Cholecysto-hepatic fistula in type III gallbladder perforation: A rare etiology of liver abscess; case report

Abstract

Introduction and importance

Gallbladder perforation (GBP) with cholecystohepatic fistula is an extremely rare complication of acute and/or chronic gallbladder diseases. Niemeier classified GBP into three types each characterized by specific signs and symptoms. Radiological investigations such as abdominal ultrasonography (USG) and computed tomography (CT) are crucial to evaluate and diagnosing GBP, while fistulae are usually identified intraoperatively.

Case presentation

A 77-year-old female patient, with a background medical history of multiple comorbidities, presented to our hospital with a one-week history of abdominal pain. Laboratory investigations showed abnormal values. The abdominal CT scan revealed a mildly enlarged liver, distended gallbladder, and liver abscess. Then, ultrasound-guided aspiration was done, and the clinical picture was consistent with calcular cholecystitis complicated with liver abscess. So, laparoscopic cholecystectomy was decided and the intraoperatively detected fistula was excised. The resected perforated gallbladder was sent for histopathological studies. The postoperative period was uneventful.

Clinical discussion

GBP is an unusual entity that is categorized into three types. It is considered a gallbladder complication and surgical emergency. The clinical features of GBP are non-specific and radiological tools aid in diagnosis demonstration. In the presented case of type III GBP, cholecystohepatic fistula was detected and excised intraoperatively.

Conclusion

Due to the rare entity of type III GBP in association with liver abscess, we report the case of a 77-year-old female with right lower quadrant pain, found to be caused by type III GBP with cholecystohepatic fistula and liver abscess.

Highlights

• •Gallbladder perforation is a life-threatening complication of calcular cholecystitis and type III is even rarer.
• •Gallbladder perforation type III with cholecystohepatic fistula is an extremely rare complication of gallbladder diseases.
• •The signs and symptoms of GBP are usually non-specific, while fistulae are mostly detected intraoperatively.
• •Both USG and CT provide high diagnostic values for GBP.
• •The laparoscopic cholecystectomy with abscess drainage and/or fistula excision is a viable treatment option.

1. Introduction

Gallbladder perforation (GBP) is an infrequent life-threatening complication of calcular cholecystitis [1]^, [2]^, [3]. Historically, Duncan reported the first case of GBP in 1844 [1]^, [2]. This condition is associated with high morbidity and mortality rates [1]^, [3]^, [4]. In 1934, Niemeier categorized the perforation of the gallbladder into three types [1]^, [3]^, [5] explained in Table 1.

Table 1.

Types of GBP according to Niemeier classification [2]^, [6]^, [7].

Type I	Acute, generalized peritonitis.
Type II	Subscute, localized peritonitis.
Type III	Chronic, characterized by cholecystoenteric fistula.

Here we report a 77-year-old female patient who presented with right lower quadrant pain that was due to type III GBP, cholecystohepatic fistula, and liver abscess. Our work has been reported in line with the SCARE Guidelines 2020 criteria [8].

2. Presentation of case

A 77-year-old female patient, with a background medical history of diabetes mellitus type II, hypertension, chronic renal disease, epilepsy, and ischemic heart disease, presented to our hospital with a one-week history of abdominal pain. The abdominal pain was localized in the right lower quadrant, gradual onset, sharp and colicky in nature, non-radiating, and associated with nausea. No urinary symptoms or peritonitis features were reported. The patient experienced a similar pain episode 4 days back that was not subsided by antibiotics.

The patient was conscious, alert, oriented, and vitally stable on physical examination. She had mild tenderness over the upper and lower quadrants with no peritonitis features. The laboratory investigations on admission showed Amylase 44 IU/L, total bilirubin 7.1 umol/L, conjugate bilirubin 1.67 umol/L, Alkaline phosphatase 103 IU/L, serum sodium 125 mmol/L, serum potassium 4.86 mmol/L, and white cell count 23 10⁹/L.

