Gallbladder torsion: A Systematic Review of the Recent Literature

Introduction

Gallbladder torsion (GBT) is a rare disorder that manifests as an acute surgical abdomen and has the potential to incur significant morbidity and mortality in case of delay in recognition and management 1-2. It was described for the first time in 1898 by Wendel and since then only about 600 cases have been reported in peer reviewed English literature 3.

GBT is characterized by a mechanical organo-axial twist along the longitudinal axis of the gallbladder involving cystic artery and cystic duct. Its exact aetiology of GBT remains unknown, although certain anatomical variants are thought to predispose GB to torsion. There are five recognized congenital variations in relationship of the GB with respect to the liver as depicted in Figure 1.

Figure 1: Congenital variations in anatomical relationship of the gallbladder (GB) with respect to the liver (Image credits: Sajad Ahmad Salati – author).

In the types 4 and 5, the GB hangs free, floating in the peritoneal cavity and has the predisposition to volvulus around the cystic pedicle in presence of inciting events like sudden changes in body position , blunt trauma , peristalsis in underlying viscera , and fatty meals leading to increased release of cholecystokinin and the resultant GB peristalsis 4-5 .Loss of visceral fat and elasticity with aging or after bariatric surgical procedures also tend to result in an elongated GB mesentery and predisposition for GBT.

GBT can be clockwise or anticlockwise and termed complete if the torsion is greater than 180 degrees and incomplete /partial if torsion is lesser than that 6. Women in their seventh and eighth decade are more likely to be affected than the men (3-4:1), according to published case reports; however, in the pediatric age range, it is more common in boys than in girls (4:1). The diagnosis usually comes as a surprise intraoperatively, and correct preoperative diagnosis is reached in less than 10% of patients 1.

In recent times, improved investigations and imaging tools have become available which are increasingly used in emergency settings for diagnosis of patients with acute abdomen. Hence, against that backdrop, this article was compiled to review the case reports of gallbladder torsion published after 2020, and present an up-to-date summary with respect to demographics, clinical presentation, imaging, and management and thereby build on existing knowledge related to it. The ultimate objective to enhance the physician’s awareness about GBT and thereby improve the ability to attain its timely diagnosis.

Materials and methods

Methods

A systematic review was performed according to PRISMA (preferred reporting items for systematic reviews and meta-analyses) guidelines. A literature search was performed using the PubMed, Google Scholar and PsycInfo databases, with ‘gallbladder’, ‘torsion’ and ‘volvulus’ as individual keywords with a combination of Boolean logic (AND). Further case reports were then compiled from the references of the initial articles.

Only the articles published in English language were considered for inclusion in this review and the time frame fixed was 2020–2024.

Criteria for considering studies

Articles, including case series and case reports, that provided an account of the selected variables were included in the review process.

Participants and Outcome measures

Only the articles where the diagnosis of GBT had been established on imaging or surgical exploration were included. The variables reported and analyzed include: [i] age; [ii] gender; [iii] clinical features; [iv] time between onset of symptoms and reporting to healthcare facility; [v] co-morbidities; [vi] findings on imaging; [vii] provisional diagnosis before definitive treatment ; [viii] delay between onset of symptoms and definite treatment ; [ix] treatment ; [x] operative findings; [xi] histopathology of retrieved specimen , and [xii] postoperative course.

Exclusion

The original articles, and systematic reviews that provided no information regarding the variables were excluded. The articles in languages other than English were also excluded.

Methodological quality checking

Checklist items from PRISMA and previously published systematic reviews were used for comparison with the checklist that was self-drafted for this study.

Data Synthesis (extraction and analysis)

Data related to the variables was derived and arranged in the form of Tables 1 & 2. The collected data was then analyzed with Statistical Package for Social Sciences (IBM-SPSS version 27.0) and Microsoft Excel (Office Version 16). The information was presented using frequencies, summary measures, tables, and figures as shown in the results.

RESULTS

Study selection

As seen in Figure 2, the electronic database search yielded a total of 114 publications. Sixty-three duplicate articles were eliminated and then titles and abstracts of 51 articles were screened. The full text of 4 articles was not in the English language, leaving 47 articles that met the qualifying requirements. After further exclusion of 3 articles due to unavailability and one case series that had details of cases before 2020, the review ultimately analyzed 43 articles.

Figure 2: Flowchart of the reviewed articles.

Study characteristics: Study characteristics are presented in Tables 1 to 4.

