Gallbladder volvulus in a patient with chronic lymphocytic leukemia treated with laparoscopic cholecystectomy.

Gallbladder volvulus is a rare condition that most commonly occurs in elderly women and often mimics acute cholecystitis in its presentation. This condition is a surgical emergency requiring cholecystectomy as it can lead to gallbladder perforation and bilious peritonitis with high morbidity to the patient. An 85-year-old woman with chronic lymphocytic leukemia presented with acute-onset right upper-quadrant abdominal pain and associated nausea with emesis. After admission to the surgical service and initiation of intravenous antibiotics, the patient was taken to the operating room for surgical management due to the persistence of symptoms. Intraoperative findings included a necrotic appearing gallbladder that was twisted on the cystic duct. Laparoscopic cholecystectomy was performed, which was complicated by bile leak requiring endoscopic retrograde cholangiopancreatography with bile duct stenting followed by operative washout. Gallbladder volvulus can be challenging to diagnose. This condition should be suspected in elderly women with acute-onset abdominal pain and imaging concerning for acute cholecystitis. Emergent cholecystectomy is the treatment of choice for gallbladder volvulus.

INTRODUCTION

Gallbladder volvulus is an uncommon condition that can mimic acute cholecystitis. Since first described by Wendel in 1898,[1] approximately 500 cases have been reported.[2] This condition is seen most commonly in elderly women, and as described by Gross in 1936, is associated with loss of adhesions between the liver and gallbladder.[3] This anatomical abnormality allows the gallbladder to rotate partially or completely on its long axis, resulting in compromised vascular perfusion to the gallbladder.[4] Preoperative diagnosis is challenging due to symptoms, laboratory assays, and radiographic imaging that are similar to those of acute cholecystitis.[5] It is usually diagnosed intraoperatively, and the treatment is detorsion and cholecystectomy.[6] We report the case of an elderly woman initially diagnosed with acute cholecystitis who was found intraoperatively to have complete gallbladder volvulus that was treated with laparoscopic cholecystectomy.

CASE REPORT

An 85-year-old woman presented to the emergency department with complaints of acute-onset abdominal pain. She reported a 1-day history of epigastric pain that radiated to the right upper quadrant and right flank. It was associated with anorexia, nausea, and emesis. She denied fever, chills, change in bowels, or any other symptoms. She was afebrile with mild tachycardia at initial presentation. Physical examination was notable for a soft, nondistended abdomen with tenderness to palpation in the epigastrium and right upper quadrant. She was found to have a positive Murphy sign. Her past medical history was significant for chronic lymphocytic leukemia for which she was currently treated with ibrutinib. Laboratory assay revealed a white blood cell count of 168.7, which was within her baseline levels. Her liver function tests were normal, with a total bilirubin of 0.5, alkaline phosphatase of 98, and normal transaminases. A computed tomography (CT) scan of the abdomen revealed a dilated gallbladder with a thickened wall and pericholecystic edema concerning for acalculous cholecystitis [Figure 1]. No obvious gallstones were noted. An ultrasound of the right upper quadrant was obtained in order to attempt to better visualize gallstones. This ultrasound did not visualize gallstones or sludge, but did confirm the presence of a distended gallbladder with a thickened wall. She was admitted to the general surgery service, and intravenous ciprofloxacin and metronidazole were administered [Table 1]. Due to the persistence of symptoms, she was taken to the operating room for laparoscopic cholecystectomy with a presumed diagnosis of acute cholecystitis. After the induction of general anesthesia, she had a palpable mass in the right upper quadrant. Upon insertion of the laparoscope into the abdomen, it was noted that the gallbladder was completely gangrenous. There was only one attachment from the underside of the liver to the gallbladder, which measured approximately 1 cm. Distal to this attachment, the gallbladder was completely torsed. After detorsion, the infundibulum was also noted to be gangrenous, but the proximal cystic duct was not, and laparoscopic cholecystectomy was performed. Given the very short attachment of the gallbladder to the liver fossa, great care was taken to completely define the anatomy prior to resection. The total operative time was 135 min with minimal blood loss. Histopathologic findings of the gallbladder specimen revealed acute cholecystitis with involvement by chronic lymphocytic leukemia. Postoperatively, her course was complicated by abdominal distention, right shoulder pain, and hyperbilirubinemia. On postoperative day #3, a repeat ultrasound of the right upper quadrant was performed, which revealed a small amount of perihepatic fluid concerning for bile leak. There was no choledocholithiasis or intrahepatic biliary dilatation. A hepatobiliary iminodiacetic acid (HIDA) scan confirmed leakage of the bile posteriorly and at the dome of the liver. The patient then underwent endoscopic retrograde cholangiopancreatography that showed normal biliary anatomy with peripheral bile leakage therefore a common bile duct stent was placed. She had a CT scan of the abdomen on postoperative day #4 for persistent symptoms, revealing persistent biloma measuring 11 cm at the greatest diameter. She returned to the operating room for laparoscopic washout and drain placement. The patient was discharged to home on postoperative day #9/#5. One week after the discharge, the patient had her drain removed in the outpatient clinic and after further recovery, her biliary stent was removed endoscopically.

