Xanthogranulomatous cholecystitis: Diagnostic dilemma and surgical solution in geriatric patients: A case report.

This concerns a 70-year-old patient with a history of pulmonary embolism 3 years ago treated with vitamin K antagonists for one year. The patient initially presented with pain in the right hypochondrium, without associated jaundice, in a context of afebrile state lasting for 2 months. Moreover, there is no history of general condition deterioration. Clinical examination revealed slight tenderness in the right hypochondrium with a palpable gallbladder. Laboratory tests revealed a biological inflammatory syndrome with a high CRP of 100 mg/l and white blood cell count of 15,200 10^9/l, along with disrupted liver function and biological cholestasis ( Table 1 ). The patient underwent an initial abdominal ultrasound, which revealed a distended lithiasic gallbladder with thickened walls and multiple peri-vesicular collections, along with a nonspecific nodule in segment V of the liver. Table 1 Table of main laboratory indicators (first day, 3rd day, 5th day, 10th day). Table 1 First day (admission) 3rd day 5th day 10th day Reference range CRP 100 mg/l 65 mg/l 11 mg/l <8 mg/l <8 mg/l WBC 15,200 10^9/l 14,900 10^9/l 13,400 10^9/l 5000 10^9/l 4000–10,000 10^9/l ASAT 240 UI/l 94 UI/l – 39 UI/l 10–42 UI/l ALAT 74 UI/l 41 UI/l – 8 UI/l 10–60 UI/l PAL 857 UI/l 512 UI/l – 238 UI/l 42–121 UI/l GammaGT 823 UI/l 555 UI/l – 240 UI/l 7–64 UI/l BIL T/BIL C 217/126 μmol/l 76–35 μmol/l – 18/- μmol/l <17 μmol/l CA19–9 461 U/ml – – – <30 U/ml CEA 22,9 ng/ml – – – <5 ng/ml CRP (C-reactive protein); WBC (White blood cells); ASAT (Aspartate Aminotransferase); ALAT (Alanine Aminotransferase); Gamma GT (Gamma-Glutamyl Transferase); PAL (Alkaline phosphatase); BIL T (Total bilirubin); BIL C (Conjugated bilirubin); CEA (Alpha-fetoprotein Converting Enzyme); CA19-9 (Carbohydrate Antigen 19-9). Table of main laboratory indicators (first day, 3rd day, 5th day, 10th day). CRP (C-reactive protein); WBC (White blood cells); ASAT (Aspartate Aminotransferase); ALAT (Alanine Aminotransferase); Gamma GT (Gamma-Glutamyl Transferase); PAL (Alkaline phosphatase); BIL T (Total bilirubin); BIL C (Conjugated bilirubin); CEA (Alpha-fetoprotein Converting Enzyme); CA19-9 (Carbohydrate Antigen 19-9). Given the suspicious appearance of the gallbladder, an abdominal CT scan was performed, confirming the findings from the ultrasound ( Fig. 1 ). Further investigation with MRI demonstrated a distended lithiasic gallbladder with irregular thickening of the gallbladder wall and fat densification around it, suggesting the diagnosis of xanthogranulomatous cholecystitis ( Fig. 2 ). Additionally, there was a nodule in segment V of the liver and thickening of the duodenal bulb ( Fig. 2 ). A scanno-guided biopsy of the liver nodule was performed due to persistent uncertainty between chronic xanthogranulomatous cholecystitis and gallbladder carcinoma with hepatic metastasis. The biopsy ruled out a malignant origin. Fig. 1 Computed tomography imaging showing xanthogranulomatous cholecystitis. Coronal [A] and axial [B] scans of the abdomen display diffuse thickening of the gallbladder wall [yellow arrow], the border between the gallbladder and liver is indistinct. Fig. 1 Fig. 2 T2W axial MRI scan [A, B]. A dilated multilithiasic gallbladder with irregularly thickened wall located within a large collection measuring 100 × 80 mm [white arrow] extending into segment V of the liver with close contact with the first and second duodenum [suspicion of bilio-digestive fistula due to the presence of air]. Fig. 2 Computed tomography imaging showing xanthogranulomatous cholecystitis. Coronal [A] and axial [B] scans of the abdomen display diffuse thickening of the gallbladder wall [yellow arrow], the border between the gallbladder and liver is indistinct. T2W axial MRI scan [A, B]. A dilated multilithiasic gallbladder with irregularly thickened wall located within a large collection measuring 100 × 80 mm [white arrow] extending into segment V of the liver with close contact with the first and second duodenum [suspicion of bilio-digestive fistula due to the presence of air]. The patient underwent surgery via subcostal approach. Intraoperatively, no carcinosis or hepatic metastasis was found. The gallbladder was distended with thickened walls and a fine bile duct, with initial suspicion of a cholecystic-duodenal fistula ( Fig. 3 ). The patient underwent cholecystectomy with catheterization of the cystic duct through a trans-cystic drain and drainage with a subhepatic drain. A cholangiography on postoperative day 5 revealed early duodenal passage, a non-dilated bile duct, and no contrast extravasation ( Fig. 4 ). The trans-cystic drain was clamped on postoperative day 6. Two days post-clamping, a well-directed low-flow bile fistula developed, which spontaneously dried up. The patient was discharged on postoperative day 10 after removal of the subhepatic drain. Histological examination confirmed chronic xanthogranulomatous cholecystitis without any signs of malignancy ( Fig. 5 ). A clinical and radiological follow-up at 6 months postoperatively showed no abnormalities. Fig. 3 Surgical specimen after resection. The gallbladder appears atrophied with diffuse thickening of the wall and strong adhesion to surrounding organs. Fig. 3 Fig. 4 Cholangiography showing a trans-cystic drain in place with good early duodenal passage of contrast agent, without contrast extravasation, no lacunar image observed, with a non-dilated main bile duct and visualization of both right and left hepatic ducts. Fig. 4 Fig. 5 Histological examination of the gallbladder mucosa showing hyperplasia [A], HE ×2.5. The surface epithelial lining is ulcerated, while the submucosa hosts a dense polymorphic inflammatory infiltrate comprising foamy macrophages and giant cells extending throughout the wall [B], HE ×20. The muscular layer appears hypertrophic. No signs of malignancy are observed. Fig. 5 Surgical specimen after resection. The gallbladder appears atrophied with diffuse thickening of the wall and strong adhesion to surrounding organs. Cholangiography showing a trans-cystic drain in place with good early duodenal passage of contrast agent, without contrast extravasation, no lacunar image observed, with a non-dilated main bile duct and visualization of both right and left hepatic ducts. Histological examination of the gallbladder mucosa showing hyperplasia [A], HE ×2.5. The surface epithelial lining is ulcerated, while the submucosa hosts a dense polymorphic inflammatory infiltrate comprising foamy macrophages and giant cells extending throughout the wall [B], HE ×20. The muscular layer appears hypertrophic. No signs of malignancy are observed.