The abdominal ultrasound showed bilateral grade III nephropathy, calculary gallbladder with a heterogeneous cystic lesion in segment V right liver lobe measuring 6.6 × 4 × 4.8 cm with related echogenic edema. The mildly enlarged liver, markedly distended gallbladder, and liver abscess were visualized. Then, the US and fluoroscopic guided aspiration of the abdominal collection was performed with pigtail drain insertion, which drained about 50 cc of pus. Moreover, the drainage of the right hepatic lobe abscess was uneventful. Further CT assessment was required.

The CT scan of the abdomen and pelvis revealed a multinodular cystic lesion located in the right liver lobe (segment V) adjacent to the gallbladder, measuring about 2 × 2 × 2 cm with an inserted pigtail catheter within. Also, a distended gallbladder with two stones inside measuring about 3 cm and 0.7 cm in length was detected [Fig. 1].

Fig. 1.

Abdominopelvic CT scan showing cystic lesions of the liver that are adjacent to the gallbladder.

Once the drain was checked under fluoroscopy, it denoted a fistulous tract between the liver abscess cavity and gallbladder [Fig. 2].

Fig. 2.

The fluoroscopic guided aspiration drain insertion, showing fistula.

At this point, laparoscopic cholecystectomy was performed. A small liver abscess adjacent to the gallbladder fundus was identified and drained laparoscopically. During the procedure, the gallbladder wall was perforated and through this opening, a fistula tract between the gallbladder fundus and the liver abscess was identified [Fig. 3]. The tract was dissected and tract.

Fig. 3.

A: Intraoperative findings of fistula. B: GBP at the fundus part.

The perforation site was noted to be in the fundus. The resected perforated gallbladder was sent for histopathological studies that are important to rule out gallbladder malignancies in such cases [Fig. 4]. It revealed chronic cholecystitis with adenomyomatosis with mucosal erosion. The postoperative period was uneventful and the patient was discharged on the seventh postoperative day.

Fig. 4.

Histopathology showing,

A: Rokitansky-Aschoff sinuses.

B: Adenomyomatosis.

C: Chronic Cholecystitis.

3. Discussion

GB diseases, either acute or chronic, can develop rare complications. GB and intra-hepatic perforations are even rarer and extraordinary complications [3]^, [9]^, [10]. In all acute cholecystitis cases, the prevalence of GBP ranges from 2 to 18 % [1]^, [6]. Cholecystitis is classified and graded according to the Tokyo Guidelines (TG) which rely on different grades of inflammation [11]. The diagnostic standards of TG are based on a combination of regional and systemic inflammatory symptoms, imaging, and clinical suspicion. TG categorizes the cholecystitis severity into mild (grade I), moderate (grade II), and severe (grade III) [11]. Furthermore, the American Association for Surgery of Trauma Grading Scale (AAST) developed a grading severity based on physiologic and anatomic severity, patient age, and comorbidity [11]. AAST described the cholecystitis severity into five grades as shown in Table 2.

Table 2.

AAST disease grade description [11].

	AAST	Acute Cholecystitis
Grade I	Local disease	Acute cholecystitis
Grade II	Confined to the organ Minimal abnormality Local disease	Gallbladder empyema or gangrenous cholecystitis or emphysematous cholecystitis
Grade III	Confined to the organ Severe abnormality Local extension beyond the organ	Gallbladder perforation with local contamination
Grade IV	Regional extension beyond the organ	Gallbladder perforation with pericholecystic abscess or gastrointestinal fistula
Grade V	Widespread extension beyond the organ	Gallbladder perforation with generalized peritonitis

The risk factors of GBP include infections, diabetes mellitus, malignancies, traumas, and atherosclerotic cardiac diseases [1]^, [2]^, [9]. The most common site of perforation is the GB fundus [5]^, [12]. The exact etiology is not well understood [10]. GBP can be due to gallbladder wall necrosis, ischemia, and increased intraluminal pressure within the gallbladder [7]. In the current case, liver abscesses arise secondary to intrahepatic GBP [7].