Figure 1

Gallbladder volvulus. (a) Coronal reconstruction of contrast-enhanced computed tomography demonstrates a distended and conical gallbladder freely suspended outside its normal anatomic fossa, associated with pericholecystic fluid and mild intrahepatic ductal dilatation. The transition from the distended lumen to the angulated and abruptly tapered infundibulum may resemble a curved beak at the suspected point of twist (arrow). (b) Gray-scale ultrasound image shows diffuse thickening of the gallbladder wall with trace pericholecystic fluid but no intraluminal stones or sludge

Table 1

Timeline of events for case

Timeline of case
HD #1	Patient presents to the emergency department and admitted to surgical service. Laparoscopic cholecystectomy late on HD #1
POD #1–2	Patient complains of right shoulder pain, abdominal distention, and total bilirubin increased to 1.2 from 0.6
HD #4, POD #3	Right upper-quadrant ultrasound reveals perihepatic fluid. HIDA scan confirms bile leak. ERCP performed revealed low-grade bile leak. 10-Fr, 7-cm plastic biliary stent placed. Laparoscopic washout with perihepatic drain placement performed
HD #10, POD #9/6	Discharged to home in good condition with plastic biliary stent and perihepatic drain in place
POD #17/14	Perihepatic drain removed in the clinic
POD #41/38	Repeat ERCP performed, no evidence of persistence of bile leak, plastic biliary stent removed

HD: Hospital day, POD: Postoperative day, HIDA: Hepatobiliary iminodiacetic acid, ERCP: Endoscopic retrograde cholangiopancreatography

DISCUSSION

Gallbladder volvulus, also known as gallbladder torsion, refers to the twisting of the gallbladder around its mesentery. First described by Wendel in 1898, it remains a rare phenomenon as approximately only 500 cases have been reported in the literature.[17] The condition is most prevalent in elderly women, but has also been documented in pediatric populations.[89] It typically presents as acute-onset right upper-quadrant pain and is frequently misdiagnosed as acute acalculous cholecystitis, as in the current report.

Gallbladder volvulus is associated with a “free-floating” gallbladder, in which there is minimal fixation of the gallbladder to the liver bed. This configuration is associated with an abnormal gallbladder mesentery which allows the gallbladder to twist along the axis of the cystic artery and duct.[10] The Gross classification describes two anatomical variants of gallbladder mesentery.[3] In Type I, the mesentery is wide and attached to the cystic duct as well as a portion of the gallbladder body. In Type II, the mesentery only supports the cystic duct and artery. These characteristic mesenteries have been postulated to occur congenitally or are acquired through loss of fat, liver atrophy or visceroptosis, and the relaxation and atrophy of previously normal mesentery in the elderly.[11] Triggers for gallbladder torsion have also been proposed, including vigorous peristalsis of the neighboring organs.[8] Kyphoscoliosis and atherosclerosis of the cystic artery may contribute as fulcrums for torsion.[12] Interestingly, cholelithiasis does not appear to play a dominant role in the development of volvulus, as a review of 235 cases in the Japanese literature found gallstones in only ~25% of patients afflicted.[13]

Diagnosing gallbladder volvulus preoperatively is challenging due to low prevalence, nonspecific radiographic findings, and a clinical presentation that mimics acute cholecystitis. Sonographic features are often nonspecific to gallbladder volvulus, such as gallbladder wall thickening and pericholecystic fluid.[14] However, findings of the gallbladder out of the fossa, conical shape, and/or floating anteriorly without gallstones should increase should increase suspicion for gallbladder torsion.[415] Doppler imaging may be helpful to demonstrate compromised blood flow.[16] CT of the abdomen may show abnormal positioning and impairment of blood flow, but is not specific to gallbladder volvulus.[5] Magnetic resonance imaging of the abdomen has also been utilized in the pediatric population for diagnosis.[89] HIDA scan and magnetic resonance cholangiopancreatography (MRCP) are additional imaging modalities reportedly utilized to help diagnose gallbladder volvulus. Tracer accumulation can lead to a “bull's eye” appearance of the gallbladder on HIDA scan,[16] and MRCP can reveal gallbladder dilatation and often twisting of the cystic duct.[17]

A review of the literature found that only 9.8% of the reported cases of gallbladder volvulus were correctly identified preoperatively.[18] Due to challenges with diagnosing gallbladder volvulus, Lau et al. proposed the “Triad of Triads” [Table 2] comprising factors related to appearance (elderly female, thin, and spinal deformities), symptoms (sudden onset, right upper-quadrant pain, and early emesis), and physical examination findings (nontoxic presentation, palpable right upper-quadrant mass, and pulse–temperature discrepancy), in order to more easily identify potential patients with the condition.[19]

Table 2

Triad of triads for early recognition of potential gallbladder volvulus[19]

Appearance	Symptoms	Physical examination
Elderly woman	Right upper-quadrant pain	Palpable right upper-quadrant mass
Thin habitus	Sudden onset	Nontoxic appearance
Kyphotic spinal deformity	Early emesis	Pulse–temperature discrepancy

Prompt diagnosis is critical in the management of gallstone volvulus because progression to necrosis is rapid once torsion has occurred. Delay in intervention can result in perforation and bilious peritonitis, which increases the associated mortality to approximately 5%.[20] Emergent cholecystectomy is the preferred treatment for gallbladder volvulus upon diagnosis.[5] The laparoscopic approach is appropriate, as the long mesentery and separation of the gallbladder from the liver bed may ease laparoscopic removal of the gallbladder. Laparoscopic cholecystectomy also results in quicker postoperative recovery. It is important to note that delay in diagnosis resulting in progression to necrosis can lead to increased difficulty in the identification of anatomical structures, often necessitating laparotomy.[10] To facilitate early postoperative recovery for patients with gallbladder volvulus, swift diagnosis and surgical intervention are essential.

CONCLUSION

Gallbladder volvulus is a relatively rare condition that can be an elusive diagnosis. The condition is most prevalent in elderly women presenting with right upper-quadrant pain and a mass on physical examination, and suspicion should be heightened if symptoms are refractory to antibiotic therapy. Once diagnosed, emergent cholecystectomy is indicated to prevent gallbladder necrosis, perforation, and bilious peritonitis.

Informed consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.