The author[s] declared that no grants were involved in supporting this work.

Please specify the contribution of each author to the paper, e.g. study concept or design, data collection, data analysis or interpretation, writing the paper, others, who have contributed in other ways, should be listed as contributors. TYA and HG contributed to manuscript writing, editing and data collection; HB and AC contributed to data analysis; KT contributed to data interpretation; MBK contributed to conceptualization and supervision; All authors have read and approved the final manuscript.

Ethical approval is not applicable/waived at our institution. Due to the specific nature of case reports, which involve detailed descriptions of observations and interventions that have already been conducted on patients, as opposed to prospective studies involving planned interventions, our institution does not require formal ethical approval for such cases. We recognize the importance of ethics in medical research and are fully committed to upholding ethical standards in our medical and research practices.

This research did not receive funding from any specific grant provided by public, commercial, or not-for-profit organizations.

Written informed consent was obtained from the patient for the publication of this case report and its accompanying images. A copy of the written consent is available for the Editor-in-Chief of this journal to review upon request.

N/A

Dr. Taha Yassine AYADI

Managing Xanthogranulomatous Cholecystitis (XGC) poses significant diagnostic and therapeutic challenges due to its rare nature and potential resemblance to gallbladder carcinoma (GBC) [ 4 , 6 ]. This case emphasizes the importance of a multidisciplinary approach involving surgeons, radiologists, and pathologists in the management of XGC. Additional studies on a larger scale are imperative to refine diagnostic methods and therapeutic strategies for XGC, ultimately improving patient outcomes and treatment efficacy.

