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The Internet Journal of Surgery ISPUB.COM Volume 28 Number 2

Common Bile Duct Stone With Mirizzi’s Syndrome: Another Exception To Double Duct Sign And Courvoisier’s Law? S Chandramohan, J Madhusudhanan, A Anbazhagan, B Duraisamy, B Dhalla, S Chandrasekaran

Citation S Chandramohan, J Madhusudhanan, A Anbazhagan, B Duraisamy, B Dhalla, S Chandrasekaran. Stone With Mirizzi’s Syndrome: Another Exception To Double Duct Sign And Courvoisier’s Law?. The Internet Journal of Surgery. 2012 Volume 28 Number 2.

Abstract The combined dilatation of pancreatic and common bile ducts is known as double duct sign and occurs most commonly in malignancy of the head of pancreas. Courvoisier's law states that, in a patient with obstructive , if the is distended and palpable, it is not due to common bile duct stones. We present our case, a 62-year old female patient who presented to our department with obstructive jaundice. She was found to have a large common bile duct stone exhibiting both double duct sign and Courvoisier's sign. We discuss the current literature pertaining to these signs and highlight their significance in day-to-day clinical practice.

CASE REPORT Figure 1 A 62-year-old female presented with 8 days’ history Figure 1. Axial CT scan showing distended gallbladder with a large stone and a dilated common bile duct suggestive of obstructive jaundice. She had right upper quadrant discomfort, dark urine and clay stool. She denied any similar history in the past. She also had loss of appetite and weight. Clinically, she was icteric and abdominal examination revealed palpable and gallbladder and there was no other mass lesion. She was worked up with the possibility of obstructive jaundice due to a mitotic disease.

Liver function tests confirmed obstructive jaundice with a total bilirubin of 4.2 with increase in direct bilirubin. Alanine and asparte aminotransaminase levels were normal but serum alkaline phosphatase was elevated (305 IU/l; normal range, 20-140 IU/l).

She underwent ultrasound and CECT of the abdomen which revealed the following: Hepatomegaly with dilatation of intrahepatic biliary radicals in both lobes and grossly distended gallbladder with a stone in the dependent portion. The common bile duct measured 2.5cm with a solitary stone of 2.5 x 2.0cm in the distal common bile duct and dilated main (Figures 1, 2 & 3). There was no demonstrable mass lesion either in the head of pancreas or in the periampullary region. Both end and side-viewing endoscopies were done to rule out periampullary pathology.

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Figure 2 choledochoduodenostomy using 000 vicryl (Figure 4, 5, 6). Figure 2. Axial CT scan showing combined dilatation of She had an uneventful recovery and is on regular follow-up. common bile duct and main pancreatic duct: ‘double duct sign’ Figure 4 Figure 4. Intraoperative picture showing cholecysto- choledochal fistula (Mirizzi syndrome - arrow) with dilated common bile duct (forceps pointing to the stone inside)

Figure 3 Figure 3. Axial CT scan showing a large common bile duct stone in the distal common bile duct

Figure 5 Figure 5. Retrieved common bile duct stone

She was optimized and taken up for surgery. At surgery, she was found to have a grossly distended and thick-walled gallbladder with a stone, Type II Mirizzi’s syndrome with a cholecysto-choledochal fistula, and a 2.5-cm common duct with a solitary mobile stone. There was no malignancy anywhere. Careful dissection of Calot’s triangle was done and the site of the cholecysto-choledochal fistula was visualized. As the fistula was broad, she underwent subtotal and the gallbladder stump was closed with 00 vicryl without any compromise of the common bile duct lumen. Choledochotomy was then done, the CBD stone was extracted and the procedure completed with a

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Figure 6 concluded that this sign will be more useful in localising the Figure 6. Completed choledocho-duodenostomy level of obstruction to the head region rather than identifying the etiology. Our patient exhibited this first peculiar feature of a 'double duct sign' due to a common bile duct stone. Though it presented with double duct sign, the stone was a mobile one which could be easily removed through supraduodenal choledochotomy.

