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0International journal of Surgery Case Reports 3 (2012) 346-348
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International Journal of Surgery Case Reports
journal homepage www.elsevier.com/locate/ijscr
JOURNAL OF SUHCERY
CASE REPORTS
An unusual case of acalculous cholecystitis heralding presentation of acute mesenteric ischaemia with typical radiological findings
Emma Aitken *, Alison Lyon, Itamar Felstenstein
Department of General Surgery, Crosshouse Hospital, Kilmarnock I(A2 0BE, United Kingdom
ARTICLE INFO
Article history:
Received 22 October 2011
Received in revised form 28 March 2012
Accepted 12 April 2012
Available online 17 April 2012
Keywords:
Acalculous cholecystitis Mesenteric ischaemia Pneumatosis intestinalis
ABSTRACT
INTRODUCTION: Acalculous cholecystitis accounts for 10-15% of cases of cholecystitis. It is often associated with critical illness and has a high morbidity and mortality.
PRESENTATION OF CASE: We report an unusual case of an elderly lady who presented with acalculous cholecystitis as the herald event for subsequent fatal intestinal ischaemia. She demonstrated classical radiological features of pneumatosis coli and hepatic porto-venous gas (HPVG).
DISCUSSION: The pathogenesis of acalculous cholecystitis remains uncertain but theories including biliary stasis, sepsis and ischaemia have been proposed. The gallbladder is particularly vulnerable to ischaemia which may precipitate the inflammatory process. In this case, we propose that acute acalculous cholecystitis was triggered by ischaemia and was a herald sign of the ischaemia that would later affect the entire gastrointestinal tract. We suggest that the gallbladder's tenuous blood supply made it more vulnerable to the ischaemia that the rest of the bowel subsequently suffered from.
CONCLUSION: Intramural and hepatic porto-venous gas are classical, though rarely seen, CT findings in acute intestinal ischaemia. In these situations HPVG is often associated with poor outcome. In this case the acute acalculous cholecystitis may have been a herald sign of mesenteric ischaemia.
© 2012 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved.
1. Introduction
Acalculous cholecystitis accounts for 10-15% of cases of cholecystitis.1 It is often associated with critical illness and has a high morbidity and mortality.2 We report a case of acalculous cholecystitis presenting as the herald event for intestinal ischaemia with classical radiological features of pneumatosis coli and porto-venous gas.
2. Presentation of case
A 69-year-old woman initially presented with a 6-h history of sudden onset upper abdominal pain. Her past medical history was unremarkable. She was apyrexial with a borderline tachycardia of 92 beats per minute. Examination revealed localised peritonism in her right upper quadrant. Inflammatory markers were elevated withWCC31 x 109/l and CRP 57 mg/l.LFTs and amylase levels were within normal limits. CT scan of her abdomen revealed free air in the right upper quadrant with ill-defined hypodense changes in the region of the gallbladder. She underwent emergency laparo-tomy and open cholecystectomy for a perforated gallbladder. She made a relatively uneventful recovery and was discharged home on
* Corresponding author at: 14/3 350 Meadowside Quay Walk, Glasgow G11 6DT, United Kingdom. Tel.: +44 141 339 4093.
E-mail address: emmaaitken@doctors.net.uk (E. Aitken).
post-operative day 10. Pathology subsequently confirmed gallbald-der perforation secondary to acalculous cholecystitis.
She re-presented to Accident and Emergency on post-operative day 19 with a 2-day history of vomiting, diarrhoea and vague lower abdominal pain. She was hypotensive (blood pressure 80/40) and tachycardiac (heart rate 110 beats per minute). Her abdomen was distended with mild generalised tenderness, but no periton-ism. Again her inflammatory markers were elevated with WCC 28.5 x 109/l and CRP 417 mg/l. She had a profound metabolic acidosis with a base excess of -12.2 mmol/l. She was resuscitated, then under went an urgent contrast-enhanced CT scan of her abdomen. This revealed air within the wall of the stomach, small bowel and colon, along with free intraperitoneal air. Additionally there was extensive air within the portal venous system, portal vein (Fig. 1) and mesenteric vessels including the superior mesenteric artery (Fig. 2). Immediate laparotomy was performed. The entire small and large bowel was found to be ischaemic, necrotic and non-viable. The abdomen was closed and the patient died shortly afterwards.
3. Discussion
We present a case of an elderly lady presenting with acute acalculous cholecystitis as a herald sign for catastrophic mesenteric infarction.
Acalculous cholecystitis is a disease process that mainly affects the critically ill. It accounts for 5-15% of all cases of acute cholecystitis and is associated with a relatively high morbidity and mortality.1
2210-2612/$ - see front matter © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijscr.2012.04.006
E. Aitken et al.
Fig. 1. CT scan showing porto-venous gas, pneumatosis intestinalis and air within the wall of the splenic vein (red arrow).