The clinical manifestations of patients with GBP are non-specific and differ from cholecystitis complication features, leading to delayed diagnosis [3]^, [13]^, [14]^, [15]. GBP signs and symptoms are imitated by multiple pathologies such as liver abscess, cholecystitis, and gallbladder malignancy [10]^, [17]. On the other hand, fistulae are mostly asymptomatic and usually incidentally detected intraoperatively [3]^, [4]^, [17]. The clinical picture of our case was consistent with type III GBP with cholecystohepatic fistula.

Abdominal ultrasonography (USG) and computed tomography (CT) play an important role in the evaluation and diagnosis of GBP. USG is the initial diagnostic modality to be used [2]^, [6]^, [16]^, [17]. This radiological method provides an excellent outcome in clinical cases showing sonographic hole signs, indicating gallbladder defects [3]^, [5]. Moreover, a CT scan is used following the ultrasound to demonstrate the GBP diagnosis [1]^, [2].

A CT scan is more precise in identifying abscesses, intraperitoneal fluid, and pericholecystic fluid [1]. In the presenting case, an abdominal CT scan was performed and its findings were mentioned above in the case presentation section.

Gallbladder perforation is managed by surgical techniques [4]. Cholecystectomy and/or abscess drainage are the main surgical approach to treat GBP [3]^, [4]. Cholecystectomy can be performed following percutaneous US-guided abscess drainage. Acute, gangrenous, perforated cholecystitis as well as uncomplicated cholecystitis can be treated by laparoscopic cholecystectomy [3]. Additionally, there are other alternative management tools available for patients who are unfit for cholecystectomy. These include endoscopic transpapillary gallbladder stenting and ultrasound-guided transgastric or transdudenal drainage of the gallbladder with stenting [3].

As a result, the current case is type III GBP and its definitive treatment modality is cholecystectomy and/or abscess drainage [1]^, [9]^, [16]. Laparoscopic cholecystectomy was performed after the liver abscess drainage under radiological guidance. The procedure was associated with difficulties and surgical challenges due to the gallbladder being acutely inflamed liver abscess formation, and an extremely rare cholecystohepatic fistula requiring excision [1]^, [9]^, [10].

Date et al. reported a gallbladder perforation case series that demonstrated the most efficient method of managing patients with such disease by a multimodal diagnostic workup and precise diagnosis of the type of gallbladder perforation [18]. Also, the study suggested adherence to the original Neimeier's categorization for reporting data on gall bladder perforation in future investigations, pending the emergence of additional information that would permit the creation of a more comprehensive classification scheme [18].

4. Conclusion

Despite the rarity of type III GBP, it should be considered in elderly patients with unusual abdominal pain. Imaging modalities help to demonstrate the diagnosis of type III GBP and its related clinical features. It is rare to be diagnosed preoperatively. To cure type III GBP and liver abscess, laparoscopic cholecystectomy and/or abscess drainage is a viable treatment option. Our case report emphasizes the diagnostic and surgical challenges of GBP as a life-threatening complication.

Funding

No funding or grant support.

Ethical approval

Ethical approval is exempt/waived at our institution.

Consent

Written informed consent was obtained from the patient to publish this case report and accompanying images. On request, a copy of the written consent is available for review by the Editor-in-Chief of this journal.

Research registration

Not applicable.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Guarantor

Athary Saleem, B.Med.Sc., M.D.; Department of General Surgery, Al Adan Hospital, Kuwait

CRediT authorship contribution statement

Athary Saleem: literature review, writing, paper, picture editing, manuscript drafting.

Maznah Almutairi: assisted in surgery, and paper editing.

Ahmed Hassan: assisted in surgery, and paper editing.

Nimer Al-Shadidi: performed surgery, and paper editing.

Khaled Alshammari: performed surgery, critical review, picture editing, supervision, and final approval.

Declaration of competing interest

There are no conflicts of interest to declare by all the authors.