Biliary pancreatitis in a duplicate gallbladder: a case report and review of literature

Biliary pancreatitis in a duplicate gallbladder: a case report and review of literature Duplicated gallbladder is a rare congenital anomaly that require special attentions due to its clinical, surgical and diagnostic difficulties. We present a case of a 39-year-old female patient with a duplicated gallbladder who presented with an acute bil- iary pancreatitis, a case to our knowledge is the first in the literature. A double gallbladder in an abdominal ultrasonography was doubtful, thus a computed tomography scan, a magnetic resonance cholangiopancreatography and an endoscopic retrograde cholangiopancreatography were done that confirmed the double gallbladder. A laparoscopic cholecystectomy with an intraoperative cholangiography was performed safely two months after the acute attack. The histopathological report revealed a Y-shaped type 1 double gallbladder according to the Harlaftis et al. classification. both gallbladders should be removed to avoid complications [3]. INTRODUCTION Reinisch et al. [4] re-operated a 73-year-old patient 17 years after Double gallbladder is a rare congenital variation, with an inci- his cholecystectomy due to an acute cholecystitis in his accessory dence of 1:4000, twice high in women as in men [1]. An autopsy gallbladder which was not detected during the first surgery. done by Blasius reported the first accessory gallbladder in 1674. However, it was not until 1911 that Sherren documented the first CASE REPORT ever double gallbladder in a living patient [2]. Accurate preopera- tive diagnosing is crucial, double gallbladder can be missed during A 39-year-old healthy female, presented to our emergency preoperative imaging [3]. Abdominal ultrasonography (US) (which department due to abdominal pain, nausea and vomiting. She is operator dependent) and computed tomography (CT) scans may was febrile, with a tender right hypochondrial and epigastric not give us sufficient visualization of the biliary anatomy to detect region. Her laboratory results showed a normal complete blood theses kind of anomalies [3]. Magnetic resonance cholangiopan- count and C-reactive protein level. Liver function test showed creatography (MRCP) has a superior diagnostic capability than US. an elevation in total bilirubin at: 42 mmol/L, direct bilirubin level However, endoscopic retrograde cholangiopancreatography (ERCP) at: 29 mmol/L, gamma-glutamyl transferase (GGT) level at :160 U/L is considered the gold standard for diagnosing these rare anomal- and Lipase level at: 34 000 U/L. An abdominal US showed two sep- ies. When confronting an accessory gallbladder intraoperatively, arate gallbladders with a sludge (Fig. 1). Abdominal CT scan Contributed equally to the work. Received: February 27, 2018. Accepted: May 3, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 H. Alratrout et al. showed a Balthazar grade C pancreatitis and confirmed the pres- ence of a double gallbladder (Fig. 2). An MRCP confirmed the dou- ble gallbladder (Fig. 3). An ERCP was performed with evacuation of biliary debris in the common bile duct (CBD). The patient was dis- charged home after appropriate medical treatment a couple of days later with a full normal liver function test. Two months later, a laparoscopic cholecystectomy was performed where the two gallbladders were dissected with a dome-down technique, from the gallbladder fundus towards the neck (Fig. 4), the cystic duct and artery were identified. An intraoperative cholangiography was performed which showed patent intrahepatic ducts, cystic and CBD (Fig. 5). A Hem-o-lock clip (WECK Closure System; Teleflex Inc., Morrisville, NC, USA) was then placed on the main cystic duct (Fig. 6), and another Hem-o-lock clip was placed on the cystic artery. Figure 7 showing the gross specimen. The final histopath- ology report concluded two separate gallbladders, each having its own cystic duct, with both cystic ducts joining to form a main cys- Figure 1: Ultrasonography showing the two distinct gallbladders (1 and 2) with tic duct. sludge in one of them (arrowhead). Figure 2: CT scan in frontal and axial slices showing two distinct gallbladders (1 and 2) with cholelithiasus in one of them. Figure 3: MRI with T2-weighted, T1-weighted images and MRCP showing two distinct gallbladders and one main cystic duct (arrowhead). Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Biliary pancreatitis in a duplicate gallbladder 3 Figure 4: Laparoscopic dissection of the double gallbladder. Figure 6: Laparoscopic view showing two separate cystic ducts joining to form one main cystic duct, with a clip on it. Figure 7: Gross specimen examination. (Fig. 8). Type 1, or split primordial group, has one single cystic duct draining in the CBD, it’s subdivided into septated, V- Figure 5: Intraoperative cholangiography. shaped (two gallbladders joining at the neck level), or Y-shaped (two separate cystic ducts joining to form one single cystic duct DISCUSSION that drains into the CBD). Type 2, or the accessory gallbladder During the seventh week of gestation, the gallbladder arises group, has more than one cystic duct draining into the CBD. H or from the caudal aspect of the hepatic diverticulum which is a Ductular type and right or left hepatic duct trabecular type. In the H or Ductular type, the accessory cystic duct connects to the ventral outgrowth of the human foregut. The duplication of the gallbladder usually occurs due to outpouching from the extra- CBD. In the trabecular type the accessory cystic duct connects to the right hepatic duct. Type 3 accessory gallbladder includes hepatic biliary system during the fifth and sixth weeks of gesta- tion. If those outpouchings persist, they form an accessory rare anomalies that do not fit either type 1 nor type 2 (e.g. triple gallbladder). A modified Harlaftis classification has been gallbladder [5]. The anatomical variations of accessory gallblad- ders have been classified by several authors, Boyden being the reported in the literature describing a left trabecular variant to type 2 classification [7] (Fig. 8). In our case, the double gallbladder first in 1929, where he reported 20 cases of double gallbladder in the literature from 1674 to 1929 [6]. He classified these anom- was a Y-shaped type 1. Any complications that can occur in a alies into: vesica fellea divisa (bi-lobed gallbladder with a single single gallbladder may occur in a double gallbladder [8, 9]. The cystic duct), and vesica fellea duplex (true gallbladder duplica- most common being stone formation. Preoperative diagnosis of double gallbladder is important to prevent possible surgical tion). The latter being divided into Y-shaped (two cystic ducts uniting before entering the CBD); and H-shaped (two cystic complications. However, only one half of all cases of double gall- ducts enter separately into the CBD) duplication. In 1977, bladders are diagnosed preoperatively.When a double gallblad- Harlaftis et al. [5] modified the classification by describing three der is suspected, MRCP, allows visualization of the two independent gallbladders and, as the case may be, the presence main types of gallbladder duplication based on embryogenesis Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 4 H. Alratrout et al. Figure 8: Harlaftis et al. classification for accessory gallbladder. of separate cystic ducts. We highly recommend intraoperative 2. Paraskevas GK, Raikos A, Ioannidis O, Papaziogas B. cholangiography that provides an outline to the biliary anatomy Duplicated gallbladder: surgical application and review of and reduces the risk of injury to the biliary tree. Some surgeons the literature. Ital J Anat Embryol 2011;116:61–6. have recommended open surgery in case of accessory gallblad- 3. Fazio V, Damiano G, Palumbo VD, Spinelli G, Scio A, der anomalies, especially in type 2 double gallbladder [10], Tomasello G, et al. An unexpected surprise at the end of a because the risk of injuring the CBD or the right hepatic artery ‘quiet’ cholecystectomy. A case report and review of the lit- during dissection is higher. Yet, we believe that accurate pre- erature. Ann Ital Chir 2012;83:265–7. operative planning, makes the laparoscopic approach by an 4. Reinisch A, Brandt L, Fuchs K-H. Doppelt angelegte experienced surgeon a safe option. Gallenblase Laparoskopische Cholezystektomie 17 Jahre nach konventioneller Cholezystektomie. Zentralblatt Für Chir 2009;134:576–9. CONCLUSION 5. Harlaftis N, Gray SW, Skandalakis JE. Multiple gallbladders. Surg Gynecol Obstet 1977;145:928–34. Double gallbladder is a challenging rare congenital anomaly. 6. Boyden EA. The accessory gallbladder—an embryological Accurate preoperative imaging and high index of suspicion are and comprarative study of aberrant biliary vesicles occur- required in order to avoid surgical complications. We recom- ring in man and the domesticmammals. Am J Anat 1926;38: mend these cases to be managed by an experienced laparo- 177–231. scopic surgeon or a hepatobiliary surgeon. 7. Kim RD, Zendejas I, Velopulos C, Fujita S, Magliocca JF, Kayler LK, et al. Duplicate gallbladder arising from the left hepatic duct: report of a case. Surg Today 2009;39:536–53. CONFLICT OF INTEREST STATEMENT 8. Brady K, Mitchell A. Cholecystitis of a duplicated gallbladder No conflict of interest. complicated bya cholecystoenteric fistula. Pediatr Radiol 2009;39:385–8. 9. Garcia JC, Weber A, Berry FS, Tanur B. Double gallbladder REFERENCES treated successfullyby laparoscopy. J Laparoendosc Surg 1993; 3:153–5. 1. Boyden EA. The accessory gallbladder—an embryological 10. Miyajima N, Yamakava T, Varma A, Uno K, Ohtaki S, Kano and comparative study of aberrant biliary vesicles occurring N. Experience with laparoscopicdouble gallbladder removal. in man and the domestic mammals. Am J Anat 1926;38: Surg Endosc 1995;9:63–6. 177–231. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Biliary pancreatitis in a duplicate gallbladder: a case report and review of literature