Acalculous cholecystitis is commonly seen secondary to burns, sepsis, trauma, diabetes mellitus, vasculitis or the prolonged use of total parenteral nutrition.1
The exact pathogenesis of acalculous cholecystitis remains uncertain but theories including biliary stasis, sepsis and ischaemia have been proposed. It may be that a combination of these factors plays a role. Some authors suggest that the primary insult is cholestasis, with highly viscous biliary sludge causing distension and increased tension within the gallbladder wall, thus compromising arterial flow.1 Others suggest that the causative factor precipitating the inflammatory process is in fact ischaemia.2
Arteriography of the gallbladder has demonstrated arterial occlusion and venous opacification in acute acalculous cholecystitis in contrast to the arterial dilatation and large vessel venous filling seen in calculous cholecystitis.3 Acute acalculous cholecystitis is commonly seen post-operatively following major cardiac surgery. Mechanisms, including atherosclerosis, cardiac failure and prolonged hypotension with visceral arterial hypoperfusion, have been postulated as causative. Ischaemia and disturbed microcirculation coupled with visceral hypotension and poor capillary filling of the small vessels of the gallbladder is likely the final common
irgery Case Reports 3 (2012) 346-348 347
pathway.4 Additionally, acute acalculous cholecystitis is seen when the vascular supply to the gallbladder is compromised in vasculi-tides, e.g. polyarteritis nodosa.5
In this case, we propose that acute acalculous cholecystitis was triggered by ischaemia, a herald sign of the ischaemia that would later affect the entire gastrointestinal tract. We suggest that the gallbladder's tenuous blood supply made it more vulnerable to the ischaemia that the rest of the bowel subsequently suffered from.
To our knowledge, there has only been one other report in the literature of acute acalculous cholecystitis heralding bowel ischaemia. Dessailloud et al., 1998 describe the case of a 29-year-old patient with anti-phospholipid syndrome who presented with acute acalculous cholecystitis and subsequently died two months later from thromboses in her coeliac axis and superior mesenteric artery.6
The CT scan findings in this case are a classical, but rarely seen, radiographic presentation of mesenteric ischaemia. The presentation, with free intraperitoneal air on abdominal CT is ususual, though previously described within the literature.7
Pneumatosis intestinalis is normally seen in cases mesen-teric infarction and necrotising enterocolitis, but can also occur in Crohn's disease, diverticulitis, intestinal obstruction, cholangi-tis, pancreatitis, pneumonia and graft versus host disease. Whilst indicative of severe disease, it is not necessarily an indication for urgent laparotomy.8 Mortality would appear to be directly correlated with the length of bowel involved, with 14% mortality ifjust one vascular segment is affected; 71% mortality if two segments are affected; or, as in the case described, 100% mortality when three vascular segments are involved.9
Hepatic porto-venous gas (HPVG) is more rarely encountered. It is thought to arise as a result of intramural intestinal gas, produced by bacteria, travelling into the portovenous system, perhaps secondary to damage to the intestinal mucosa from bowel distension or sepsis. First described in 1955 by Wolfe and Evans, only about 100 cases of HPVG are seen each year in England and Wales. HPVG is indicative of ischaemia and impending bowel necrosis, the severity of which determines outcome.
HPVG on imaging is an "ominous sign",8 although with the increasing availability of CT in recent years, earlier detection of bowel ischaemia rather than necrosis has improved outcome. Twenty years ago HPVG indicated necrotic bowel in over two-thirds of cases with mortality in the region of 75%. Recent studies suggest a 71% survival in patients with HPVG on CT, half of whom do not require surgery.9 This improved prognosis is attributed to detection prior to bowel necrosis and increasing numbers of patients with iatrogenic or inflammatory causes for HPVG rather than ischaemia. There are a number of case reports of conservative treatment for HPVG,8 however some authors still advocate immediate laparotomy for HPVG.8 Despite immediate laparotomy in the case described the patient died because of the extent of ischaemia and bowel necrosis.
4. Conclusion
Intramural and hepatic porto-venous gas are classical, though rarely seen, CT findings in acute intestinal ischaemia. In these situations HPVG is often associated with poor outcome. In this case the acute acalculous cholecystitis may have been a herald sign of mesenteric ischaemia.
Conflict of interest
Fig. 2. CT scan showing porto-venous gas and air within wall of superior mesenteric artery (red arrow) and splenic artery.
None declared.
348 E. Aitken et al. / International Journal of Surgery Case Reports 3 (2012) 346-348
Funding
Ethical approval
Written informed consent was obtained from the patient's family for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-inChief of this journal on request.
Author contributions
Emma Aitken wrote case report and submitted. Itamar Felsten-stein consultant surgeon in charge of patient's care and revision of manuscript. Alison Lyon sourced and prepared images and revision of manuscript.
References
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