Repeat Cholecystectomy. A Mummified Gallbladder was the Clue to Diagnose Duplicate Gallbladder: A Case Report and Review of Literature

Introduction

Gallbladder (GB) duplication is a congenital anomaly that is considered rare being present in about one in 4000 births. The first report of duplication of the gallbladder is credited to Blasius in 1675. GB Duplication has many forms including the true duplication form being two completely separated gallbladders or Y-shaped in which the two gallbladders are united with a single cystic duct prior to joining the common hepatic duct. Gallbladder duplication is suggested to be the result of exuberant budding of the developing biliary tree during the division of the caudal bud of the hepatic diverticulum [1]. Additionally, it may be associated with duplication of cystic duct. Thus, the anatomy should be precisely visualized during surgery to avoid injuries to other structures [2,3].

Gallstone is the commonest complication associated with duplicate gallbladder. Although it usually affects one lobe, both can be involved with equal risk for the disease. Hence, resection of both gallbladders is the rule [3,4].

Ultrasonography (US) is a very popular imaging modality for the assessment of gallbladder diseases. Its primary benefit comes from evaluating the gallbladder wall and contents. However, it cannot assess the anatomy of the biliary tree, while magnetic resonance cholangiopancreatography (MRCP) can. Therefore, it is considered the investigation of choice to visualize the biliary anatomy even when complicated by certain diseases [1,5-7].

The identification of duplicate gallbladder before attempting to perform cholecystectomy is crucial to prevent or at least decrease the rate of complications. Moreover, the gallbladder duplication is diagnosed preoperatively in only fewer than half of the cases, which can be attributed to the presence of many variations. For instance, some of them were found to be more difficult to be detected when the duplicate organ is positioned remotely or deeper in the viscera, e.g. above the right or rarely the left hepatic artery [4]. Thus, appropriate diagnosis is essential to avoid recurrence of the symptoms, and repetition of the procedure, with a risk of special surgical complications mainly due to distorted biliary anatomy [1,5,8,9].

Methods

Case Report

We report a case of cholecystitis after open cholecystectomy in a private center, according to SCARE criteria [10].

Systematic Review

We performed a systematic review and meta-analysis according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. (Supplementary table 1). The literature review was performed in PubMed and SCOPUS in October 2022 using the following search term (((repeat cholecystectomy) OR (second cholecystectomy)) AND ((double gallbladder) OR (duplicate gallbladder) OR (gallbladder duplication) OR (multiple gallbladders))). Then, title and abstract screening followed by full-text screening was performed by two independent reviewers according to our selection criteria. We included all case reports that reported repeat cholecystectomy without any restrictions to language, age, or gender. Any reviews, letters, or abstract only articles were excluded. Afterwards, the data of the included articles were extracted using a standardized excel sheet by two independent reviewers. All conflicts were solved through discussion and the senior author was consulted to reach a consensus. The extracted data was analyzed qualitatively and summarized in tables. Risk of bias was performed using JBI (Joanna Briggs Institute) critical appraisal tool for case reports [11]. The quality of the studies was divided into good, fair, or poor according to the score. In detail, the quality was considered good if it scored (6-8). On the other hand, a poor quality was considered if the study got (0-2).

Results

Case Report

Patient Information

A 34-year-old male patient presented to our private center with recurrent episodes of severe right hypochondrium pain, radiating to the right shoulder, lasting for 20 to 40 minutes. Additionally, the patient complained from epigastric pain which suggested cholecystitis [12].

The patient was not diabetic or hypertensive and didn’t suffer from any chromic or acute comorbidities. He had a history of mini-laparotomy cholecystectomy, which was done by the same consultant; Dr. Madany MEDM 2 years and 7 months ago. Also, the patient reported administration of symptomatic treatment such as proton pump inhibitor for epigastric pain.

On examination the patient was vitally stable, and the scare of the previous operation was observed through inspection. On palpation, there was mild tenderness in the right hypochondrium and epigastric region. The laboratory results including CBC, Bilirubin, AST, and ALT were within the normal range. An US report, which was performed before visiting our center, revealed the presence of non-calcular cholecystitis. Reviewing his records revealed that the previous cholecystectomy passed without any complications. Moreover, his mother had brought the specimen of the removed GB in the same piece of gauze she received! (Figure 1). On gross examination, it was complete GB, measured 4x2 cm2, with clearly ligated cystic duct with Vicryl suture. Abdominal US was repeated and revealed GB-like structure containing small stones in the GB fossa, followed by MRCP to confirm the diagnosis. Our proposed differential diagnoses were 1) Dilated cystic duct containing stones, 2) GB remnant, 3) Duplication of GB, and 4) Choledochal cyst.

