Ann Thorac Surg 1995;59:1021-1022
© 1995 The Society of Thoracic Surgeons
Case Reports
Massive Esophageal Bleeding in Achalasia Complicated by Cytomegalovirus Esophagitis
Roche J. Featherstone, MD,
L. Gabriel Camero, MD,
Riad Khatib, MD,
Daniel Snower, MD,
Praveena Mungara, MD
St. John Hospital and Medical Center, Detroit, Michigan
Accepted for publication August 20, 1994.
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Abstract
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A 69-year-old man with achalasia who had received cytoxan and prednisone over a 6-week period for presumed Wegener's granulomatosis presented with massive esophageal bleeding. He did not respond to aggressive medical management, and an emergent esophagectomy was performed. Histologic examination revealed extensive cytomegalovirus esophagitis. He had a long but progressive hospital course and was discharged 1 month after admission. This case illustrates that cytomegalovirus esophagitis may cause massive hemorrhage that necessitates surgical intervention.
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Introduction
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The patient, a 69-year-old man, presented to our Emergency Department with hematemesis. His past medical history was significant for achalasia of greater than 10 years' duration. Six weeks before this admission he was noted to have persistent proteinuria; a kidney biopsy was performed, the results of which were thought to be consistent with Wegener's granulomatosis. He was started on a regimen of prednisone (20 mg every 6 hours) and cyclophosphamide (50 mg every 12 hours). One week after beginning therapy, upper gastrointestinal bleeding developed that resolved spontaneously. Endoscopy at that time revealed no active bleeding, and he was treated with ranitidine (150 mg every 12 hours). On this admission bleeding was massive and his hemoglobin level fell to 8.0 g/dL.
He was admitted to the surgical intensive care unit for resuscitation. Esophagogastroscopy performed at bedside revealed profuse active bleeding from the mid-esophagus that precluded a thorough assessment of the esophagus. The stomach could not be visualized. Attempts to control the bleeding with intravenous pitressin and balloon tamponade with a Sengstaken-Blakemore tube were unsuccessful. The bleeding was massive, and after multiple blood transfusions, emergency surgical intervention was performed.
The patient was taken to the operating room immediately and a right posterolateral thoracotomy was performed. A huge, purplish esophagus, secondary to venous congestion, and a large bloody pleural effusion were found. Dissection of the esophagus was cumbersome due to diffuse periesophageal adhesions and scarring. These areas bled easily as the dissection was performed. The aortic branches to the esophagus were large and friable, as were the veins. This led to significant, continuous bleeding. Nevertheless, the dissection continued in spite of hypotension into the 60 mm Hg systolic range, until the intrathoracic esophagus was excised. At this point, the bleeding slowed and the patient's condition stabilized with a systolic blood pressure of 120 to 130 mm Hg. The options of an esophagostomy and gastrostomy versus completion of gastric mobilization with esophagogastrostomy were considered. Because the patient's condition remained stable, we elected to proceed with the gastric mobilization and esophagogastrostomy, which the patient tolerated well.
Postoperatively, blood continued to ooze from the chest tube, and 24 hours later the patient was taken for a repeat thoracotomy. This exploration revealed bleeding from a vessel involved in the gastric mobilization. Bleeding was controlled and the patient was returned to the intensive care unit. The patient remained in the intensive care unit for approximately 4 weeks. He received 31 units of packed red blood cells and 12 units of fresh frozen plasma. Pathologic study showed the esophageal wall to be markedly thickened. The mucosal surface had multiple areas of small ulceration. Histologic examination confirmed the marked hypertrophy of the muscularis propria consistent with achalasia. The areas of ulceration revealed a fibropurulent exudate with acute inflammation and granulation tissue (Fig 1
) reaching to the superficial layers of the muscularis propria. Within the inflammatory exudate and in the granulation tissue at the base of the ulcers were numerous intranuclear and intracytoplasmic inclusions consistent with cytomegalovirus (CMV) infection (see Fig 1, inset a). The inclusions within the inflamed granulation tissue were located in both endothelial cells and fibroblasts. Immunohistochemical staining with antibodies to CMV and herpes simplex virus found the inclusions to be positive for CMV (Fig 1, inset b) and negative for herpes simplex virus, confirming that the inclusions were due to CMV. No evidence of neoplasm or vasculitis was identified. Review of previous medical records from other facilities showed that he had evidence of membranous glomerulonephritis. The diagnosis of Wegener's granulomatosis was not substantiated. Administration of cyclophosphamide was not resumed, the prednisone dose was tapered, and he received a 2-week course of intravenous gancyclovir. Testing for human immunodeficiency virus was negative.
