XANTHOGRANULOMATOUS CHOLECYSTITIS
A SURGICAL CHALLENGE

Bestoun H.A., Al Aali A.Y. and Abu Sabib A.R.
General Surgery Section, Department of Surgery Hamad Medical Corporation, Doha, Qatar

Abstract:

Xanthogranulomatous cholecystitis is a severe inflammatory process, which gives rise to dense adhesions that complicate cholecystectomy. Four cases of significant injuries to the main biliary passages and surrounding structures are discussed. We suggest greater caution in the dissection of the gall bladder, a lower threshold for early conversion, examining frozen sections in suspicious cases and accurate preoperative diagnosis whenever possible.

Introduction:

Xanthogranulomatous cholecystitis is a destructive type of gall bladder inflammation, which may be focal or diffuse and is mostly chronic. It is a histopathological diagnosis with peculiar preoperative findings. It requires special consideration because of the dense inflammation and adhesions to the structures surrounding the gall bladder that necessitate pre- and per-operative awareness with precautions to avoid complications.

We discuss four cases of xanthogranulomatous cholecystitis with severe postoperative complications following cholecys-tectomy that occurred in Hamad General Hospital, Doha, Qatar.

Case 1:

A 20-year-old man presented with clinical and ultrasonic diagnosis of acute cholecystitis with gall bladder sludge. Laparoscopic exploration showed dense adhesions between the gall bladder and the surrounding structures. After conversion and trial of dissection, the transverse colon and the second part of the duodenum were injured. This was managed by partial cholecystectomy, repair of the duodenal injury and tube duodenostomy. Because of the severity of the duodenal injury, a pyloric exclusion and gastro-jejunostomy were added. The transverse colon was resected with an end-to-end anastomosis. Postoperatively he did well. Pertubal duodenostomy showed no dye leak and a patent lumen (Figure 1). The patient was later discharged home in a good condition.

Figure 1: Pertubal Duodenostomy

Case 2:

A 49-year-old man presented with clinical and ultrasonic diagnosis of chronic cholecystitis and gall bladder sludge. Laparoscopic exploration revealed dense adhesions around the gall bladder. Five days following laparoscopic exploration, the patient was re-admitted for abdominal pain, fever and jaundice. A diagnosis of postoperative bile collection was confirmed by ultrasound examination and was managed by insertion of a pigtail catheter.

Endoscopic retrograde cholangiopancreatography (ERCP) showed complete transection of the common bile duct with an obvious dye leak to the sub-hepatic area (Figure 2).

Figure 2: ERCP showing complete transection of the CBD with free dye leak.

Percutaneous transhepatic cholangiography (PTC) was performed to identify the proximal anatomy of the biliary passages. This showed a complex injury with the left main hepatic, right upper and right lower hepatic ducts all opening separately (Figure 3). Re-exploration confirmed the PTC findings, which were managed by mucosal graft hepatico-jejunostomy (Figure 4). The patient was discharged home in a good condition.

Figure 3: Complex injury at the confluence of the CHD with the Shiba needle inside

Figure 4: Post hepaticojejunostomy, showing free passage of the dye to the jejunal loop.

Figure 5: Peroperative cholangiography, showing complete transection of the CBD.

Case 3:

A 57-year-old man presented with a clinical and ultrasonic picture of chronic cholecystitis and gall bladder sludge. Laparoscopic examination revealed dense adhesions in the area of the gall bladder. Peroperative cholangiography showed complete transection of the common hepatic duct (Figure 5). After conversion an iatrogenic duodenal injury was also identified. Roux-en-Y hepaticodochojejunostomy was performed and the patient was discharged later in a good condition.

Case 4:

A 50-year-old man presented on emergency basis with typical clinical and ultrasonic picture of acute calculous cholecystitis. Laparoscopy revealed dense adhesions and the procedure was terminated by partial cholecystectomy without conversion. One week later he was re-admitted with abdominal pain, fever and jaundice due to bile collection, which was drained with a pigtail catheter under ultrasound guidance. ERCP was done because of a large and increasing bilious output. This showed a free leak of dye from the cystic duct, which was managed by endoscopic insertion of a common bile duct stent. The patient did well post-operatively and was discharged home with outpatient follow up for stent removal.

