Palchick BA, Alpert MA, Holmes RA, Tully RJ, Wilson RC. Esophageal hemangioma: diagnosis with computed tomography and radionuclide angiography. S Med J 1983;76:1582–4.
- Riemenschneider HW, Klassen KW. Cavernous esophageal hemangioma. Ann Thorac Surg 1968;6:554–6.
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Abstract
Introduction