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Fig 1. . Light microscopic view of the esophagus: extensive erosion of surface epithelium is visible, with inflammatory reaction and numerous internuclear inclusions. (Hematoxylin and eosin; original magnification, x40.) Inset (a): intranuclear inclusions at higher magnification. (Hematoxylin and eosin; original magnification, x400.) Inset (b): positive immunohistochemical stain for cytomegalovirus. (Cytomegalovirus immunoperoxidase stain; original magnification, x400.)
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During his hospitalization, the patient was supported with enteral feedings. On postoperative day 20, a contrast swallow demonstrated a patent gastroesophageal anastomosis with no evidence of leak. The patient was started on oral feedings and tolerated his diet well. He was then transferred to the step-down unit and subsequently was discharged home.
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Comment
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Cytomegalovirus esophagitis is a well-known entity in the medical literature [1–3]. Most often, it is found in severely immunocompromised hosts such as patients with acquired immunodeficiency syndrome or organ transplants. Occasionally, it also has been reported in immunocompetent hosts. It usually presents with dysphagia and odynophagia. Hemorrhage, although a common presenting manifestation in CMV involvement of the rest of the gastrointestinal tract, is extremely rare in esophagitis [2, 4]. Mayeux and Smith [5] described a patient on hemodialysis with chronic renal failure in whom massive hemorrhage developed a few days after she had experienced odynophagia and a small amount of hematemesis. Bleeding did not stop and she died of associated complications. Our patient had a previous episode of hemorrhage that stopped spontaneously. The second episode, however, was more severe and life-threatening. We believe that CMV was the major cause of hemorrhage, based on the extent of CMV inclusion throughout the mucosa and muscularis.
The significance of detecting CMV inclusions in the gastrointestinal mucosa is a matter of controversy. Whether it represents a superinfection of already diseased tissue or an original process is uncertain. In our patient, preexisting esophageal tissue damage secondary to achalasia was probably extensive. Cytomegalovirus reactivation after immunosuppression with cyclophos- phamide and prednisone resulted in superimposed infection with subsequent massive esophageal bleeding. Whatever the mechanism may be, this report illustrates that CMV esophagitis should be considered as a possible cause of massive and life-threatening upper gastrointestinal bleeding intractable to medical therapy. It also shows that this pathologic condition may develop after a short course of immunosuppression in patients with previous esophageal disorders. Finally, it demonstrates that prompt surgical intervention may be essential to change the outcome of this potentially devastating complication.
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Footnotes
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Address reprint requests to Dr Featherstone, St. John Hospital and Medical Center, 22101 Moross Rd, Detroit, MI 48236.
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References
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- Villar LA, Massanari RM, Mitros FA. Cytomegalovirus infection with acute erosive esophagitis. Am J Med 1984;76:924–8.[Medline]
- Wilcox CM, Diehl DL, Cello JP, Margaretten W, Jacobson MA. Cytomegalovirus esophagitis in patients with AIDS. Ann Intern Med 1990;113:589–93.[Abstract/Free Full Text]
- Balthazar EJ, Meigbow AJ, Hulnick DH. Cytomegalovirus esophagitis and gastritis in AIDS. Am J Radiol 1985;144:1201–4.[Abstract/Free Full Text]
- Cheung ANY, Ng IOL. Cytomegalovirus infection of the gastrointestinal tract in non-AIDS patients. Am J Gastroenterol 1993;88:1882–6.[Medline]
- Mayeux GP, Smith JW. Massive esophageal bleeding from cytomegalovirus esophagitis [Letter]. Am J Gastroenterol 1990;85:626.
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Abstract
Introduction
