Acute esophageal necrosis: possible association with terlipressin.

A 75-year-old man was admitted to our department with abdominal pain, hematemesis, and melena. His significant medical history included erosive gastritis, alcohol-related chronic liver disease, and chronic pancreatitis. He was not receiving any medication. His blood pressure was low (80/50mmHg); results of laboratory testing showed macrocytic anemia and liver dysfunction (hemoglobin 11.8g/dL, mean cell volume [MCV] 106.4 fL, international normalized ratio [INR] 1.53). After a second episode of hematemesis, his hemoglobin dropped to 8.9g/dL and he was treated by infusion of a colloidal solution, two units of packed red blood cells, a proton pump inhibitor, and terlipressin (2mg every 4 hours). Endoscopy showed a black mucosa (●" Fig.1a) that started from the upper esophagus and ended abruptly at the cardia. At that level, we identified an ulcer extending circumferentially in which there was a large exposed vessel (●" Fig.1b), which was treated by application of a Hemoclip.The stomach and duodenum were intact. Brushings were negative for cytomegalovirus. Broad-spectrum antibiotics, antifibrinolytic drugs, and parenteral nutrition were commenced; terlipressinwas stopped. Endoscopy at day 8 showed a clear margin between the intact proximal esophagus and its lower portion (●" Fig.2a). The luminal circumference decreased craniocaudally, ending in a stricture at the cardia (●" Fig.2b). At day 16, the distal esophagus appeared stenotic but was passable and enteral nutrition was resumed. The patient was discharged 25 days after admission. A month later, endoscopy revealed almost complete restoration of the mucosa. Notably, at the cardia we observed a Schatzki ring (●" Fig.3). A further endoscopy 8 months later showed no abnormal esophageal findings. Acute esophageal necrosis is characterized by a circumferential mucosal blackening involving the distal esophagus and occasionally extending upstream that stops abruptly at the gastroesophageal junction [1]. Ulceration of the cardia, as in this case, is uncommon; however, similar cases have been reported [2]. Ischemia, impaired mucosal defenses, and chemical insult seem to contribute to its pathogenesis [3]. The distal esophagus has been shown to be less vascularized in angiographic studies [2,3], arguably making it susceptible to local hypoperfusion caused by low splanchnic blood flow. In the case described, such a state could have resulted from hemorrhage and hypotension. Furthermore, because of the signs of liver dysfunction and the history of alcohol abuse, which suggested variceal bleeding, the patient received terlipressin, a splanchnic vasoconstrictor that may have reduced microcirculatory perfusion, further contributing to the local ischemia [4]. Although cutaneous necrosis following terlipressin treatment has been reported [5], this is the first reported case of a possible association with acute esophageal necrosis.