Xanthogranulomatous cholecystitis (XGC) is a rare inflammatory disease of the gallbladder characterized by severe proliferation of fibrotic tissue accompanied by the accumulation of lipid-laden macrophages and acute and chronic inflammatory cells [ 1 ]. Despite being benign, XGC often extends into neighboring organs, and its macroscopic appearance may be mistaken for gallbladder cancer [ 1 , 5 ]. XGC affects both sexes without significant predominance [ 5 ]. The average age of operated patients is 53 years [range: 49 to 62 years], but exceptionally, XGC can occur in children or adolescents [ 6 ]. Clinical symptoms of XGC are usually nonspecific, presenting as either acute or chronic cholecystitis [ 7 ]. Between a third and three-quarters of XGC cases are diagnosed in an emergency setting [ 8 ]. In the context of chronic gallbladder inflammation and episodes resembling cholecystitis, common findings include fever and recurrent abdominal pain over an average of 11 months [ 8 ]. Serological evaluation remains nonspecific and may not aid in the diagnosis of XGC. Liver function derangement or inflammatory marker elevation [white cell count or C-reactive protein] may correspond with episodes of XGC [ 9 ]. Additionally, tumor markers such as CA19.9, although correlating well with the incidence of cholangiocarcinoma, may be elevated in isolated XGC cases, providing no diagnostic certainty [ 9 ]. A literature review identified 111 cases with recorded CA19-9 values, with almost two-thirds (63 %] showing elevated levels [ 9 ]. The imaging characteristics of XGC closely resemble those of gallbladder cancer, including thickening of the gallbladder wall and involvement of neighboring organs [ 3 , 9 ]. Ultrasonography reveals thickening of the gallbladder wall, which can be focal or diffuse, accompanied by gallbladder stones and intramural hypoechoic nodules [ 5 ]. CT examinations have also highlighted these findings, aiding in differentiating XGC from gallbladder cancer [ 10 ]. More precisely, the CT diagnostic criteria for XGC encompass diffuse thickening of the gallbladder wall, continuity of the mucosal line, presence of intramural hypoattenuated nodules, and absence of invasion into adjacent liver parenchyma [ 5 , 11 ]. In MRI, the intramural T2 high signal intensity serves as a key feature for diagnosing XGC and is valuable in distinguishing it from gallbladder cancer [ 11 ]. The regions showing hyperintensity on T2-weighted images correlate with areas rich in xanthogranulomas [ 11 ]. However, in the absence of enhancement, these regions may also indicate necrosis and/or abscesses [ 11 ]. Surgery is the definitive treatment for XGC [ 12 ]. Simple cholecystectomy is usually sufficient [ 3 ]. XGC can destroy the gallbladder border, produce dense fibrotic adhesions between surrounding organs, and result in fistula formation with adjacent structures, mimicking gallbladder cancer [ 6 ]. Therefore, the possibility of gallbladder cancer should be excluded during surgery [ 6 ]. However, intraoperative differentiation between XGC and GB carcinoma remains challenging, especially in cases of tumor formation and adhesions to adjacent organs [ 12 ]. Intraoperative frozen section analysis is useful in distinguishing between XGC and gallbladder cancer, helping to avoid unnecessarily extended surgery [ 1 ]. Given the possibility of coexistence between GBC and XGC, radical resection, such as liver resection, may be justified when malignancy cannot be completely excluded [ 6 ]. Laparoscopic cholecystectomy, the gold standard for treating benign gallbladder diseases, often proves challenging in cases of XGC [ 12 ]. XGC can lead to chronic inflammation, compromising the integrity of the gallbladder wall, causing border irregularities, advanced fibrotic adhesions to surrounding structures, and biliary fistulas [ 12 ]. As a result, some studies suggest that the laparoscopic approach may not be suitable for XGC [ 13 , 14 ]. The frequency of conversion to open surgery is higher in patients with XGC than in those with other forms of cholecystitis, with a conversion rate ranging between 10 % and 80 % [ 12 , 14 ]. Many studies recommend open cholecystectomy when there is a suspicion of malignancy or when severe adhesions are present [ 14 ]. In their study, Qasaimeh et al. opted for direct open cholecystectomy in seven patients with preoperative tumor suspicion [ 15 ]. They reported a 31.4 % conversion rate in cases that began laparoscopically [ 15 ]. Additionally, the same study noted that the incidence of cholecystectomy conversion was ten times higher in XGC cases compared to other cholecystectomy procedures in their series [ 14 , 15 ]. The high rate of cholecystectomy conversions was attributed to the difficulty in identifying the anatomy and controlling bleeding [ 13 , 14 ].

AI tools were not used for the elaboration of the manuscript.

Xanthogranulomatous cholecystitis (XGC) is a rare inflammation of the gallbladder characterized by the presence of xanthogranulomas, which are yellow structures within the gallbladder wall often associated with gallstones [ 1 ]. In some cases, XGC can resemble gallbladder carcinoma (GBC), especially when it takes on a pseudo-tumoral form and affects nearby organs [ 1 ]. The development of XGC involves inflammation that damages the Rokitansky-Aschoff sinuses in the gallbladder wall, leading to an intense inflammatory reaction [ 1 ]. The actual incidence of XGC is unknown, ranging from 0.7 % to 13.2 % of all inflammatory pathologies of the gallbladder [ 2 ]. Symptoms of XGC are nonspecific and can resemble those of acute or chronic cholecystitis [ 3 ]. In individuals with underlying health issues, the presence of XGC may raise concerns for GBC [ 3 ]. XGC can also occur suddenly as a complication of gallstones, particularly when they migrate [ 3 ]. While ultrasound and CT scans can provide suggestive evidence of XGC, magnetic resonance imaging (MRI) offers more specific diagnostic information [ 4 ]. In challenging cases, a biopsy may be necessary to definitively rule out cancer [ 3 ]. If GBC is ruled out, laparoscopic cholecystectomy is typically recommended as the treatment of choice for XGC, despite the risk of complications [ 3 ]. This case report aims to evaluate the clinical and radiological features of XGC and its surgical outcomes to determine the most appropriate treatment approaches. This work has been reported in line with the SCARE 2023 criteria [ 5 ].

No conflicts of interest.