Medical treatment was started immediately, and the patient was followed up monthly. There was no detected improvement in the symptoms for 20 months till the patient agreed to laparoscopic intervention. Intra-operative diagnosis of duplicate GB was established, and laparoscopic cholecystectomy of the 2nd gall bladder was performed after extensive meticulous adhesiolysis especially in the presence of a caterpillar hump of the right hepatic artery (Figure 3).

Surgical Technique

Under general anesthesia a classic laparoscopic cholecystectomy was performed. We inserted a 5 mm port in the upper margin of the umbilicus for the 5 mm 30° scope. During exploration, there was a minimal adhesion to the right of the falciform ligament. The sub-xiphoid and the subcostal midclavicular line 5 mm ports were inserted.

Thereafter, we found an accessory Riedel’s lobe in the liver situated to the left of the gallbladder fossa, where extensive adhesions of the previous surgery were found. Thus, a meticulous adhesiolysis was performed until we surprisingly found a missed gallbladder. Finally, we inserted a fourth 5 mm port in the lateral subcostal port.

We caught the fundus of the gallbladder and used it for retraction. Then, we continued meticulous adhesiolysis using monopolar diathermy and hydro dissection until we reached the Calot triangle, where a caterpillar hump of the right hepatic artery was found (Figure 3). Using 2/0 vicryl, we performed two sutures to ligate the cystic duct, then we divided the cystic duct.

The previous gallbladder had a clear print on the liver bed, we carefully continued the dissection of the gallbladder from its surrounding adhesions with concerns to the caterpillar hump. We found the second gallbladder situated to the left of the previous gallbladder and above the caterpillar hump.

We removed the second gallbladder and explored the hilum of the liver where we found a tubular structure which was assumed to be the cystic duct of the previously excised gallbladder. Therefore, we can describe that both cystic ducts were situated distal to the caterpillar hump of the right hepatic artery. Finally, we retrieved the gallbladder through the 5 mm lateral subcostal port.

After the laparoscopic cholecystectomy, the patient had a smooth post-operative period without any complications.

Histopathology

Histopathologic examination of both gallbladders; the mummified and the recent one confirmed the diagnosis of duplicate gallbladder.

Gross Picture

1. The 1st gall bladder: A mummified gall bladder measured 4x2 cm, with dark outer surface and intraluminal stone mud.

2. The recently removed cholecystectomy specimen: Gall bladder measured 3x2 cm, with smooth outer surface, greenish velvety mucosa and average wall thickness 0.3 cm. Lumen was distended with bile.

Microscopic Picture

1. Sections examined from the 1st gall bladder revealed dense fibrosis and hyalinosis of the wall without definite mucosal lining and with remnants of stone.

2. Sections examined from the recent gall bladder:

a. Revealed partially ulcerated mucosa.

b. The wall was infiltrated by lymph-plasma cells and showed fibrosis.

There was no evidence of malignancy in the received specimens.

Histopathologic Diagnosis

The histopathologic report concluded a gall bladder duplication (congenital anomaly), with chronic cholecystitis in both of them, with no malignancy.

Systematic Review

The database search revealed 238 articles of which only 20 articles were eligible according to our selection criteria. (Figure 1)

The demographic and characteristics of included studies were summarized in Table 1. The age ranged from 20 to 75 years. There were 12 female and 9 male cases, and a case was undefined.

Laparoscopic cholecystectomy was performed to remove the first gall bladder in 15 cases of whom only one converted to open surgery. While 6 patients undergone open cholecystectomy and only one case performed Laparoscopic Endoscopic Cooperative Surgery (LEC). On the other hand, the number of patients undergone open cholecystectomy for the 2nd operation was 11, while 9 patients undergone laparoscopic cholecystectomy. The remaining two patients had either LEC or unreported. The time between the 1st and the 2nd operation ranged from 5 days to 40 years. The rate of shifting from open in the 1st operation to laparoscopic in the 2nd operation was 2 of 22 cases, and the rate of shifting form laparoscopic to open was 7 of 22 cases.

Although, the type of 1st operation was laparoscopic cholecystectomy in 15 cases, we suggest that the type of operation isn’t the cause of the repeat cholecystectomy, as the 1st case reported in the literature was Deans & Brough, 1994, which was after the beginning laparoscopic era, so the surgeons started to rely on laparoscopic more than open in cholecystectomy [13]. Moreover, the availability of some investigations like MRCP and ERCP facilitate the diagnosis of missed GB in addition to the development of new generations of ultrasonographic devices with a better resolutions and efficacy. Also, laparoscopic assessment encourages surgeons to evaluate these patients more than open surgery.