Table 1: Study characteristics of the patients of gallbladder torsion (demographics, clinical presentation, & preoperative diagnosis).

Table 2: Study characteristics of the patients of gallbladder torsion (imaging studies).

Table 3: Study characteristics of the patients of gallbladder torsion (management).

Table 4: Study characteristics of the patients of gallbladder torsion (histopathology and postoperative course).

There were 43 included case reports with 44 cases (n = 36, 83.3% females, and n = 8, 16.7% males), ranging in age from 01 to 94 years (mean 65 ± 31.3 years). There were 38 adults (33 females, 5 males) and 6 (3 females, 3 males) children, and hence, the ratio of females to males was 6.6:1 and 1:1 in the adults and children, respectively. As depicted in Figure 3, there was an early peak comprising 6 (13.6%) pediatric cases aged below 10 years, and there were 32 (72.7%) cases aged above 60 years.

Figure 3: Age distribution of the patients of gallbladder torsion.

Abdominal pain was the most prominent symptom in 43 (97.7%) cases, and one (2.3%) patient had chest pain. Nausea and vomiting was experienced by 21 (47.7%) cases. Abdominal tenderness of various degrees was found in all (100%) cases. Murphy’s sign of cholecystitis was positive in 10 (22.7%) cases, and an abdominal lump was palpable in 9 (20.4%) cases. There were significant comorbidities (hypertension, ischemic heart disease, chronic obstructive pulmonary disease, hiatus hernia) or a history of previous abdominal operations in 18 (40.9%) cases. One patient was pregnant (34 weeks).

Time from onset of symptoms to presentation at the healthcare facility was unknown in 2 cases. The majority (n=36; 81.8%) presented within 72 hours, and 25 (56.8%) cases presented within 24 hours, as depicted in Figure 4.

Figure 4: Time elapsed between the onset of symptoms and reporting to the healthcare facility .

Ultrasound of the abdomen was the imaging modality performed in 37 (84.1%) cases, and in 30 (68. %), USG was followed with a CT scan. In 6 (13.6%) cases, CT was undertaken as the first imaging modality without USG. Furthermore, MRCP (n = 6.8%), MRI with MRCP (n = 2; 4.5%), HIDA (n = 2; 4.5%), HIDA with MRCP (n = 2; 4.5%), and X-ray abdomen (n = 1; 2.3%) were other imaging tools used for diagnosis. In 1 (2.3%), no imaging was undertaken due to non-availability. After clinical assessment and imaging, a provisional diagnosis of GBT was made in only 18 (40.1%) as depicted in Figure 5, and 26 (59.9%) were misdiagnosed as AAC (n=43.2%), ACC (n=4; 11.4%), non-specific acute abdomen (n=1; 2.3%), and appendicitis (n=1; 2.3%).

Figure 5: Preoperative diagnosis of the patients .

Time delay from onset of symptoms till definitive management was not clearly mentioned in 8 (18.2%), and in the remaining 36 cases, the mean delay was 84 hours. Twenty (45.4%) cases were managed within 48 hours, 13 (29.5%) cases between 2-5 days, and only 3 (6.8%) cases beyond 5 days. The treatment was surgical in 43 (97.7%) and non-surgical in 1 (2.3%). The approach to surgical treatment was laparoscopic in 32 (72.7%), including one robot-assisted, and open/laparotomy in 11 (25%). The operations involved detorsion of GB followed with cholecystectomy (n= 39, 90.7%), percutaneous decompression of GB followed with cholecystectomy (n= 1, 2.3%), cholecystectomy (n= 2, 4.7%), and partial cholecystectomy with staplers (n= 1, 2.3%). In the lone pregnant patient, a caesarean section was conducted along with detorsion of GB and cholecystectomy. The non-surgical management adopted in one patient was imaging-guided percutaneous drainage of an abscess and antibiotics.

In 37 (84.1%) cases, features of gangrene were visible on surgical exploration. These changes were confirmed on histopathological analysis of the retrieved specimen, which revealed features of ischemic necrosis with accompanying hemorrhage and inflammation. In none of the specimens was there any evidence of dysplasia or malignancy. The direction of torsion was mentioned in 31 (70.5%) cases, and it was counterclockwise in 20 (45.4%) and clockwise in 11 (25%). The magnitude of torsion was mentioned in 27 (61.3%) cases, and it ranged from 180 to 720 degrees, being 180 in 8 (18.2%), 270 in 4 (9.1%), 360 in 12 (27.7%), and 720 degrees in 3 (6.8%) cases.