Discussion:

Xanthogranulomatous cholecystitis is characterized macroscopically by yellowish tumour-like masses in the wall of gall bladder. Microscopically, it is characterized in early stages by large number of foamy histiocytes and acute inflammatory cells. This is followed by extensive fibrosis in later stages(1). The disease is seen in a small proportion of cholecystectomy specimens (0.7%-9.0%). It has no gender preference and 70% of the reported cases have been between 40 and 60 years of age(2, 3).

The pathogenesis is not fully understood but it is generally agreed upon that it follows intramural rupture and extravasation of bile and mucin from occluded Rockitansky-Aschoff sinuses. Another possible cause is chronic infection and gallstones with bile stasis(4). Some authors have attributed the process to delayed type hypersensitivity reaction(5). Immunohistochemical studies have demonstrated a closer correlation with bacterial infection in the sub-acute form than in the chronic one(6).

Peroperative findings do not always fit with the preoperative picture. Two of our patients were admitted on an elective basis for routine laparoscopic cholecystectomy, but, because of the dense adhesions, the common bile duct was transected in one case and the common hepatic duct in the second.

Different imaging techniques have been used to facilitate preoperative diagnosis, which is not without difficulties. Ultrasound examination may show, in addition to marked thickening (>3mm) of the gall bladder wall, a focal or diffuse multiple hypo-echoic circular intramural shadows in around 70% of the reported cases(7) (Figure 6). Some centres have good experience in diagnosing the condition by endoscopic ultrasound, which may clearly demonstrate the thickened wall and the nodules(8). CT scan with intravenous contrast may show the same hypo-attenuated areas and bands in the gall bladder wall(9) (Figure 7). CT Scan may be of significant help in peroperative differentiation between xanthogranulomatous cholecystitis and gall bladder cancer(10, 11, 12) (Table 1).

The differentiation is sometimes difficult even peroperatively, and may necessitate frozen section histopathology but it is important because of different treatment strategies(13, 14)(Figure 8).

Figure 6: An ultrasound picture, showing multiple hypo-echoic intramural shadows.

Figure 7: ACT-scan picture, showing hypo-attenuated areas in the gall bladder wall.

Figure 8: ACT-scan picture, showing tumor irregularity of the mucosa and infilteration to the liver.

Magnetic resonance imaging (MRI) may show diffuse wall thickening and viewing high signal foci with signal ovoid lesions(15). Few authors support Fine Needle Aspiration Cytology (FNAC) examination of the gall bladder mass lesions to aid in the preoperative differentiation between xanthogranulomatous cholecystitis and gall bladder cancer(16). As both pathologies may co-exist, sectioning of the whole resected specimen is necessary. CA 19.9 (Carbohydrate Antigen Tumour Marker) is reported to be high in all forms of xanthogranulomatous cholecystitis. This returns to baseline shortly following cholecystectomy whereas it remains high in cases of gall bladder cancer(17).

Was it possible to avoid the troublesome complications in our patients with such a long hospital stay ranging from 16 to 51 days? Was it possible to have an accurate preoperative diagnosis to be more cautious during dissection? The answer is “ Yes, by having a high index of suspicion for the condition”.

Like others, we believe in the importance of preoperative diagnosis. Firstly because xanthogranulomatous cholecystitis may mimic gall bladder cancer both clinically and radiologically (18) and secondly because it is asssociated with a high incidence of operative complications(19). Finally, initial laparoscopic exploration helps in the planning of surgery with an early low threshold of conversion when dense adhesions are seen.

We would rather rely on ultrasonic examination for preoperative diagnosis by making radiologists aware of the ultrasonic criteria of the disease, because it is not practical to send every patient with gallstones for other sophisticated imaging procedures. Ultrasound-guided FNAC examination of gall bladder mass lesions may help in this respect(20).

We conclude that preoperative diagnosis of xanthogranulomatous cholecystitis is important. Because of the high incidence of complications, subtotal cholecystectomy is advisable. Early conversion and frozen section examination are essential when in doubt.

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