International Journal of Surgery 8 (2010) 6–14 Contents lists available at ScienceDirect International Journal of Surgery journal homepage: www.theijs.com Review Acute oesophageal necrosis: A case report and review of the literature Andrew Day*, Mazin Sayegh Worthing and Southlands Hospitals NHS Trust, Worthing Hospital, Lyndhurst Road, Worthing BN11 2DH, UK article info abstract Article history: Aims: We discuss a case of acute oesophageal necrosis and undertook a literature review of this rare Received 18 March 2009 diagnosis. Received in revised form Methods: The literature review was performed using Medline and relevant references from the published 24 September 2009 literature. Accepted 27 September 2009 Results: One hundred and twelve cases were identified on reviewing the literature with upper gastro- Available online 1 October 2009 intestinal bleeding being the commonest presenting feature. The majority of cases were male and the mean age of presentation is 68.4 years. This review of the literature shows a mortality rate of 38%. Keywords: Black oesophagus Conclusion: Acute necrotizing oesophagitis is a serious clinical condition and is more common than Acute oesophageal necrosis previously thought. It should be suspected in those with upper GI bleed and particularly the elderly with Endoscopy comorbid illness. Early diagnosis with endoscopy and active management will lead towards an Gastrointestinal haemorrhage improvement in patient outcome. Ó 2009 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved. 1. Introduction performed. Whilst recovering from her operation, she spiked a temperature on the 3rd postoperative day and was commenced Oesophageal necrosis, which is also known as ‘‘black oesoph- on intravenous amoxicillin. On the 4th day, she had an episode of agus’’ or ‘‘oesophageal stroke’’ is defined as a dark pigmentation of coffee ground vomiting for which she was treated conservatively. A the oesophagus associated with necrosis of the oesophageal nasogastric tube was placed and 1000 ml of dark fluid was drained mucosa on histological examination. It is important not to assume overnight. that every case of black oesophagus is caused by oesophageal On the 6th postoperative day, she developed signs of peritonitis necrosis, and for this reason we prefer to use the term acute and was referred to the surgical team. A laparoscopy was per- oesophageal necrosis (AON) to avoid any confusion. formed which showed a turbid coloured fluid within the peritoneal We present a case of acute oesophageal necrosis and we will be cavity. We proceeded to perform a laparotomy through an upper discussing the literature surrounding this rare entity. midline incision which showed a partial gastric volvulus, which was reduced, and a moderate to large hiatus hernia. There were multiple pin point ischaemic perforations on the posterior aspect of 2. Case report the fundus which were resected using a GIA stapler (Ethicon, Route 22, West Somerville, NJ 08876) and the staple line was oversewn A 77 year-old lady was admitted to the hospital for a routine with absorbable sutures. The decision was made to deal with this vaginal hysterectomy. Her past medical history included hyper- acute episode as quickly as possible and to repair the large hiatal tension, ischaemic heart disease, hypothyroidism, diverticulosis defect with fundoplication and gastropexy at a later date after and a hiatus hernia. Her medications on admission included daily making a full recovery. thyroxine, atenolol and statins. Following an initial improvement for a week, she then became During the vaginal hysterectomy, a dark brownish fluid was increasingly short of breath and suffered an episode of melaena on noted coming from the pelvis. As there was a concern of possible the 8th day after her second laparotomy. Her blood results showed bowel injury, a lower midline laparotomy was carried out which a gradual decrease in her haemoglobin with a drop of 2 g/dl along revealed a normal looking small and large bowel. There was an with an increase in her inflammatory markers (CRP-176 and WCC- endometriotic cyst assumed to be the source of the darkish brown 31.7). Her antiphospholipid antibodies were negative. An urgent fluid. An uncomplicated right salpingo-ophorectomy was oesophageo-gastro-duodenoscopy was performed which showed circumferential black pigmentation of the entire oesophagus with * Corresponding author. Mobile: þ44 078 6636 5877. a large hiatus hernia (Fig. 1). There was also a gastric volvulus and E-mail address: [email protected] (A. Day). a large duodenal ulcer, but there was no evidence of fresh bleeding. 