Introduction

Dyskinesia of the gallbladder causes gallstone-like symptoms including abdominal pain and vomiting as a result of functional abnormalities of bile flow, in the absence of any organic abnormalities in the biliary system, including the gallbladder. Cholecystectomy is a common treatment, but it is often difficult to diagnose because its clinical symptoms resemble those of irritable bowel syndrome and some psychogenic disorders.

Here, we report a case of biliary dyskinesia in which biliary scintigraphy performed before and after endoscopic gallbladder stenting (EGBS) was predictive of successful cholecystectomy.

Case report

A 40-year-old woman presented with a several-year history of epigastric and right quadrant pain and vomiting after eating. She had been treated for functional dyspepsia but her symptoms did not improve, and she was, therefore, referred to our hospital for a thorough examination.

Her height was 158.2 cm, weight was 53.1 kg, and her body mass index was 21.2 kg/m2. She had a history of nephrotic syndrome, which was diagnosed when she became pregnant, and was in remission on 3 mg prednisolone at the time of examination. Blood tests showed free T3 2.18 U/ml, which was slightly below the normal range, but otherwise within normal limits (Table 1).

Table 1 Laboratory findings at first medical examination
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During a medical interview, she complained of pain in the right quadrant, especially after lipid intake, and biliary pain was suspected. Abdominal plain computed tomography showed no organic disease of the biliary system, such as gallstones or common bile duct stones (Fig. 1).

Fig. 1
figure 1

Abdominal plain computed tomography at first medical examination. There were no stones or strictures in the biliary tract.

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Endoscopic ultrasound also revealed no gallstones or stenosis, including debris in the gallbladder or bile ducts (Fig. 2).

Fig. 2
figure 2

Endoscopic ultrasound images after first medical examination are shown. a An image of the gallbladder observed from the duodenal bulb. Endoscopic ultrasound showed no gallstones and debris in the gallbladder. The wall of the gallbladder was thickened and a 2 mm polyp was observed. b An image of the bifurcation of the cystic duct observed from the duodenal bulb. There was no obvious choledochal stone or stenosis in the common bile duct and cystic duct. c An image of the common bile duct and main pancreatic duct observed from the descending part of the duodenum. Pancreaticobiliary maljunction was not observed, and no other abnormalities including choledochal stone and stenosis were found

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In light of the patient’s biliary pain but absence of organic disease, we suspected a functional gallbladder or Oddi’s sphincter disorder, according to the Rome IV criteria, and performed biliary scintigraphy for supplementary diagnostic purposes (Fig. 3).

Fig. 3
figure 3

In the first biliary scintigraphy, the figure shows the amount of radionuclides over time. It can be seen that the radionuclides accumulate in the gallbladder (yellow) without decreasing over time. Radionuclides in the cystic duct (purple), common bile duct (light blue), and intrahepatic bile duct (red, green and blue) do not accumulate, and there is no ejection into the duodenum

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Biliary scintigraphy shows that the accumulation of radionuclides increases with time without decreasing from about 20 min, leading to a suspicion of functional gallbladder and Oddi’s sphincter disorder. We, therefore, administered a calcium channel blocker to loosen Oddi’s sphincter, but the patient’ symptoms did not improve. Endoscopic sphincterotomy (EST) was then performed to abolish Oddi’s sphincter, but this also failed to improve her symptoms (Fig. 4a, b). Biliary scintigraphy was repeated to confirm bile flow and, as in the first case, radionuclide accumulation increased over time (Fig. 4c).

Fig. 4
figure 4

The images at the time of endoscopy when EST was performed and biliary scintigraphy after treatment are shown. a EST was performed to abolish Oddi’s sphincter. A small incision was made in this case. b Fluoroscopic images during the first endoscopic retrograde cholangiopancreatography. Flexion of the cystic duct was observed. c Biliary scintigraphy after EST also shows that radionuclides accumulate in the gallbladder (yellow) without decreasing with time. Radionuclides in the cystic duct (purple), common bile duct (light blue), and intrahepatic bile duct (red, green and blue) do not accumulate and there is no ejection into the duodenum

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Based on these results, we diagnosed dyskinesia of the gallbladder and recommended cholecystectomy; however, the patient refused to undergo cholecystectomy immediately because she was uncertain if the treatment would be effective after such a long history of disease. The patient had a strongly flexed cystic duct, which is thought to be the cause of gallbladder dyskinesia, and EGBS was performed to try to correct the flexed part of the cystic duct (Fig. 5a, b). After EGBS, patient’s symptoms improved remarkably. A third biliary scintigraphy was performed to determine the efficacy of the treatment, and showed that radionuclides in the gallbladder and common bile duct accumulate up to 30 min, but after 30 min, the radionuclides in the gallbladder decrease due to excretion into the duodenum (Fig. 5c). This indicated that cholecystectomy would be effective, and cholecystectomy was accordingly performed on a standby basis. The patient’s subsequent course was good, and a laparoscopic cholecystectomy was performed two months after EGBS. After laparoscopic cholecystectomy, there was no recurrence of symptoms such as right quadrant pain and vomiting up to 8 months, and the patient continues to be followed clinically.