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Abstract

Duplicated gallbladder is a rare congenital anomaly that require special attentions due to its clinical, surgical and diagnostic difficulties. We present a case of a 39-year-old female patient with a duplicated gallbladder who presented with an acute bil- iary pancreatitis, a case to our knowledge is the first in the literature. A double gallbladder in an abdominal ultrasonography was doubtful, thus a computed tomography scan, a magnetic resonance cholangiopancreatography and an endoscopic retrograde cholangiopancreatography were done that confirmed the double gallbladder. A laparoscopic cholecystectomy with an intraoperative cholangiography was performed safely two months after the acute attack. The histopathological report revealed a Y-shaped type 1 double gallbladder according to the Harlaftis et al. classification. both gallbladders should be removed to avoid complications [3]. INTRODUCTION Reinisch et al. [4] re-operated a 73-year-old patient 17 years after Double gallbladder is a rare congenital variation, with an inci- his cholecystectomy due to an acute cholecystitis in his accessory dence of 1:4000, twice high in women as in men [1]. An autopsy gallbladder which was not detected during the first surgery. done by Blasius reported the first accessory gallbladder in 1674. However, it was not until 1911 that Sherren documented the first CASE REPORT ever double gallbladder in a living patient [2]. Accurate preopera- tive diagnosing is crucial, double gallbladder can be missed during A 39-year-old healthy female, presented to our emergency preoperative imaging [3]. Abdominal ultrasonography (US) (which department due to abdominal pain, nausea and vomiting. She is operator dependent) and computed tomography (CT) scans may was febrile, with a tender right hypochondrial and epigastric not give us sufficient visualization of the biliary anatomy to detect region. Her laboratory results showed a normal complete blood theses kind of anomalies [3]. Magnetic resonance cholangiopan- count and C-reactive protein level. Liver function test showed creatography (MRCP) has a superior diagnostic capability than US. an elevation in total bilirubin at: 42 mmol/L, direct bilirubin level However, endoscopic retrograde cholangiopancreatography (ERCP) at: 29 mmol/L, gamma-glutamyl transferase (GGT) level at :160 U/L is considered the gold standard for diagnosing these rare anomal- and Lipase level at: 34 000 U/L. An abdominal US showed two sep- ies. When confronting an accessory gallbladder intraoperatively, arate gallbladders with a sludge (Fig. 1). Abdominal CT scan Contributed equally to the work. Received: February 27, 2018. Accepted: May 3, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 H. Alratrout et al. showed a Balthazar grade C pancreatitis and confirmed the pres- ence of a double gallbladder (Fig. 2). An MRCP confirmed the dou- ble gallbladder (Fig. 3). An ERCP was performed with evacuation of biliary debris in the common bile duct (CBD). The patient was dis- charged home after appropriate medical treatment a couple of days later with a full normal liver function test. Two months later, a laparoscopic cholecystectomy was performed where the two gallbladders were dissected with a dome-down technique, from the gallbladder fundus towards the neck (Fig. 4), the cystic duct and artery were identified. An intraoperative cholangiography was performed which showed patent intrahepatic ducts, cystic and CBD (Fig. 5). A Hem-o-lock clip (WECK Closure System; Teleflex Inc., Morrisville, NC, USA) was then placed on the main cystic duct (Fig. 6), and another Hem-o-lock clip was placed on the cystic artery. Figure 7 showing the gross specimen. The final histopath- ology report concluded two separate gallbladders, each having its own cystic duct, with both cystic ducts joining to form a main cys- Figure 1: Ultrasonography showing the two distinct gallbladders (1 and 2) with tic duct. sludge in one of them (arrowhead). Figure 2: CT scan in frontal and axial slices showing two distinct gallbladders (1 and 2) with cholelithiasus in one of them. Figure 3: MRI with T2-weighted, T1-weighted images and MRCP showing two distinct gallbladders and one main cystic duct (arrowhead). Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Biliary pancreatitis in a duplicate gallbladder 3 Figure 4: Laparoscopic dissection of the double gallbladder. Figure 6: Laparoscopic view showing two separate cystic ducts joining to form one main cystic duct, with a clip on it. Figure 7: Gross specimen examination. (Fig. 8). Type 1, or split primordial group, has one single cystic duct draining in the CBD, it’s subdivided into septated, V- Figure 5: Intraoperative cholangiography. shaped (two gallbladders joining at the neck level), or Y-shaped (two separate cystic ducts joining to form one single cystic duct DISCUSSION that drains into the CBD). Type 2, or the accessory gallbladder During the seventh week of gestation, the gallbladder arises group, has more than one cystic duct draining into the CBD. H or from the caudal aspect of the hepatic diverticulum which is a Ductular type and right or left hepatic duct trabecular type. In the H or Ductular type, the accessory cystic duct connects to the ventral outgrowth of the human foregut. The duplication of the gallbladder usually occurs due to outpouching from the extra- CBD. In the trabecular type the accessory cystic duct connects to the right hepatic duct. Type 3 accessory gallbladder includes hepatic biliary system during the fifth and sixth weeks of gesta- tion. If those outpouchings persist, they form an accessory rare anomalies that do not fit either type 1 nor type 2 (e.g. triple gallbladder). A modified Harlaftis classification has been gallbladder [5]. The anatomical variations of accessory gallblad- ders have been classified by several authors, Boyden being the reported in the literature describing a left trabecular variant to type 2 classification [7] (Fig. 8). In our case, the double gallbladder first in 1929, where he reported 20 cases of double gallbladder in the literature from 1674 to 1929 [6]. He classified these anom- was a Y-shaped type 1. Any complications that can occur in a alies into: vesica fellea divisa (bi-lobed gallbladder with a single single gallbladder may occur in a double gallbladder [8, 9]. The cystic duct), and vesica fellea duplex (true gallbladder duplica- most common being stone formation. Preoperative diagnosis of double gallbladder is important to prevent possible surgical tion). The latter being divided into Y-shaped (two cystic ducts uniting before entering the CBD); and H-shaped (two cystic complications. However, only one half of all cases of double gall- ducts enter separately into the CBD) duplication. In 1977, bladders are diagnosed preoperatively.When a double gallblad- Harlaftis et al. [5] modified the classification by describing three der is suspected, MRCP, allows visualization of the two independent gallbladders and, as the case may be, the presence main types of gallbladder duplication based on embryogenesis Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018 4 H. Alratrout et al. Figure 8: Harlaftis et al. classification for accessory gallbladder. of separate cystic ducts. We highly recommend intraoperative 2. Paraskevas GK, Raikos A, Ioannidis O, Papaziogas B. cholangiography that provides an outline to the biliary anatomy Duplicated gallbladder: surgical application and review of and reduces the risk of injury to the biliary tree. Some surgeons the literature. Ital J Anat Embryol 2011;116:61–6. have recommended open surgery in case of accessory gallblad- 3. Fazio V, Damiano G, Palumbo VD, Spinelli G, Scio A, der anomalies, especially in type 2 double gallbladder [10], Tomasello G, et al. An unexpected surprise at the end of a because the risk of injuring the CBD or the right hepatic artery ‘quiet’ cholecystectomy. A case report and review of the lit- during dissection is higher. Yet, we believe that accurate pre- erature. Ann Ital Chir 2012;83:265–7. operative planning, makes the laparoscopic approach by an 4. Reinisch A, Brandt L, Fuchs K-H. Doppelt angelegte experienced surgeon a safe option. Gallenblase Laparoskopische Cholezystektomie 17 Jahre nach konventioneller Cholezystektomie. Zentralblatt Für Chir 2009;134:576–9. CONCLUSION 5. Harlaftis N, Gray SW, Skandalakis JE. Multiple gallbladders. Surg Gynecol Obstet 1977;145:928–34. Double gallbladder is a challenging rare congenital anomaly. 6. Boyden EA. The accessory gallbladder—an embryological Accurate preoperative imaging and high index of suspicion are and comprarative study of aberrant biliary vesicles occur- required in order to avoid surgical complications. We recom- ring in man and the domesticmammals. Am J Anat 1926;38: mend these cases to be managed by an experienced laparo- 177–231. scopic surgeon or a hepatobiliary surgeon. 7. Kim RD, Zendejas I, Velopulos C, Fujita S, Magliocca JF, Kayler LK, et al. Duplicate gallbladder arising from the left hepatic duct: report of a case. Surg Today 2009;39:536–53. CONFLICT OF INTEREST STATEMENT 8. Brady K, Mitchell A. Cholecystitis of a duplicated gallbladder No conflict of interest. complicated bya cholecystoenteric fistula. Pediatr Radiol 2009;39:385–8. 9. Garcia JC, Weber A, Berry FS, Tanur B. Double gallbladder REFERENCES treated successfullyby laparoscopy. J Laparoendosc Surg 1993; 3:153–5. 1. Boyden EA. The accessory gallbladder—an embryological 10. Miyajima N, Yamakava T, Varma A, Uno K, Ohtaki S, Kano and comparative study of aberrant biliary vesicles occurring N. Experience with laparoscopicdouble gallbladder removal. in man and the domestic mammals. Am J Anat 1926;38: Surg Endosc 1995;9:63–6. 177–231. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy112/5017815 by Ed 'DeepDyve' Gillespie user on 21 June 2018

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Published: May 29, 2018

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