Skip to main content

Spontaneous perforation of common hepatic duct: an intraoperative surprise

Abstract

Background

Pediatric spontaneous bile duct perforation is one of the rare causes of acute abdomen in infants. With a highly variable presentation, diagnosis and treatment can prove challenging. We report a case of spontaneous common hepatic duct perforation mistaken for appendicular peritonitis.

Case presentation

An otherwise healthy 3-year-old boy presented to the emergency department with complaints of abdominal pain, distention, bilious vomiting, and fever evolving for 4 days. Preoperative ultrasound was suggestive of appendicular peritonitis. Laparotomy revealed abundant biliary fluid, a distended gallbladder with thickened and inflammatory wall, and a macroscopically normal appendix. Intraoperative cholangiography showed a leak of contrast from the anterior wall of the common hepatic duct, 2–3 mm below the upper biliary confluence and 2 cm above the junction of the cystic duct to the common hepatic duct. A cholecystostomy was performed, and two external intraabdominal drains were placed. A retrograde cholangiogram was performed on postoperative day 21 showing no extravasation of contrast product. The patient was discharged on postoperative day 25.

Conclusion

Spontaneous perforation of the common hepatic duct is a rare phenomenon. It should be considered as a differential diagnosis in pediatric patients that present with unexplained peritonitis. The optimal method of diagnosis and management remains controversial.

Background

Spontaneous common bile duct perforation is an unusual cause of acute abdomen in children, most often diagnosed at the stage of biliary peritonitis [1]. It is potentially fatal, and preoperative diagnosis is very difficult since the clinical presentation is non-specific [1]. The most common presentation is abdominal distension and jaundice. Diagnosis is often made during surgery. Although the actual etiopathogenesis of this uncommon disease has yet to be determined, several hypotheses have been suggested. Mortality decreased from 40 to 12% as a result of improved care. However, in the lack of a clear consensus, surgical management remains variable [2, 3].

Herein, we report a rare case of spontaneous common bile duct perforation in a 3-year-old boy mistaken for appendicular peritonitis. Through our case, we detailed our surgical management and we reviewed the literature on the surgical options for this rare condition.

Case presentation

An otherwise healthy 3-year-old boy presented to the emergency department with complaints of diffuse abdominal pain, distention, bilious vomiting, and fever evolving for 4 days. On examination, the patient was febrile, dehydrated, and hemodynamically stable. Abdominal examination revealed abdominal distension with mild tenderness. Abdominal ultrasound was suggestive of appendicular peritonitis. It showed periappendiceal fat stranding, cecal apical wall thickening, and echogenic intraperitoneal effusion of great abundance without visualization of the appendix. A midline laparotomy was performed revealing an abundant bilious effusion, a distended gallbladder without any calculus and perforations, and a macroscopically normal appendix. Intraoperative cholangiography through the gallbladder showed a leak of contrast from the anterior wall of the common hepatic duct, 2–3 mm below the upper biliary confluence and 2 cm above the insertion of the cystic duct (Fig. 1).

Fig. 1
figure 1

Intraoperative cholangiography showing the leak of contrast from the anterior wall of common hepatic duct

Biliary diversion with a cholecystostomy tube was performed. A drain was placed under the liver and a wavy blade at the level of the right iliac fossa. The postoperative course was uneventful. A retrograde cholangiogram was performed on postoperative day 21 showing no extravasation of contrast product (Fig. 2). Enteral feeding was introduced on day 15 post-operatively and was well tolerated. The cholecystostomy tube was removed after the cholangiography. The patient was discharged on postoperative day 25. He is doing well on follow-up.

Fig. 2
figure 2

Postoperative retrograde cholangiogram showing a permeable normal-sized bile duct with no extravasation of contrast product

Discussion

Spontaneous or idiopathic bile duct perforation is a rare entity, mostly seen in children less than 4 years of age [1]. The actual etiopathogenesis is unknown and remains obscure. Congenital weakness of the common hepatic duct, trauma, choledochal cyst, viral infection, intramural thrombosis, necrotizing enterocolitis, pancreatitis, diverticulum in the common hepatic duct, stenosis of the common hepatic duct, and anomalous union of the pancreatico-biliary ductal system are all believed to be involved [2]. Usually, the site of weakness is at the junction of the cystic duct and the common hepatic duct. The site of the perforation at the common hepatic duct is exceptional and has been reported in few adult cases [3]. To our knowledge, this is the second pediatric case which reported such location [3].

As for our patient, the preoperative diagnosis is extremely rare given the absence of specific signs. Most common clinical presentations are abdominal distension, jaundice, symptoms of peritonitis, septicemia, and symptoms of biliary tract disease [3]. Abdominal ultrasound visualizes the defect in 38.4% of cases. An abdominal CT scan helps to make the diagnosis in 69.2% of cases [4].

Controversy exists regarding the optimal surgical modality. Most of the authors use T-tube drainage placed through the bile duct and a subhepatic drain [3, 58]. T-tube drainage ablation is usually done 3 weeks post-operatively. Other techniques are described such as biliary diversion using a cholecystostomy tube with or without placing a Kehr drain [1, 3, 914], surgical repair of the common hepatic duct, and cholecystectomy with intraabdominal drainage [12]. Insertion of a metal fully covered stent with the tip beyond the leak [15] and hepaticojejunostomy by means of a Roux-en-Y loop were also described as surgical modalities [8,9,10, 14, 16,17,18]. Table 1 summarizes the different surgical approaches used in the treatment of common bile duct perforations described in the literature. The particularity of our patient is that we opted for a non-invasive treatment using a single biliary diversion with cholecystostomy tube and drainage of the peritoneal cavity. The cholecystostomy drains the bile, thus reducing the pressure at the level of the common hepatic duct, and facilitates its cicatrization to healing.

