acute acalculous cholecystitis complicated by mrcp confirmed mirizzi syndrome a case report

CASE REPORT – OPEN ACCESS International Journal of Surgery Case Reports (2012) 193–195 Contents lists available at SciVerse ScienceDirect International Journal of Surgery Case Reports journal homepage: www.elsevier.com/locate/ijscr Acute acalculous cholecystitis complicated by MRCP-confirmed Mirizzi syndrome: A case report Yuri N Shiryajev a,b,∗ , Anna V Glebova a,b , Tatyana V Koryakina c , Nikolay Y Kokhanenko a a Department of Faculty Surgery named after Professor A.A Rusanov, Saint-Petersburg State Pediatric Medical Academy, Saint-Petersburg, Russian Federation Sixth Department of Surgery, Mariinsky Hospital, Saint-Petersburg, Russian Federation c Department of MRI, Mariinsky Hospital, Saint-Petersburg, Russian Federation b a r t i c l e i n f o Article history: Received 30 October 2011 Received in revised form November 2011 Accepted 15 November 2011 Available online 23 November 2011 Keywords: Acute acalculous cholecystitis Mirizzi syndrome Magnetic resonance cholangiopancreatography a b s t r a c t INTRODUCTION: Acute acalculous cholecystitis can be complicated by extrinsic compression of the common hepatic/common bile duct by the enlarged and inflamed gallbladder followed by jaundice Its mechanism is very similar to that of Mirizzi syndrome, when the bile duct is compressed from outside due to a stone impacted in the gallbladder neck or cystic duct This complication of acalculous cholecystitis is rare, with very little number of published cases PRESENTATION OF A CASE: We present a patient with compression of the common hepatic duct by an inflamed and enlarged gallbladder in the absence of stones as confirmed by magnetic resonance cholangiopancreatography (MRCP) Acute cholecystitis and jaundice resolved after conservative treatment, and the changes were shown by a follow-up MRCP five months later DISCUSSION: We were able to find only three similar cases reported in the literature In these cases, compression of the common hepatic/common bile duct by the inflamed gallbladder was confirmed by endoscopic retrograde cholangiopancreatography and intraoperatively Terminology to describe this condition has not been agreed upon We consider it as a special kind of Mirizzi syndrome CONCLUSION: To the best of our knowledge, this is the first reported case of MRCP-confirmed Mirizzi syndrome in acute acalculous cholecystitis © 2011 Surgical Associates Ltd Published by Elsevier Ltd All rights reserved Introduction Mirizzi syndrome (MS) is a relatively rare complication of gallstone disease Nowadays, it is considered not only as an extrinsic compression of the common hepatic duct by a stone impacted at the cystic duct or gallbladder neck, but also a result of a cholecystobiliary fistula caused by inflammation and pressure necrosis of the gallbladder and common hepatic duct walls by the impacted stone.1–3 The most typical clinical symptoms in MS are upper abdominal (especially right upper quadrant) pain, jaundice and fever.4–5 Surgery is the most effective treatment of MS, but it is accompanied with an increased risk of bile duct injury Severe inflammation and adhesions in the subhepatic area are very common and almost always involve the hepatoduodenal ligament and Abbreviations: MS, Mirizzi syndrome; AAC, acute acalculous cholecystitis; ERCP, endoscopic retrograde cholangiopancreatography; MRCP, magnetic resonance cholangiopancreatography; US, ultrasonography ∗ Corresponding author at: Department of Faculty Surgery named after Professor A.A Rusanov, Saint-Petersburg State Pediatric Medical Academy, Litovskaya Str., 2, 194100 Saint-Petersburg, Russian Federation Tel.: +7 812 2757362; fax: +7 812 2757326 E-mail addresses: shiryajev@yandex.ru (Y.N Shiryajev), glebova.anna@mail.ru (A.V Glebova), KoryaTV@yandex.ru (T.V Koryakina), kohanenko@list.ru (N.Y Kokhanenko) they distort the normal anatomic relationships and proportions Therefore, it is highly desirable to diagnose MS preoperatively to improve orientation in the surgical field and provide maximal safety of the procedure The clinical symptoms of MS are non-specific, and are very similar to the symptoms of choledocholithiasis Ultrasonography (US) and computed tomography have limited sensitivity,3,6 and they are considered as screening tests A diagnosis of MS has to be confirmed by other tests The most helpful diagnostic tools to show the variations in MS are either endoscopic retrograde cholangiopancreatography (ERCP) or percutaneous transhepatic cholangiography.1,4 However, these examinations are invasive and may lead to complications and even fatal outcomes During the last decade magnetic resonance cholangiopancreatography (MRCP) has been widely used and it is now considered as a good non-invasive and a very accurate imaging modality in MS and in many other diseases of the hepatopancreatobiliary area.7,8 Acute acalculous cholecystitis (AAC) can lead to the development of a condition which