The radiological investigations available for the diagnosis of missed gallbladder after cholecystectomy were US, X-ray, CT, MRI, ERCP, MRCP, intraoperative cholangiogram, HIDA scan, barium follow through and CT cholangiogram. The most used investigation was US in 21 cases, MRCP in 8 cases, ERCP in 7 cases and CT in 7 cases. The proposed differential diagnoses (DD) for these cases were as follow: 1) a remnant infundibulum from subtotal cholecystectomy, 2) a pseudocyst of the common bile duct, 3) a choledochal cyst, 4) a recurrent internal hernia with postoperative changes on imaging, 5) a duplicate gall-bladder, 6) acute pancreatitis, 7) bile duct diverticulum, 8) free fluid or abscess formation, 9) gallbladder diverticulum, 10) focal adenomyomatosis, 11) Phrygian cap, 12) walled off collection as a result of granulomatous reaction to dropped gallstones, 13) bilobed or duplex gallbladder with one half having been removed at previous surgery, 14) incomplete cholecystectomy or a collection in the gallbladder bed, and 15) remnant GB. The risk of bias was assessed in the included articles which yielded a good score in 18 studies, fair score in two studies and no article got bad score. The detailed assessment is shown in supplementary table 1.

Figure 1: Prisma Flow Diagram Showing the Search Results and Screening Process of Included Articles

Figure 2: The Specimen of the Removed GB in the Same Piece of Gauze she Received 2 Years and 7 Months Ago in A Mummified State!

Figure 3: MRCP Showing the Biliary Tree and Structure Like Gall Bladder

Figure 4: Laparoscopic Image Showing 1) Caterpillar Artery and 2) Cystic Duct

Figure 5: Timeline of the Case Events

Discussion

Gallbladder duplication is one of the rarest congenital anomalies of the hepatobiliary system accounting for 1:4000 births. However, this ratio reports only symptomatic cases and cases discovered incidentally during surgery, imaging modalities and autopsy. Hence, the true incidence of this anomaly is not precisely known. In addition, the usual initial preoperative imaging modalities used in cases of cholelithiasis, and cholecystitis often miss the detection leading to increased risk of intraoperative iatrogenic complications. Incidental detection of double gall bladder during surgery requires intraoperative cholangiography with meticulous delineation of biliary tree [8,14-17].

According to Desolneux et al., the incidence of gall bladder disorders is the same in both single and duplicated gall bladder.

Meanwhile, Pillay study reported that the inadequate bile drainage, which is usually present in this anomaly, increases the risk of cholelithiasis [18,19].

Asymptomatic gallbladder duplication does not require surgical intervention. However, when surgery is decided, removal of both is crucial once become symptomatic [20].

In our case, when patient presented to us 4 years ago, the diagnosis of duplicate gallbladder was missed preoperatively with ultrasound and intraoperatively during open surgery which could be justified by that this duplicate gall bladder was type II and it was remote and deeply situated in viscera above the right hepatic artery. Also, the mini-laparotomy approach may be a factor. The second time the patient presented to our clinic with similar complaint of biliary colic and after appropriate history taking, physical examination, revision of patient’s profile recorded in our database and lastly the mummified gall bladder kept with the patient’s mother, we decided to perform an abdominal ultrasound followed by MRCP to finally conclude intraoperatively that this is a case of duplicate gallbladder [1,5,8].

The operative data in the reported cases was limited, so we can’t identify a risk factor for missing a gall bladder e.g., the use of an angled lens (30 degrees) or Zero-degree lens.

According to our knowledge, this is the 1st article to report duplicate GB removal by the same surgeon, the 1st was mini laparotomy, and the 2nd was lap. Chole in addition to the presence of the old and the new GB at time of pathological examination. Also, the presence of an isolated mummified GB that belongs to a living person for 4 years and 3 months, is reported in literature for the first time.

Conclusion

This study suggests including duplicate gallbladder as one of the causes of post cholecystectomy syndrome. Moreover, it highlights that duplicate GB can be missed preoperatively, and operatively even with a senior experienced consultant. Preoperative ultrasound can often miss the diagnosis of gallbladder duplication. Additionally, the lack of performing a careful and meticulous dissection of biliary system will likely lead to missing the diagnosis. MRCP is the investigation of choice for proper identification of the anatomy of the biliary system.

                                                         Table 1: Baseline Characteristics of Similar Case Reports

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