Table 1 Literature review of surgical management of common bile duct perforations in children

Conclusion

Spontaneous perforation of the common hepatic duct is rare. The optimal method of diagnosis and management remains controversial. Intraoperative cholangiography is essential to locate the perforation. Several therapeutic methods are used. Biliary diversion via a cholecystostomy tube is simple and safe and proves to be an effective method of treatment.

Availability of data and materials

Not applicable.

References

  1. Jeanty C, Derderian SC, Hirose S, et al. Spontaneous biliary perforation in infancy: management strategies and outcomes. J Pediatr Surg. 2015;50:1137–41.

    Article  Google Scholar 

  2. Evans K, Marsden N, Desai A. Spontaneous perforation of the bile duct in infancy and childhood: a systematic review. J Pediatr Gastroenterol Nutr. 2010;50:677–81.

    Article  Google Scholar 

  3. Sunil K, Gupta A, Verma AK, et al. Spontaneous common hepatic duct perforation in a child: a rare case report. Afr J Paediatr Surg. 2018;15(1):53–5.

    Article  Google Scholar 

  4. Sood B, Jain M, Khandelwal N, et al. MRI of perforated gall bladder. Australas Radiol. 2002;46:438–40.

    Article  Google Scholar 

  5. Goel P, Jain V, Manchanda V, et al. Spontaneous biliary perforations: an uncommon yet important entity in children. J Clin Diagn Res. 2013;7(6):1201–6.

    PubMed  PubMed Central  Google Scholar 

  6. Ozdemir T, Akgül AK. Arpaz yet al. Spontaneous bile duct perforation: a rare cause of acute abdominal pain during childhood. Ulus Travma Acil Cerrahi Derg. 2008;14(3):211–5.

    PubMed  Google Scholar 

  7. Haller JO, Condon VR, Berdon WE, et al. Spontaneous perforation of the common bile duct in children. Radiology. 1989;172(3):621–4.

    CAS  Article  Google Scholar 

  8. Upadhyaya VD, Kumar B, et al. Spontaneous biliary peritonitis: is bed side diagnosis possible? Afr J Ped Surg. 2013;10(2):112–7.

    Article  Google Scholar 

  9. Chardot C, Iskandarani F, De Dreuzy O, et al. Spontaneous perforation of the biliary tract in infancy: a series of 11 cases. Eur J Pediatr Surg. 1996;6:341–6.

    CAS  Article  Google Scholar 

  10. Sai Prasad TR, Chui CH, Low Y, et al. Bile duct perforation in children: is it truly spontaneous? Ann Acad Med Singap. 2006;35:905–8.

    CAS  PubMed  Google Scholar 

  11. Fukuzawa H, Urushihara N, Miyakoshi C, et al. Clinical features and risk factors of bile duct perforation associated with pediatric congenital biliary dilatation. Pediatr Surg Int. 2018;34(10):1079–86.

    Article  Google Scholar 

  12. Xanthakos SA, Yazigi NA, Ryckman FC, et al. Spontaneous perforation of the bile duct in infancy: a rare but important cause of irritability and abdominal distension. J Pediatr Gastroenterol Nutr. 2003;36(2):287–91.

    Article  Google Scholar 

  13. Mirza B, Ijaz L, Saleem M, et al. Management of biliary perforation in children. Afr J Ped Surg. 2010;7(3):147–51.

    Article  Google Scholar 

  14. Kohli S, Singhal A, Arora A, et al. Spontaneous biliary peritonitis in children. J Clin Imaging Sci. 2013;3(2):1–4.

    Google Scholar 

  15. Sharma C, Desale J, Waghmare M, et al. A case of biliary peritonitis following spontaneous common bile duct perforation in a child. Euroasian J Hepatogastroenterol. 2016;6(2):167–9.

    PubMed  PubMed Central  Google Scholar 

  16. Howard ER, Johnston DI, Mowat AP. Spontaneous perforation of common bile duct in infants. Arch Dis Child. 1976;51(11):883–6.

    CAS  Article  Google Scholar 

  17. Lloyd DA, Mickel RE. Spontaneous perforation of the extra-hepatic bile ducts in neonates and infants. Br J Surg. 1980;67(9):621–3.

    CAS  Article  Google Scholar 

  18. Ford WD, Sen S, Morris L, LeQuesne G. Spontaneous perforation of the common bile duct in the neonate: imaging and treatment. Aust Paediatr J. 1988;24(5):306–8.

Download references

Acknowledgements

Not applicable.

Funding

No funding.

Author information

Authors and Affiliations

Authors

Contributions

KY drafted the manuscript. JR supervised the study. TF, ZA, and BA performed perioperative management of the patient. The authors read and approved the final manuscript.

Corresponding author

Correspondence to Y. Kerkeni.

Ethics declarations

Ethics approval and consent to participate

Not applicable.

Consent for publication

Written informed consent was obtained from the patient’s parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Competing interests

The authors declare that they have no competing interests.

Additional information

Publisher’s Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

Reprints and Permissions

About this article

Verify currency and authenticity via CrossMark

Cite this article

Kerkeni, Y., Thamri, F., Zouaoui, A. et al. Spontaneous perforation of common hepatic duct: an intraoperative surprise. Ann Pediatr Surg 18, 21 (2022). https://doi.org/10.1186/s43159-022-00159-2

Download citation

  • Received:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1186/s43159-022-00159-2

Keywords

  • Biliary tract
  • Common hepatic duct
  • Spontaneous perforation