The postoperative phase was uneventful without any significant complications in 41 (93.2%) cases. One patient suffered from hospital-induced delirium, and in another, there was a stroke (left parietal infarct), leading to an extended in-hospital stay.

Discussion

Only 44 documented cases of GBT from 2020–24 were found in the current study, confirming the disorder's rarity. In adults, the female to male ratio was 6.6:1, while in children, it was 1:1. This contrasts with other findings in the literature, which show that the female to male ratio is 3–4:1 for adults and 1: 4 for children 1. As shown in Figure 2, 32 (72.7%) of the cases were over 60, 25 (56.7%) were in their ninth or tenth decade of life, and 18 (40.9%) had substantial comorbidities or a history of prior abdominal surgeries. This agrees with the literature that links this disorder to advancing age and significant comorbidities, therefore it's critical to get the right diagnosis as soon as possible. In the systematic review of 324 patients of GBT by Reilly et al. 1, the median age was 77 years.

Abdominal pain with abdominal tenderness (with or without rebound tenderness) were clinical features in 43 cases (97.7%) cases whereas nausea and vomiting was experienced by 21 (47.7%) cases. Murphy’s sign of cholecystitis was positive in 10 (22.7%) cases and an abdominal lump was palpable in 9 (20.4%) cases. A clear clinical pattern that can be characterized as three triads (triad of triads ), as shown in Figure 6, emerged when Lau et al. reviewed the clinical features of GBT reported in the literature. They reaffirmed that if the majority, but not necessarily all, of these features in three triads are present, a clinical suspicion or diagnosis of GBT should be raised.

Figure 6: Lau’s ‘triad of triads’ for clinical features related to gallbladder torsion.

In the current review, an 87-year- old female patient reported by Muenyi et al. 29 had chest pain as the presentation of GBT and a 94-year-old male patient reported by Zillman et al. 5 had chest pain in addition to abdomen pain. These cases demonstrate that GBT can present with sudden onset of chest pain, and hence GBT must be borne in mind as a differential diagnosis as was suggested by Ferguson and Bhalerao 51 when they reported an 88-year-old female patient who had presented with severe central chest pain and in whom, the diagnosis was made intraoperatively on laparotomy.

Most cases (n=36; 81.8%) reported to the healthcare facility within 72 hours of the onset of symptoms, and 25 (56.8%) cases presented within 24 hours, as depicted in Figure 3. Time delay (mean) from the onset of symptoms till definitive management mentioned in 36 (81.8%) was 84 hours. This delay can be attributed to the fact that despite clinical assessment and imaging, GBT was not suspected, and a correct diagnosis was made in only 18 (40.1%) as depicted in Figure 4, leading erroneously to the adoption of a conservative line of action. This figure concurs with the data in the previous literature 1. Reilly et al. 1 in 2012 conducted an extensive review of a total of the case reports of GBT published between 1991 and 2011. They found that a correct preoperative diagnosis was made in only 32 of 125 (26%) patients. Nakao et al. 52 in 1999 reviewed 245 cases reported in the Japanese literature and found that only 9.8% of all GBT cases (pediatric and adult) could be diagnosed preoperatively.

During evaluation, imaging was undertaken in 43 cases (97.7%), and only in 1 (2.3%) patient, surgical exploration was undertaken without any imaging due to its non-availability. Ultrasound of the abdomen was the primary imaging undertaken in 37 (84.1%) cases, whereas in 6 (13.6%) cases, CT was undertaken as the first imaging. In 30 (68. %), USG was followed with a CT scan of the abdomen. In 6 (13.6%) cases, MRCP and HIDA were undertaken either alone or in combination. Distended GB with thickened wall was the common finding in USG, and cholelithiasis was detected in only 5 (11.36%) cases. This is in contrast with the review by Reilly et al. 1, where cholelithiasis was found in 32% of cases, though calculi are deemed as incidental findings and not believed to play any significant role in the etiopathogenesis of GBT of the condition 52.