is very similar to MS in its clinical course and imaging findings The common hepatic duct is compressed not by a stone at the cystic duct or Hartmann’s pouch, but by the severely inflamed and enlarged gallbladder Such cases are exclusively rare We were able to identify only three well-documented cases in the literature.9–11 All these patients were operated on, and in all three cases a preoperative diagnosis was made by ERCP 2210-2612/$ – see front matter © 2011 Surgical Associates Ltd Published by Elsevier Ltd All rights reserved doi:10.1016/j.ijscr.2011.11.006 CASE REPORT – OPEN ACCESS 194 Y.N Shiryajev et al / International Journal of Surgery Case Reports (2012) 193–195 Fig MRCP demonstrated extrinsic compression of the common hepatic/common bile duct by an inflamed, enlarged gallbladder without any stones in the biliary system Fig Check MRCP months later demonstrated post-inflammatory changes of the gallbladder which became reduced in size and was deformed However its wall was not thickened The intra- and extrahepatic bile ducts were not dilated and free of stones Recently, the authors observed a patient with jaundice, developed on the background of acute cholecystitis On US examination stones in the gallbladder and bile ducts were not found The signs of biliary obstruction disappeared promptly on conservative treatment MRCP confirmed significant compression of common hepatic/common bile duct by the inflamed and enlarged gallbladder, as well as absence of stones in the biliary system Conservative treatment was undertaken, and symptoms of acute cholecystitis and jaundice gradually resolved 3rd day after admission demonstrated enlargement of the gallbladder with its wall thickening and extrinsic compression of the common bile duct without any stones in the biliary system (Fig 1) The patient’s pain and jaundice gradually resolved with conservative treatment, and laboratory findings significantly improved to normal/subnormal ranges He was discharged on the ninth day On check-up in the clinic months later the patient demonstrated no complaints, no jaundice, his abdomen was soft and not painful US findings were unremarkable The follow up MRCP (Fig 2) showed the gallbladder to be reduced in size and it was deformed Its wall was not thickened (3 mm) and without any filling defects The intra- and extrahepatic bile ducts were not dilated and free of stones Case presentation A 52-year male patient N was admitted urgently on 24.02.2011 with severe upper abdominal pain, which was especially marked in the right hypochondrium He had nausea and repeated bile vomiting These complaints started two days ago and they increased with time This was his first episode of such an attack Past medical history was unremarkable Physical examination showed moderate tenderness in the right hypochondrium and epigastrium There were no peritoneal signs Ortner’s sign and Murphy’s sign were weakly positive Laboratory data revealed leucocytosis 12,600/ml US findings: gallbladder 103 mm × 38 mm, and its wall was thickened to mm The common bile duct was dilated (12 mm); there were no stones in the gallbladder and in bile ducts After crystalloid infusion with drotaverin and metamizole sodium the patient’s pain decreased, and vomiting stopped On the next morning the pain increased again, and his skin and sclera became yellowish Palpation showed severe tenderness and mild muscular rigidity in the right hypochondrium Ortner’s sign and Murphy’s sign became highly positive Biochemical blood tests revealed increase in bilirubin (total 101 ␮mol/l, direct 25 ␮mol/l) and transaminases (ALT 295 IU/l (normal range 0–55), AST 123 IU/l (5–34)), whereas his amylase level remained normal (46 IU/l, normal range 25–110) A repeated US showed the gallbladder to be 110 mm × 43 mm, its wall was not thickened, intrahepatic bile ducts and common bile duct (3 mm) were not dilated Gastroduodenoscopy showed no significant changes AAC, complicated with jaundice, was diagnosed on the basis of clinical, laboratory and instrumental findings Conservative management was continued with addition of antibiotics MRCP on the Discussion AAC can be complicated with extrinsic compression of the common hepatic/common bile duct by the enlarged and inflamed gallbladder, which was followed by jaundice Its mechanism is very similar to MS, when the bile duct is compressed from outside due to a stone impacted at the gallbladder neck or cystic duct The first description of this condition using US and ERCP was published by R.J Ippolito in 1993.9 A very similar case was described by Italian authors in 1992.12 They described a young male patient who complained of repeated attacks of right upper abdominal pain, accompanied with jaundice, though, the biliary obstruction was not caused by AAC, but by an extremely enlarged “congestive” gallbladder due to “cystic duct syndrome” Compression of the common hepatic/common bile duct was confirmed by ERCP and intraoperatively In the literature we managed to find only