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Augustin G, Romic I, Miličić I, Mikuš M, Herman M. Maternal choledochal cysts in pregnancy: A systematic review of case reports and case series. World J Gastrointest Surg 2023; 15:1784-1798. [PMID: 37701693 PMCID: PMC10494602 DOI: 10.4240/wjgs.v15.i8.1784] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/20/2022] [Revised: 02/14/2023] [Accepted: 06/11/2023] [Indexed: 08/25/2023] Open
Abstract
BACKGROUND Choledochal cysts (CC) are cystic dilatations of the biliary tract, usually diagnosed during childhood, with an estimated incidence in the general population of 1:100000. Complications related to CC include rupture, biliary obstruction, and cholangitis. Maternal CC in pregnancy are rarely reported, and there are no guidelines on optimal management. AIM To systematically review maternal CC diagnosed during pregnancy or postpartum with regard to the clinical presentation of CC, the mode of treatment and delivery, and maternal outcomes. METHODS A literature search of cases and case series of maternal CC in pregnancy and postpartum was conducted using MEDLINE/PubMed, Web of Science, Google Scholar, and Embase. There were no restrictions on language or publication year. Databases were lastly accessed on September 1, 2022. RESULTS Overall, 71 publications met the inclusion criteria, reporting 97 cases. Eighty-eight cases were diagnosed during pregnancy and nine in the puerperium. The most common symptoms were abdominal pain (81.2%) and jaundice (60.4%). Interventions for CC complications were required in 52.5% of the cases, and 34% of pregnancies were induced. Urgent cesarean section (CS) was done in 24.7%. The maternal mortality was 7.2%, while fetal mortality was inconsistently reported. Cholangitis, CC > 15 cm, and bilirubin levels > 80 mmol/L were associated with a higher likelihood of urgent CS and surgical intervention for CC. Bilirubin levels positively correlated with CC size. There was no correlation between age and cyst dimension, gestational age at cyst discovery, and CC size. CONCLUSION Although rare, maternal CC in pregnancy should be included in the evaluation of jaundice with upper abdominal pain. Symptomatology and clinical course are variable, and treatment may range from an expectative approach to emergent surgical CC treatment and urgent CS. While most cases were managed by conservative measures or drainage procedures, CC > 15 cm and progressive cholangitis carry the risk of CC rupture and septic complications, which may increase the rates of unfavorable maternal and fetal outcomes. Therefore, such cases require specific surgical and obstetric interventions.
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Affiliation(s)
- Goran Augustin
- Department of Surgery, School of Medicine, University of Zagreb, Zagreb 10000, Croatia
- School of Medicine University of Zagreb, Zagreb 10000, Croatia
| | - Ivan Romic
- Department of Surgery, University Hospital Centre Zagreb, Zagreb 10000, Croatia
| | - Iva Miličić
- Department of Obstetrics and Gynecology, University Hospital Centre Zagreb, Zagreb 10000, Croatia
| | - Mislav Mikuš
- Department of Obstetrics and Gynecology, University Hospital Centre Zagreb, Zagreb 10000, Croatia
| | - Mislav Herman
- Department of Obstetrics and Gynecology, University Hospital Centre Zagreb, Zagreb 10000, Croatia
- School of Medicine University of Zagreb, Zagreb 10000, Croatia
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Zhang K, Wu L, Gao K, Yan C, Zheng C, Guo C. Strict Surgical Repair for Bile Leakage Following the Roux-en-Y Hepaticojejunostomy. Front Surg 2021; 8:641127. [PMID: 34017852 PMCID: PMC8130580 DOI: 10.3389/fsurg.2021.641127] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/13/2020] [Accepted: 03/17/2021] [Indexed: 11/13/2022] Open
Abstract
Background: The optimal bile leakage management strategy in the pediatric population following the initial Roux-en-Y hepaticojejunostomy is still a matter of discussion today. Here, we assessed the roles of bile leakage management and surgical implementation on outcomes for patients with bile leakage. Materials and Methods: A revised protocol for bile leakage management with restricted surgical intervention was implemented at Chongqing Children’s Hospital on March 15, 2013 and Sanxia Hospital on April 20, 2013. We performed a retrospective, historical control analysis for the protocol implementation to compare the short- and long-term outcomes using the corresponding statistical methods. Results: There was a total of 84 patients included in the analysis, including 46 patients in the pre-protocol group and 38 patients in the post-protocol group. No statistical differences for the demographic features were found between the two groups. There was a decrease in redo surgeries in the post-protocol cohort compared to those in the pre-protocol cohort (odds ratio [OR] = 4.48 [95% CI, 1.57–12.77]; p = 0.003). Furthermore, patients in the post-protocol group were less likely to be associated with intensive care unit (ICU) admission (OR = 3.72 [95% CI, 1.11–12.49]; p = 0.024) compared to patients in the pre-protocol group, respectively. There was no mortality between the two groups. Conclusions: A restrictive surgical intervention strategy can effectively reduce the rate of redo surgery and exhibited promising outcomes for bile leakage in terms of postoperative recovery and hospitalization costs.