In the current review, USG findings were able to guide towards the correct diagnosis in only 5 (33.3%) cases where GBT was established preoperatively and USG had been conducted, pointing towards the low sensitivity of the modality. The suspicious findings included deviation of the GB fundus away from the GB bed and lack of vascular flow. Tiep et al. 9 could pick up a cone-shaped hypoechogenic structure with probe tenderness. In a case presented by Uemura et al. 23, USG and CT scan misdiagnosed a 3-year-old boy as AAC, and when USG was repeated at 48 hours due to clinical deterioration while on conservative treatment (antibiotics, intravenous fluids), GST was diagnosed due to the thickened GB wall and lack of mural blood flow. Dasyam et al. 53 have described a "knot"-like hyperechoic nodular appearance of the twisted cystic duct close to the GB neck in GBT and named it the ‘cystic duct knot sign’ in ultrasound and suggested that the presence of this sign should prompt the diagnosis of GBT, but this sign was not apparent in any case in the current review.

Like USG, the CT abdomen scan also lacked sensitivity and guided towards the correct diagnosis of GBT in only 7 out of 36 cases (19.4%) where CT had been conducted. The common findings detected by CT included GB dilatation, wall edema, shift of GB fundus away from GB bed, and pericholecystic/intraperitoneal fluid. The actual twist/swirl around the GB neck/cystic pedicle was documented in 3 (8.3%), whereas diminished enhancement of GB and abrupt cut-off/tapering were detected in 2 (5.5%) cases each. These findings concur with the literature 1, though multidetector CT (MDCT) has been reported to be successful in the preoperative diagnosis of GBT [54-55]. MDCT can reveal a V-shaped distortion of the extrahepatic biliary ducts and a twisted pedicle inferior to the liver in pre-contrast studies and poor enhancement of the GB wall and “beak” and ‘whirl’ or ‘swirl’ sign 31. The “whirl sign” describes the appearance of the twisted cystic pedicle and is best delineated when the visualization plane is perpendicular to the axis of the cystic pedicle 56. This sign was documented by Miyata et al. 27 in an MRCP study of an 84-year-old female, leading to a preoperative diagnosis of GBT.

In the current series, an 87-year-old female patient reported by Kalyanasundaram et al. 31 in 2021 had a GB-like suspicious structure to the left of the midline anterior to the gastric pylorus with significant inflammatory changes and features of ischemia, suggestive of GBT. However, in her previous CT scan recorded three years prior in 2018, GB was in the correct anatomical location, lending support to the theory that reduction of visceral fat and elasticity with aging tends to elongate the GB mesentery, thereby predisposing to GBT. MRI abdomen/MRCP and/or HIDA scans were used as supplementary imaging tools in 9 (20.4%) and were effective (100% sensitivity and specificity) in the identification of GBT, displaying features such as an ectopic enlarged GB with transverse lie, tapered cystic duct with a swirl sign, and V-shaped distortion of the extrahepatic bile ducts. These features are consistent with the description given in the literature 57. GBT has been reported to manifest as a ‘bulls-eye’ image on hydroxyiminodiacetic acid (HIDA) scans because of the accumulation of the radioisotope within the GB 58, but such an image was not reported in the cases enrolled in this review.

Lack of awareness about GBT and hence the failure to consider it as a differential diagnosis in acute abdomen leads to delays in management despite the availability and utilization of advanced imaging tools. A case included in the current review and reported by Bergeron et al. 47 is a perfect example of this situation. Their patient was an 84-year-old female (BMI = 17.3) who had reported with back pain. X-ray revealed osteoporosis with two vertebral fractures. She was discharged home with analgesics only to be admitted the following day with clinical deterioration. Abdominal USG showed distended GB with wall thickening, but no calculi. After a further delay of one day, the possible diagnosis of acute cholecystitis prompted 99mTc-labeled hepatobiliary iminodiacetic (HIDA) scintigraphy, as well as a dynamic scintigraphy of 20 minutes with a single photon emission computed tomography (SPECT-CT). Adequate hepatic uptake of radionuclide was observed, followed by a rapid biliary tree excretion and visualization of gallbladder activity within 4 minutes, raising the possibility of a pathologic process other than acute cholecystitis. Following an additional day's delay, an abdominal CT scan was requested, which revealed dilatation of the gallbladder up to 5 × 5 × 9.5 cm, fatty infiltration surrounding the GB, and a small amount of free fluid. The dimension of GB on CT scan correlated with that on USG (9 cm) but not with the HIDA scan (3 cm). After reviewing the imaging thoroughly, the discrepancy in the dimensions of the GB between the imaging tools was explained by a “missing” part of the GB on the HIDA scintigraphy, and a possibility of GBT was entertained, leading to laparoscopy where the diagnosis got confirmed. The case presented by Hong Duc et al. 46 was initially misdiagnosed as acute appendicitis, Fitz-Hugh-Curtis syndrome, and acute cholecystitis despite imaging including multiple USG scans and CT scans, and it was on laparoscopy after 3 days that GBT got detected. These cases also serve as a reminder that laparoscopy should continue to be utilized as a tool for diagnosis and therapeutics in challenging cases.

