Double cystic duct, a review of literature with report of a new case.

INTRODUCTION: Although cystic duct variation is quite common, duplication of cystic duct is an extreme rare variant. We report a case of double cystic duct with literature review. A 33-year old female presented with right upper quadrant pain of three day duration, associated with nausea and poor appetite. The patient reported previous three attacks of right upper quadrant pain within the last two years. On examination: Murphy's sign was positive and the right upper quadrant was tender. Abdominal ultrasound showed multiple gall stones. Oesophago-gastro- duodenoscopy was normal. Under general anesthesia, four port formal laparoscopy was done, double cystic duct was found. Histopathological examination showed features of chronic cholecystitis.
CONCLUSION: double cystic duct is a very rare variant of the cystic duct anomaly. Identification pre or intraoperatively is very important to prevent ductal injury.

Introduction

Although cystic duct variation is quite common, duplication of cystic duct is an extreme rare variant; only 16 cases have been reported in literature [1], [2], [3], [4], [5], [6], [7], [8], [9], [10], [11], [12], [13], [14], [15]. These variations increase risks of ductal injury, need for open conversion and postoperative complication [13]. The diagnosis is usually established during surgery [8]. However, it may be missed intraoperatively and diagnosed postoperatively during a diagnostic work up performed for persistent biliary symptoms [15].

In line with SCARE guide line, we report a case of a young female who had two separate cystic ducts exiting from a single gallbladder [16].

Patient information

A 33-year-old married female presented with right upper quadrant pain of three day duration. The pain started suddenly, sharp in character, awakening her from sleep, radiating to right shoulder and epigastric area, associated with nausea and poor appetite. The patient reported previous three attacks of right upper quadrant pain within the last two years that settled down within few days. Past surgical and medical histories were negative.

Clinical findings

the patient was in pain. Murphy's sign was positive and right upper quadrant was tender.

Diagnostic assessment

Vital signs, blood examination, electrocardiography and echocardiography were within normal range. Abdominal ultrasound showed multiple gall stones, largest one about 14 millimeters in diameter. Oesophago-gastro- duodenoscopy (OGD) was normal.

Therapeutic intervention

Under general anesthesia, four port formal laparoscopy was done, double cystic duct was found (Fig. 1), clipped, gall bladder removed, and there were multiple gall stones (Fig. 2). Gross examination of the specimen confirmed double cystic duct (DCD) (Fig. 3). Histopathological examination showed features of chronic cholecystitis.

Fig. 1

Intraoperative finding of double cystic duct. White arrows show the stumps of the first cystic duct. The blue arrow shows the second cystic duct prepared for division.

Fig. 2

Multiple stones evacuated from the gallbladder.

Fig. 3

Macroscopic examination of the specimen showing two separate opening in the gallbladder (the suture material).

Follow-up and outcomes

Two weeks later, the patient was stable and healthy.

Discussion

Presence of variations especially DCD during laparoscopic cholecystectomy is of great challenge for laparoscopic surgeons. Variation in the anatomy of cystic duct is not uncommon [7].