Fig. 5
figure 5

Images at the time of endoscopy when EGBS was performed and biliary scintigraphy after treatment are shown. a Endoscopic retrograde cholangiopancreatography images shows the strongly flexed cystic duct (arrow). b Endoscopic retrograde cholangiopancreatography image when EGBS was performed. A Zimmon (Cook Group Inc., IN, USA) 7 Fr, 14 cm plastic stent was placed into the gallbladder. c Biliary scintigraphy after EGBS shows that radionuclides flow into the gallbladder (yellow) for up to 20 min. In contrast, there is no accumulation of radionuclides in the intrahepatic bile duct (red, green) or gallbladder, and radionuclides are ejected into the cystic duct (purple), common bile duct (light blue) and duodenum (blue)

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Discussion

Gallbladder dyskinesia is a disease in which functional abnormalities of bile flow cause gallstone attack-like symptoms, such as abdominal pain and vomiting, in the absence of any organic abnormality in the biliary system, including the gallbladder. It is classified as a functional gallbladder disorder according to the Functional Gallbladder and Sphincter of Oddi Functional Gastrointestinal Disorders in the Rome IV diagnostic criteria [1]. Bile produced in the liver is secreted into the intrahepatic bile ducts, concentrated in the gallbladder, and excreted via the papilla into the duodenum. Cholecystokinin, a gastrointestinal hormone secreted from the duodenum, is involved in the coordinated movement of the gallbladder, Oddi’s sphincter, and duodenum at this time, causing the gallbladder to contract and Oddi’s sphincter to relax. This process is believed to be strongly involved in bile-excretion disorders [2].

The Rome IV classification is based on the diagnostic criteria of biliary pain and absence of gallstones or other organic disease, with low biliary ejection fraction on biliary scintigraphy and normal blood enzymes, bilirubin, and amylase/lipase as supplementary diagnoses. The diagnosis of gallbladder dyskinesia is suggested when the above criteria are fulfilled [1].

Biliary scintigraphy using 99 m technetium is commonly used to evaluate the gallbladder ejection fraction. Given that changes in radioisotopes over time reflect bile flow, changes can, thus, be expressed semi-quantitatively by the number of γ-rays [3,4,5]. However, other diseases, including functional gastrointestinal disorders, can also cause delayed excretion, and the Rome IV diagnostic criteria emphasize the importance of patient selection in the decision to perform biliary scintigraphy. In addition, although there is no correlation between biliary ejection fraction and Oddi’s sphincter pressure, elevated sphincter pressure has been reported in patients with gallbladder dyskinesia, which may make it difficult to differentiate between gallbladder dyskinesia and functional Oddi’s sphincter disorder [6]. In the current case, initial biliary scintigraphy showed excretion of radionuclides into the common bile duct after 15 min and EST was, thus, chosen as the treatment; however, there was no improvement in the patient’s symptoms. A diagnosis of gallbladder dyskinesia was therefore made based on residual delayed bile excretion from the gallbladder on biliary scintigraphy, performed to confirm the efficacy of treatment.

Treatment of gallbladder dyskinesia involves pharmacotherapy with anticonvulsants and neuromodulators, but cholecystectomy is considered if these measures are ineffective [7,8,9]. Although cholecystectomy has been reported to be effective in more than 80% of patients with gallbladder dyskinesia, there has only been one report of a randomized trial, and further investigations are therefore needed [7, 10].

In the present case, cholecystectomy was recommended after the diagnosis of gallbladder dyskinesia but the patient was hesitant to undergo surgery, and EGBS was, therefore, performed to improve gallbladder bile outflow. This helped to confirm the diagnosis of gallbladder dyskinesia and predicted the efficacy of cholecystectomy.

Conclusion

In conclusion, this is the first report of a patient with gallbladder dyskinesia in whom treatment response was confirmed by biliary scintigraphy before and after EGBS. This case suggests that that EGBS may be an effective procedure for supporting the diagnosis and treatment of gallbladder dyskinesia.