two additional case reports of AAC, complicated with the bile duct compression and jaundice.10,11 Thus, this pathological entity is exceptionally rare It is interesting to note that the terminology to describe this condition has not been agreed upon The authors of these three published case reports used different terms In Ippolito’s paper9 he called the condition “Acute acalculous cholecystitis associated with common hepatic duct obstruction: a variant of Mirizzi’s syndrome” K Mergener et al.,10 entitled their article “Pseudo-Mirizzi syndrome in acute cholecystitis”, while S Ahlawat11 used the title – “Acute acalculous cholecystitis simulating Mirizzi syndrome: a CASE REPORT – OPEN ACCESS Y.N Shiryajev et al / International Journal of Surgery Case Reports (2012) 193–195 very rare condition” We believe that the above-mentioned entity can be considered to be a special kind of MS One MS classification proposed by Japanese authors in 1997 based on their experience in 30 cases.5 They defined type IV of MS as “hepatic duct stenosis as a complication of cholecystitis in the absence of calculi impacted in the cystic duct or the neck of the gallbladder” There were such cases in the authors’ series However, neither the figure, nor the presentation of a typical case allowed the readers to say whether some of these patients had acute acalculous cholecystitis or not We consider that the cases of MS with acalculous cholecystitis can be classified as Nagakawa’s type IV T Nagakawa et al believe, that “for Type IV disease, which often presents with severe stenosis of the hepatic duct, reconstruction of the biliary tract following excision of the stricture is indicated” However, we think, such a procedure is too major and unjustified Inflammatory hepaticocholedocheal strictures heal well after simple cholecystectomy, when combined with biliary drainage (by transhepatic or nasobiliary drainage as performed in the first stage ˇ of surgical treatment, or by operative T-tube drainage) R Colovi c´ et al proposed their own classification for MS.13 These authors’ MS type IV is the same as Nagakawa’s classification However, their conception of surgery is significantly different,5 and it is very similar to our approach The diagnosis of MS in acalculous cholecystitis is usually suggested by US and ERCP,9–12 or sometimes by percutaneous transhepatic cholangiography.12 As the first stage of surgical treatment, choledochal stenting was used10 to relieve jaundice All the patients were operated on: in three cases open cholecystectomy,9,11,12 in one laparoscopic cholecystectomy10 was performed In all the three cases of AAC there was gangrenous inflammation of the gallbladder.9–11 Despite the absence of biliary drainage (it was placed only in of patients), jaundice and other symptoms successfully resolved Conclusion Our case can be considered as extraordinary because of two factors First, the diagnosis of MS in acalculous cholecystitis was confirmed by MRCP, which appears to be the first time reported in the literature Second, conservative treatment was effective and surgery was avoided It allowed us to perform a check MRCP, which showed resolution of acute inflammation of the gallbladder, as well as decompression of the common hepatic/common bile duct Conflict of interest None Funding 195 Ethical approval Written informed consent was obtained from the patient for publication of this case report A copy of the written consent is available for review by the Editor-in-Chief of this journal on request Author’s contributions Yuri N Shiryajev: idea of the article, study design, data collection, data analysis, literature search, writing, revision of the manuscript Anna V Glebova: data collection, data analysis, literature search Tatyana V Koryakina: data collection, data analysis, making and interpretation of ultrasound and MRI examination Nikolay Y Kokhanenko: critical revision and supervision of the work All authors read and approve the final version of the manuscript Acknowledgements None 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Chir Iugosl 2001;48(1):65–9 (in Croatian) None Open Access This article is published Open Access at sciencedirect.com It is distributed under the IJSCR Supplemental terms and conditions, which permits unrestricted non commercial use, distribution, and reproduction in any medium, provided the original authors and source are credited  ... literature search, writing, revision of the manuscript Anna V Glebova: data collection, data analysis, literature search Tatyana V Koryakina: data collection, data analysis, making and interpretation... presentation of a typical case allowed the readers to say whether some of these patients had acute acalculous cholecystitis or not We consider that the cases of MS with acalculous cholecystitis can... Eubanks WS, Baillie J, Branch MS Pseudo -Mirizzi syndrome in acute cholecystitis Am J Gastroenterol 1998;93(12):2605–6 11 Ahlawat S Acute acalculous cholecystitis simulating Mirizzi syndrome: a