1743-9191/$ – see front matter Ó 2009 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ijsu.2009.09.014 A. Day, M. Sayegh / International Journal of Surgery 8 (2010) 6–14 7 Fig. 1. Circumferential black pigmentation of the oesophagus encountered at OGD. Fig. 3. A post mortem specimen showing the black oesophagus with clear demarca- tion at the cricopharyngeus. The previous gastrectomy staple line was intact. The discoloration of the oesophagus was attributed to haematin staining. She was was all within the chest cavity through a large hiatus hernia. A subsequently taken to the intensive care unit for resuscitation and left thoracotomy was performed, which revealed a large pleural monitoring and commenced on intravenous proton pump effusion and the volvulus of the stomach with necrosis of the inhibitors. fundus. Upon dissecting the stomach from the hiatus, the A CT scan was performed, showing clear pulmonary arteries, necrosis was noted to extend into the oesophagus. The upper large bilateral pleural effusions and consolidation. There was a fluid extent of the necrosis was high and could not be determined. It filled dilated gastric volvulus in the posterior mediastinum. Small was therefore not possible to anastomose the oesophagus to the pockets of air were noted adjacent to the oesophagus, but no gross distal stomach. A proximal gastrectomy and oesophagostomy was free air within the abdomen. A left sub-phrenic collection was attempted. Due to the severity of the necrosis, and with deteri- noted in addition to an ill defined mass anteriosuperior to the oration of her condition and persistent sepsis she died later that pancreas and posterior to the left lobe of the liver, possibly a hae- night. matoma (Fig. 2). The post mortem showed a blackened, thinned oesophagus In view of the deterioration of her condition and the above CT compatible with necrosis involving the remaining upper half of the findings, the patient returned to theatre for a further laparotomy. oesophagus (Fig. 3). Histology confirmed acute necrosis confined to An upper midline incision was performed to drain the left sub- the mucosa but in some areas it extended more deeply to involve phrenic collection, which turned out to be an infected haema- submucosa and muscularis propria (Fig. 4). A report of acute toma. There was an organo-axial volvulus of the stomach which oesophageal necrosis of unknown aetiology was made. Fig. 2. A CT scan of the chest which shows a large well defined fluid filled structure representing a gastric volvulus in the posterior mediastinum. Fig. 4. Histology specimen identifying mucosal necrosis of the oesophagus. 8 A. Day, M. Sayegh / International Journal of Surgery 8 (2010) 6–14 2.1. Introduction Table 2 Associated conditions. Acute oesophageal necrosis or ‘‘black oesophagus’’ used to be Gastric outlet obstruction4 considered a rare entity. It was first described in post mortem cases Gastric volvulus13 1 2 Ischemia2,14,43,45 by Brennan and Lee and colleagues. A French report indicated 15,39 3 Shock a prevalence of 0.2% and its incidence has been reported by various 16,17 Hypersensitivity to antibiotics 4 5 groups to be between 0.01 – 0.0125%. There has been new interest Viral infections18,40,42 in this condition over the last few years and it seems that it may Associated with erythema multiforme or Stevens–Johnson syndrome19,20 perhaps occur more frequently than initially thought. In Others: anticardiolipin antibodies21 and hypothermia1 a prospective 1 year study by Ben Soussan and colleagues,6 they Irradiation Trauma22,23 reported that acute oesophageal necrosis was present in 0.2% of their 3900 patients who underwent an upper GI endoscopy. Augusto et al.7 reported the condition in 29 of 10,295 patients who 2.3. Histology had an upper gastrointestinal endoscopy (0.28%) during a 5 year period. In their large retrospective series they found that 83% of AON appears as severe mucosal and submucosal necrosis with those with the disease had comorbid conditions. inflammation of the muscle fibres. There may be thrombosis of the 5 It is important to differentiate acute oesophageal necrosis from blood vessels. In the majority of cases, there is a circumferential other diseases causing a black oesophagus on endoscopy such as necrosis which is more prominent in the lower third of the oesoph- 37 melanosis8 (which is more common among the Japanese), malig- agus demarcating it from the cardia which seems to be spared. 12 nant melanoma,9 pseudomelanosis,10 acanthosis nigricans,11 In a study by Jacobsen and colleagues, it was reported that exogenous dye ingestion and coal dust exposure,46 (see Table 1). At oesophageal necrosis was distinguished from autolytic changes by autopsy it is often misinterpreted as haematin discolouration, the presence of vital reaction (i.e. fibrin exudation and presence of which is the most important differential diagnosis.12 neutrophils). They found that oesophageal necrosis which pre- The majority of cases have no real known aetiology, but there is sented as dark coloured or black, punctuate, striped or confluent a large list of conditions that are associated with acute oesophageal areas, most frequently located in the distal oesophagus, was necrosis (see Table 2). Although the majority of patients have an observed in 10.3% (32 of the 310 patients). The necrosis extended adverse outcome, there have been a few cases whereby the patient into or through the muscularis propria in nine patients.