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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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Minagawa T, Dowaki S, Kikunaga H, Fujita K, Ishikawa K, Mori K, Sakuragawa T, Ichisaka S, Miura H, Kumai K, Mikami S, Kitagawa Y. Endoscopic biliary drainage as a bridging procedure to single-stage surgery for perforated choledochal cyst: a case report and review of the literature. Surg Case Rep 2015; 1:117. [PMID: 26943441 PMCID: PMC4648837 DOI: 10.1186/s40792-015-0115-4] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/16/2015] [Accepted: 10/28/2015] [Indexed: 02/07/2023] Open
Abstract
Choledochal cyst (CC)—a congenital anomaly of the bile duct—is rare. We report a 28-year-old woman complaining of epigastralgia who was transferred to our hospital. Physical examination revealed severe tenderness to abdominal palpation without symptoms of diffuse peritonitis. Urgent contrast-enhanced abdominal computed tomography indicated the dilated common bile duct (CBD) was perforated, with a presumed diagnosis of perforated CC. Endoscopic external biliary drainage was performed immediately as a bridging procedure to the definitive surgery. Additional evaluations confirmed a type IVa CC, according to Todani’s classification, but no signs of malignancy. Twenty-two days after biliary drainage, laparotomy was performed. A large cystic mass was found in the CBD with a perforated scar on the right-side wall. Because inflammation around the pancreas head was too severe to perform cyst excision safely, the patient underwent subtotal stomach-preserving pancreatoduodenectomy. The postoperative course was uneventful, and the patient was discharged on the 29th postoperative day. Pathologic examination of a specimen showed no malignancy, and the patient has remained well during the 3-year follow-up. Our experience with this case suggests that definitive single-stage surgery for perforated CC in an adult can be performed safely owing to external biliary drainage as a bridging procedure, if manifestation of diffuse peritonitis is not evident.
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Affiliation(s)
- Takuya Minagawa
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan.,Department of Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan
| | - Shoichi Dowaki
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan.
| | - Hiroyuki Kikunaga
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Koji Fujita
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Keiichi Ishikawa
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Katsuaki Mori
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Tadayuki Sakuragawa
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Shunsuke Ichisaka
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Hiroshi Miura
- Department of Radiology, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Koichiro Kumai
- Department of Surgery, Hino Municipal Hospital, 4-3-1 Tamadaira, Hino, Tokyo, 191-0062, Japan
| | - Shuji Mikami
- Division of Diagnostic Pathology, Keio University Hospital, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan
| | - Yuko Kitagawa
- Department of Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan
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Katabathina VS, Kapalczynski W, Dasyam AK, Anaya-Baez V, Menias CO. Adult choledochal cysts: current update on classification, pathogenesis, and cross-sectional imaging findings. ACTA ACUST UNITED AC 2015; 40:1971-81. [DOI: 10.1007/s00261-014-0344-1] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
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Abstract
Choledochal cyst, usually recognized as a problem in infants, is increasingly reported in adult patients. The aim of this study was to share our experience in treating adult patients with choledochal cysts. Two hundred twenty-one adult patients with choledochal cysts who were treated in our hospital from January 2002 to December 2010 were enrolled. The clinical data were retrospectively collected and analyzed. There were 168 Type I, three Type II, three Type III, 26 Type IV, and 21 Type V cysts. The presentations were nonspecific with 177 patients having abdominal pain and a few patients having other rare symptoms including back pain, cirrhosis, occupying lesions, or spontaneous rupture. The cysts were completely removed in 169 patients, and 36 patients required liver resection. There was one death resulting from abdominal bleeding. Morbidity was limited to 27 patients, 11 of whom underwent invasive interventions. Choledochal cysts remain an interesting clinical problem. In some adult cases, the diagnosis is very difficult. Treatment is type-dependent, complete removal of the cysts is widely accepted, and lifelong follow-up for these patients is recommended.
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Affiliation(s)
- Lei Gong
- Department of General Surgery, Beijing Tsinghua Hospital, Tsinghua University, Beijing, China
| | - Qiang Qu
- Department of Hepatobiliary Surgery, China PLA General Hospital, Beijing, China
| | - Xin Xiang
- Department of Hepatobiliary Surgery, China PLA General Hospital, Beijing, China
| | - Jing Wang
- Department of Hepatobiliary Surgery, China PLA General Hospital, Beijing, China
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