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Affiliation(s)
- Keying Zhang
- College of Traditional Chinese Medicine, Chongqing Medical University, Chongqing, China
| | - Linfeng Wu
- College of Traditional Chinese Medicine, Chongqing Medical University, Chongqing, China
| | - Kai Gao
- Department of Pediatric General and Neonatal Surgery, Children's Hospital of Chongqing Medical University, Chongqing, China.,Ministry of Education Key Laboratory of Child Development and Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Department of Orthopaedics, Children's Hospital of Chongqing Medical University, Chongqing, China.,China International Science and Technology Cooperation Base of Child Development and Critical Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Key Laboratory of Pediatrics, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Engineering Research Center of Stem Cell Therapy, Children's Hospital of Chongqing Medical University, Chongqing, China
| | - Chengwei Yan
- Department of Pediatric General Surgery, Sanxia Hospital, Chongqing University, Chongqing, China
| | - Chao Zheng
- Ministry of Education Key Laboratory of Child Development and Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Department of Orthopaedics, Children's Hospital of Chongqing Medical University, Chongqing, China.,China International Science and Technology Cooperation Base of Child Development and Critical Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Key Laboratory of Pediatrics, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Engineering Research Center of Stem Cell Therapy, Children's Hospital of Chongqing Medical University, Chongqing, China.,Department of Traumatology, Children's Hospital of Chongqing Medical University, Chongqing, China
| | - Chunbao Guo
- Department of Pediatric General and Neonatal Surgery, Children's Hospital of Chongqing Medical University, Chongqing, China.,Ministry of Education Key Laboratory of Child Development and Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Department of Orthopaedics, Children's Hospital of Chongqing Medical University, Chongqing, China.,China International Science and Technology Cooperation Base of Child Development and Critical Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Key Laboratory of Pediatrics, Children's Hospital of Chongqing Medical University, Chongqing, China.,Chongqing Engineering Research Center of Stem Cell Therapy, Children's Hospital of Chongqing Medical University, Chongqing, China
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Ravichandran A, Pichaimuthu A, Arcot R. Laparoscopic excision of a Type II choledochal cyst with cystolithiasis. J Minim Access Surg 2021; 17:259-261. [PMID: 33723185 PMCID: PMC8083736 DOI: 10.4103/jmas.jmas_181_20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Choledochal cysts can present with abdominal pain, jaundice and stones.MRI is the standard imaging tool and the type of biliary enteric anastomosis depends on the cyst type.
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Affiliation(s)
- Arihanth Ravichandran
- Department of General Surgery, Saveetha Institute of Medical and Technical Sciences, Saveetha Medical College, Chennai, Tamil Nadu, India
| | - Anbalagan Pichaimuthu
- Department of General Surgery, Saveetha Institute of Medical and Technical Sciences, Saveetha Medical College, Chennai, Tamil Nadu, India
| | - Rekha Arcot
- Department of General Surgery, Saveetha Institute of Medical and Technical Sciences, Saveetha Medical College, Chennai, Tamil Nadu, India
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Chaudhry MBH, Azeemuddin M, Khan MR, Parkash O. A large choledochocystolithiasis mimicking Mirizzi syndrome. BMJ Case Rep 2017. [PMID: 28647717 DOI: 10.1136/bcr-2017-219582] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
An 18-year-old man presented with spontaneous severe epigastric pain, progressing and radiating to back since 3 days. It was associated with epigastric tenderness, bilious vomiting and jaundice. He had been intermittently experiencing these symptoms for the last 1 year. No known comorbid. Ultrasound showed a poorly visualised heterogeneous focus at porta hepatis; considering poor visualisation, this might represent an enlarged calcified lymph node or cystic duct calculus causing extrinsic compression or a large sludge ball within the common bile duct (CBD), leading to dilatation of common hepatic duct and intrahepatic biliary system. Subsequent magnetic resonance cholangiopancreatography revealed a focal saccular dilatation of middle part of CBD, a type I-B choledochal cyst, large heterogeneous focus seen within it representing choledochocystolithiasis. Later, CT was performed for further characterisation of surrounding anatomy and pathology, which confused the appearance of choledochocystolithiasis for Mirizzi syndrome. Later, surgery and histopathology confirmed type I-B choledochocystolithiasis and chronic cholecystitis.
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Affiliation(s)
| | | | | | - Om Parkash
- Department of Medicine, Aga Khan University, Karachi, Pakistan
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Sastry AV, Abbadessa B, Wayne MG, Steele JG, Cooperman AM. What is the incidence of biliary carcinoma in choledochal cysts, when do they develop, and how should it affect management? World J Surg 2015; 39:487-92. [PMID: 25322698 DOI: 10.1007/s00268-014-2831-5] [Citation(s) in RCA: 59] [Impact Index Per Article: 5.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
The incidence of cancer in choledochal cysts (CCs) in adults was calculated to determine the timing and need for surgery. In 78 publications (1996-2010), 434 of 5780 reported CCs patients had cancer. Cholangiocarcinoma (70.4 %) and gallbladder cancer (23.5 %) were the most common malignancies. Only nine malignancies were reported before age 18 (0.42 %). In contrast, the incidence of malignancy in adults was 11.4 %. The median age for diagnosis of cancer was 42 years, and the incidence increased with each decade.
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Affiliation(s)
- Amit V Sastry
- Department of Surgery, Beth Israel Medical Center, New York, NY, USA,
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