A classic anatomic picture between the extrahepatic bile duct, cystic duct, and related arteries is seen in only 33% of individuals. In 66% of the cases, the entry of the cystic duct into the common bile duct (CBD) is angular. In 17% to 20%, the CBD is parallel to the cystic duct and the entry is straighter. In 5% to 8%, the cystic duct is spiral and tortuous, connecting the CBD at a variable angle. In less than 1% of the patients, it drains into the right hepatic duct [8]. DCD is an extremely rare variant [16]. It is usually more common among female. From the reported cases, 11 cases (73%) occurred among female patients [3], [4], [5], [6], [7], [8], [9], [10], [11], [14], [15]. The age at the time of awareness of the variation varies ranging from new born to 76 years old [4], [11]. Vincente et al. reported a newborn baby with VACTERL association (vertebral defects, anal atresia, cardiac defects, tracheo-esophageal fistula, renal anomalies, and limb abnormalities) having DCD discovered during cholicystectomy for sympomatic cholelithiasis [11]. The current case was a young, married female. Otaibi et al. stated that DCD associated with double gallbladder in 80% of the cases [13]. From the 15 reported cases in the literature, five cases (33%) associated with double gallbladder [1], [2], [3], [12], [15]. We did not find accessory gallbladder, the two cystic ducts drained from the same gallbladder. Flannery and Caster classified DCD into three different categories according the configuration of the ducts. The “H type” is the most common variant in which the second cystic duct drains separately into either the left, right, or common hepatic duct (CHD). The current patient had the Y type variant. In “Y type” variant, both cystic ducts unite to form a common channel that drains into the common hepatic duct. The third type variant is the trabecular type in which the accessory duct drains directly into substance of liver [8], [14]. Preoperative diagnosis is difficult due to rarity of the variation and difficulty in visualization of the two ducts by radiological imaging [2], [3], [4], [11], [14]. Ultrasound of the current case failed to reveal DCD. Some authors prefer routine use of intraoperative cholangiography to differentiate bile duct injury that might occur either because of transaction of bile duct or because of the presence of anatomical variations [7], [14]. The limitation of this study is the absence of an intraoperative investigation like cholangiogram or post operative investigation like MRCP/ERCP to confirm the diagnosis. Table 1 shows literature review of the reported cases of DCD.

Table 1

Literature review of the reported cases of DCD.

Reference. Authors	Year of publication	Age. sex	diagnosis	Number of gallbladder	Presenting pathology
1. Heyas et al	1931	35. male	Preoperative CRE	double	Not mentioned
2. Kennon	1933	69. male	Second re-exploration	double	suppurativecholecystitis
3. Wilson	1939	55. female	intraoperative	double	Acute cholecystitis
4. Paraskevas et al	1989	76. female	Postmortem	single	Postmorterm educational examination
5. Nakasugi et al.	1995	50. female	Preoperative ERCP	single	Chronic cholecystitis
6. Hirono et al	1997	74. female	Preoperative US, CT-scan	single	Chronic cholecystitis
7. Tsutsumi et al	2000	74. female	Preoperative ERCP	single	Chronic cholecystitis
8. Shivhare et al	2002	46. female	intraoperative	single	Chronic cholecystitis
9. Huston et al.	2006	43. female	intraoperative	single	Chronic cholecystitis
10. Yoo et al.	2008	55. female	Preoperative MRI, PTBD	singe	CBD cancer
11. Vicente et al.	2009	newborn	Intraoperative	single	acute cholecystitis
12. Görkem et al.	2014	10. male	Preoperative US	double	acute cholecystitis
13. Otaibi et al.	2015	54. male	intraoperative	single	Chronic cholecystitis
14. samnani et al	2015	43. female	intraoperative	single	Chronic cholecystitis
15. Wei et al	2015	66. female	Preoperative US, CT-scan	double	Chronic cholecystitis

ERCP: endoscopic retrograde cholangiopancreatography; CRE: complete roentgen examination; MRI: magnetic resonance imaging; PTBD: percutaneous transhepatic biliary drainage; US: ultrasound; CT-scan: computed tomography scan; CBD: common bile duct.

In conclusion, DCD is a very rare variant of the cystic duct variation. Identification pre or intraoperatively is very important to prevent ductal injury.

Consent

Informed consent was taken from the patient for the publication of this report.

Author contribution

Abdulwahid M. Salih: Surgeon performed the operation and follow up. Final approval of the manuscript.

F. H. Kakamad: writing the manuscript, reviewing the literature and follow up with final approval of the manuscript.

Rawezh Q.S, Shvan H.M, Imad J. Habibullah, Aso S. Muhialdeen, Hewa fatih: major contribution to the idea, literature review and final approval of the manuscript.

Sources of funding

No source to be stated.

Ethical approval

Approval has been taken from bioscience centre.

Guarantor

Fahmi Hussein kakamad.

Conflicts of interest

There is no conflict to be declared.