The second peculiar feature presented by this patient is the palpable gallbladder in the presence of a common bile duct stone. In 1890, Ludwig G. Courvoisier observed in his 187 patients with common bile duct obstruction, that gallbladder dilatation seldom occurred in stone obstruction of the duct [10]. This gradually came to be represented in the literature and taught as Courvoisier's law or sign. This is attributed to the contraction and fibrosis of the gall bladder due to recurrent . However, as it happens with any rule DISCUSSION in medicine, there are exceptions. Those are impacted stone at Hartmann's pouch, chronic , autoimmune In 1976, Freeny et al., in their study of endoscopic pancreatitis, biliary ascariasis, AIDS-related cholangiopathy, retrograde cholangiopancreatography (ERCP) in pancreatic choledochal cysts, common hepatic duct obstruction and carcinoma patients, reported that an irregular encasement or double pathology like distal malignancy with obstruction of pancreatic duct occurred exclusively in xanthogranulomatous cholecystitis as listed by Fitzgerald et carcinoma patients [1]. They stated that the accuracy al. in 2009 [11]. increased if it is associated with dilatation of common bile duct that is the presence of 'double duct sign', which is Thus, our patient with a palpable gallbladder and calculous probably its first description. Subsequently, the ‘double duct bile duct obstruction is yet another exception to this 'law.' In sign’ was used to describe the combined dilatation of bile 1999, Munzer reported a series of 86 patients with distended duct and pancreatic duct in other modalities of imaging like gallbladder: 83% resulted from a distal malignant ultrasound, CT scan and MRCP [2][3]. obstruction while 15% were due to bile duct stones [12]. This throws light on the fact that the presence of The most common causes of 'double duct sign' were Courvoisier's sign need not always mean a distal pancreatic adenocarcinoma and ampullary adenocarcinoma malignancy. However, the reverse is true that the gallbladder for which this sign was initially described [4]. However, may not be palpable in all cases of malignant distal bile duct numerous further reports also identified this to be present in obstruction. . In these patients, the duct is usually irregular with intraductal calculi unlike the smooth uniform CONCLUSION enlargement in pancreatic head malignancy [5]. Other Traditionally, Courvoisier's sign and double duct sign are pancreatic causes are lymphoma and metastasis arising in the viewed as indicators of underlying malignancy and help head region. Anecdotal causes reported were sphincter of differentiate from common bile duct stone. However, our Oddi dysfunction, primary retroperitoneal fibrosis and case and the accumulating evidence suggest that they may Kaposi sarcoma [6][7][8]. not be accurate in predicting malignancy and can even be A ductal stone producing 'double duct sign’ is rarely present in ductal stone. Hence we conclude stating that these described. It appears as a solitary case in a series of 77 signs should be interpreted in conjunction with other patients described by Edge et al. in 2007 [9]. In a study of 10 findings in a given patient to arrive at a clinical or patients with dilated pancreatic duct, Fishman et al., in 1979, radiological diagnosis. discussed about a patient (not reported) with impacted References ampullary stone producing double duct dilation [5]. They 1. Freeny PC, Bilbao MK, Katon RM: "Blind" evaluation of

3 of 5 Common Bile Duct Stone With Mirizzi’s Syndrome: Another Exception To Double Duct Sign And Courvoisier’s Law? endoscopic retrograde cholangiopancreatography (ERCP) in Gastrointest Endosc; 2000; 52(1): 74-7. the diagnosis of pancreatic carcinoma: the "double duct" and 7. Pereira-Lima JC, Krömer MU, Adamek HE, Riemann JF: other signs. Radiology; 1976; 119(2): 271-4. Cholestatic jaundice due to Ormond's disease (primary 2. Soto JA, Alvarez O, Múnera F, Velez SM, Valencia J, retroperitoneal fibrosis). Hepatogastroenterology; 1996; Ramírez N: Diagnosing bile duct stones: comparison of 43(10): 992-4. unenhanced helical CT, oral contrast-enhanced CT 8. Sharma M, Mahadevan B: An unusual cause of double , and MR cholangiography. AJR Am J duct sign. Type I sphincter of Oddi dysfunction due to Roentgenol; 2000; 175(4): 1127-34. chronic opium addiction. ; 2011; 140(3): 3. Nino-Murcia M, Jeffrey RB Jr, Beaulieu CF, Li KC, e1-2. Rubin GD: Multidetector CT of the pancreas and bile duct 9. Edge MD, Hoteit M, Patel AP, Wang X, Baumgarten DA, system: value of curved planar reformations. AJR Am J Cai Q: Clinical significance of main pancreatic duct dilation Roentgenol; 2001; 176(3): 689-93. on computed tomography: single and double duct dilation. 4. Baillie J, Paulson EK, Vitellas KM: Biliary imaging: a World J Gastroenterol; 2007; 13(11): 1701-5. review. Gastroenterology; 2003; 125(5): 1565. 10. Courvoisier LG: The pathology and surgery of the biliary 5. Fishman A, Isikoff MB, Barkin JS, Friedland JT: tract, Leipzig, FCW Vogel. 1890; 58. Significance of a dilated pancreatic duct on CT examination. 11. Fitzgerald JE, White MJ, Lobo DN: Courvoisier's AJR Am J Roentgenol; 1979; 133(2): 225-7. gallbladder: law or sign? World J Surg; 2009; 33(4): 886-91. 6. Menges M, Lerch MM, Zeitz M: The double duct sign in 12. Munzer D: Assessment of Courvoisier's law. Saudi J patients with malignant and benign pancreatic lesions. Gastroenterol; 1999; 5(3): 106-12.

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Author Information SM Chandramohan, M.S., M.Ch. FACS Professor and Head, Department of Surgical Gastroenterology and Center of Excellence for Upper Gl Surgery, Madras Medical College and Rajiv Gandhi Government General Hospital

J Madhusudhanan, M.S. Senior Resident, Department of Surgical Gastroenterology and Center of Excellence for Upper Gl Surgery, Madras Medical College and Rajiv Gandhi Government General Hospital

Amudhan Anbazhagan, M.S., M.Ch Assistant Professor, Department of Surgical Gastroenterology and Center of Excellence for Upper Gl Surgery, Madras Medical College and Rajiv Gandhi Government General Hospital

Benet Duraisamy, M.S., M.Ch. Assistant Professor, Department of Surgical Gastroenterology and Center of Excellence for Upper Gl Surgery, Madras Medical College and Rajiv Gandhi Government General Hospital

Bharati Y. Dhalla, M.D. Senior Consultant Radiologist, Medall Healthcare Pvt Ltd

Sathyabama Chandrasekaran, M.D. Medall Healthcare Pvt Ltd

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