GBT is unlikely to respond to conservative treatment or percutaneous cholecystostomy, and hence, once a diagnosis is established, emergent cholecystectomy is to be undertaken to prevent perforation with biliary peritonitis and the subsequent sepsis, which results in significant morbidity and mortality 16,51,59. Laparoscopic cholecystectomy for GBT is not considered more difficult than the operation conducted for usual acute cholecystitis and has excellent outcomes 47. In the current review, the treatment was surgical in 43 (97.7%) and non-surgical in 1 (2.3%). The approach to surgical treatment was laparoscopic in 32 (72.7%), including one robot-assisted, and open/laparotomy in 11 (25%), and procedures involved detorsion of GB followed with cholecystectomy (n=39, 90.7%), percutaneous decompression of GB followed with cholecystectomy (n=1, 2.3%), cholecystectomy (n=2, 4.7%), and partial cholecystectomy with staplers (n=1, 2.3%). The first successful laparoscopic cholecystectomy for GBT was reported in 1994, and since then, it has emerged as the approach of choice 1. Detorsion of the gallbladder before clipping of the cystic duct and artery reduces the risk of bile duct injuries 5. Bekki et al. 60 published a case study of their experiences with CBT in 2017 and concluded that laparoscopic cholecystectomy, rather than open cholecystectomy, would be the gold-standard treatment choice upon diagnosis confirmation. Garciavilla et al. 6 had also asserted in 2010, that whenever GBT is suspected, it should be promptly resolved laparoscopically.

One patient in the current series where non-surgical management was adopted was reported by Yoshida et al. 15 and is unique. A 90-year-old female presented with loss of appetite, lower back pain, abdominal pain, and fever for 30 days, and a CT scan could not visualize the GB but detected a twisted GB neck in continuity with a 10 cm encapsulated abscess, reaching the anterior abdominal wall. Percutaneous drainage of the abscess and antibiotics successfully managed the case. Remarkably, post-treatment CT showed complete resolution of the abscess, but the GB itself was no longer visible. In the previous literature, Aharoni et al. 61 have reported a successful diagnosis and non-surgical detorsion of GBT by endoscopic retrograde cholangiopancreatography (ERCP).

On surgical exploration, there were features of gangrene /necrosis in 37 (84.1%) cases and were confirmed on histopathological analysis of the retrieved specimen, pointing towards a delay in diagnosis. There were, however, no cases of perforation and biliary peritonitis. The direction of torsion was mentioned in 31 (70.5%) cases, and the ratio of counterclockwise to clockwise was 2:1. The magnitude of torsion was mentioned in 27 (61.3%) cases, and it ranged from 180 to 720 degrees, with most cases having a 360- or 180-degree twist. Torsion is termed as incomplete, in which rotation is less than 180 degrees, or complete, in which rotation is greater than 180 degrees; all the cases in this review were complete. Regarding the direction, it has been traditionally linked to the precise location of GB and its posteriorly lying organ, and it is believed that the clockwise rotation is secondary to the gastric and duodenal peristalsis, whereas anticlockwise rotation occurs because of colonic peristalsis 62.

The postoperative phase was eventful in 2 (4.7%) cases in the current review. One patient suffered from hospital-induced delirium, and in another, there was a left parietal infarct, leading to a prolonged hospital admission. There were no mortalities, though the mortality rate with GBT is 6%, according to literature 1. It is conceivable that early diagnosis and intervention can lower the morbidity and mortality associated with GBT because, based on Reilly et al.'s review of the literature 1, the deaths linked to GBT occurred in elderly patients with multiple comorbidities, and none of the reported deaths occurred in patients who had a confirmed preoperative diagnosis of GBT.

Conclusion

Gallbladder torsion is a rare but potentially life-threatening disorder that should be considered during evaluation of acute abdomen, particularly in the thin elderly female patients. Due to lack of awareness, preoperative diagnosis is uncommonly made even after the recent advances in diagnostic imaging modalities. Cholecystectomy is mandatory whenever the diagnosis is suspected or confirmed by imaging, and the laparoscopic approach is safe in most of the cases. Delay in diagnosis and management increases morbidity and mortality. There is a need to enhance awareness about this disorder among the healthcare professionals.

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