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(8): 1784-1798 [PMID: 37701693 DOI: 10.4240/wjgs.v15.i8.1784]
Corresponding Author of This Article
Goran Augustin, MD, MSc, PhD, Associate Professor, Doctor, Senior Scientist, Surgical Oncologist, Department of Surgery, University Hospital Centre Zagreb, Kišpatićeva 12, Zagreb 10000, Croatia. augustin.goran@gmail.com
Research Domain of This Article
Surgery
Article-Type of This Article
Systematic Reviews
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
World J Gastrointest Surg. Aug 27, 2023; 15(8): 1784-1798 Published online Aug 27, 2023. doi: 10.4240/wjgs.v15.i8.1784
Maternal choledochal cysts in pregnancy: A systematic review of case reports and case series
Goran Augustin, Ivan Romic, Iva Miličić, Mislav Mikuš, Mislav Herman
Goran Augustin, Department of Surgery, School of Medicine, University of Zagreb, Zagreb 10000, Croatia
Goran Augustin, Mislav Herman, School of Medicine University of Zagreb, Zagreb 10000, Croatia
Ivan Romic, Department of Surgery, University Hospital Centre Zagreb, Zagreb 10000, Croatia
Iva Miličić, Mislav Mikuš, Mislav Herman, Department of Obstetrics and Gynecology, University Hospital Centre Zagreb, Zagreb 10000, Croatia
Author contributions: Augustin G designed the research and wrote the paper; Romic I and Miličić I performed the research and wrote the paper; Mikuš M and Herman M analyzed the data and performed the literature review.
Conflict-of-interest statement: The authors declare no conflict of interest for this article.
PRISMA 2009 Checklist statement: The manuscript was prepared and revised according to the PRISMA 2009 Checklist statement.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Goran Augustin, MD, MSc, PhD, Associate Professor, Doctor, Senior Scientist, Surgical Oncologist, Department of Surgery, University Hospital Centre Zagreb, Kišpatićeva 12, Zagreb 10000, Croatia. augustin.goran@gmail.com
Received: December 20, 2022 Peer-review started: December 20, 2022 First decision: February 8, 2023 Revised: February 14, 2023 Accepted: June 11, 2023 Article in press: June 11, 2023 Published online: August 27, 2023
ARTICLE HIGHLIGHTS
Research background
No systematic data on choledochal cysts (CC) in pregnancy exist.
Research motivation
Due to the rarity, no guidelines exist for the diagnostic workup and treatment and obstetric strategy for CC in pregnancy.
Research objectives
To collect the most published case reports on CC in pregnancy.
Research methods
Descriptive statistics of available patient and disease data.
Research results
Cholangitis, CC > 15 cm, and bilirubin levels > 80 mmol/L were associated with a higher likelihood of urgent cesarean section (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.
Research conclusions
Although rare, maternal CC in pregnancy should be included in the evaluation of jaundice with upper abdominal pain. Symptomatology and clinical course are variable. 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.
Research perspectives
All cases of CC in pregnancy should be published to understand better the epidemiology, etiology, specific diagnostic imaging methods, and treatment strategy of this extremely rare condition.
