==== Front Case Rep Surg Case Rep Surg CRIS Case Reports in Surgery 2090-6900 2090-6919 Hindawi 10.1155/2023/5825045 Case Report Extensively Invasive Gallbladder Cancer from Intracholecystic Papillary Neoplasm Treated with Pylorus-Preserving Pancreaticoduodenectomy and Extended Cholecystectomy: A Case Report and Literature Review https://orcid.org/0000-0001-5374-9080 Kumagai Hideki hishioka@iwate-med.ac.jp 1 https://orcid.org/0000-0003-4181-7159 Umemura Akira 1 Nitta Hiroyuki 1 Katagiri Hirokatsu 1 Nishiya Masao 2 Uesugi Noriyuki 2 Sugai Tamotsu 2 Sasaki Akira 1 1Department of Surgery, School of Medicine, Iwate Medical University, 2-1-1 Idaidori, Yahaba, Iwate 028-3695, Japan 2Department of Molecular Diagnostic Pathology, Iwate Medical University, 2-1-1 Idaidori, Yahaba, Iwate 028-3695, Japan Academic Editor: Wei-bo Chen 2023 24 6 2023 2023 58250459 11 2022 26 4 2023 13 6 2023 Copyright © 2023 Hideki Kumagai et al. 2023 https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Background Intracholecystic papillary neoplasm (ICPN) is a rare tumor first classified by the World Health Organization in 2010. ICPN is a counterpart of the intraductal papillary mucinous neoplasm of the pancreas and intraductal papillary neoplasm of the bile duct. Previous reports on ICPN are limited; thus, the diagnosis, surgical intervention, and prognosis are controversial. Here, we report an extensively invasive gallbladder cancer arising in ICPN treated with pylorus-preserving pancreaticoduodenectomy (PPPD) and extended cholecystectomy. Case Presentation. A 75-year-old man presented to another hospital with jaundice for 1 month. Laboratory findings showed elevated total bilirubin, 10.6 mg/dL and carbohydrate antigen 19-9, 54.8 U/mL. Computed tomography showed a well-enhanced tumor located in the distal bile duct and dilated hepatic bile duct. The gallbladder wall was thickened and homogeneously enhanced. Endoscopic retrograde cholangiopancreatography revealed a filling defect in the distal common bile duct, and intraductal ultrasonography showed a papillary tumor in the common bile duct, indicating tumor invasion of the bile duct subserosa. Subsequent bile duct brush cytology revealed adenocarcinoma. The patient was referred to our hospital for surgical treatment and underwent an open PPPD. Intraoperative findings showed a thickened and indurated gallbladder wall, suggesting concurrent gallbladder cancer; thus, the patient subsequently underwent PPPD and extended cholecystectomy. Histopathological findings confirmed gallbladder carcinoma originating from ICPN, which extensively invaded the liver, common bile duct, and pancreas. The patient started adjuvant chemotherapy (tegafur/gimeracil/oteracil) 1 month after surgery and had no recurrence at follow-up after 1 year. Conclusions Accurate preoperative diagnosis of ICPN, including the extent of tumor invasion is challenging. To ensure complete curability, the development of an optimal surgical strategy considering preoperative examinations and intraoperative findings is essential. ==== Body pmc1. Introduction Intracholecystic papillary neoplasm (ICPN) is a relatively new concept established by the 2010 World Health Organization (WHO) classification [1]. According to this classification, ICPN is recognized as a counterpart of intraductal papillary mucinous neoplasm in the pancreas and intraductal papillary neoplasm of the bile duct [1]. Tumors are considered premalignant lesions [2, 3]. ICPN is rare, accounting for 0.4–1.5% of cholecystectomies and 6.4% of gallbladder cancers [1–3]. Therefore, there are limited previous studies on the diagnosis or surgical management of ICPN, and the prognosis of ICPN remains controversial. Here, we report a rare case of extensively invasive gallbladder cancer arising in ICPN treated with pylorus-preserving pancreaticoduodenectomy (PPPD) and extended cholecystectomy and review the previous literature. 2. Case Presentation A 75-year-old man presented to the hospital with jaundice that had been present for 1 month. He had a medical history of hypertension and non-tuberculosis mycobacterial infection but no surgical history. Laboratory findings showed elevated levels of serum bilirubin and liver enzymes: total bilirubin, 10.6 mg/dL; aspartate aminotransferase, 68 IU/L; alanine aminotransferase, 119 IU/L; and gamma-glutamyl transpeptidase, 393 IU/L. Serum carbohydrate antigen 19-9 was also elevated (54.8 U/mL), though carcinoembryonic antigen was within the normal range. Enhanced computed tomography (CT) revealed a well-enhanced tumor in the distal bile duct and dilation of the hepatic bile duct (Figure 1(a)). In addition, the gallbladder mucosa was thickened, with a homogeneous contrast effect (Figure 1(b)). Magnetic resonance imaging (MRI) showed a low T2 signal tumor in the common bile duct (Figure 2(a)), and magnetic resonance cholangiopancreatography (MRCP) showed dilation of the hepatic-sided bile duct from the tumor (Figure 2(b)). The thickened gallbladder walls had a homogeneous low T2 signal; however, liver invasion by the tumor was not significant. Endoscopic retrograde cholangiopancreatography (ERCP) revealed a filling defect in the distal bile duct, and the cystic duct was invisible (Figure 3(a)). Moreover, intraductal ultrasonography (IDUS) showed a papillary tumor in the common bile duct and indicated invasion of the bile duct subserosa (Figures 3(b) and 3(c)). Subsequent bile duct brushing cytology on ERCP and IDUS revealed adenocarcinoma, and the patient was diagnosed with distal bile duct cancer associated with adenomyomatosis of the gallbladder. The patient was referred to our hospital for surgical treatment, and we planned to perform PPPD for distal bile duct cancer with curative intent. Intraoperative findings revealed a thickened and indurated gallbladder wall, suggesting the coexistence of gallbladder cancer; thus, we performed PPPD and extended cholecystectomy. Intraoperative frozen-section analysis of the cut end of the hepatic bile duct was negative for the tumor. A macroscopic examination of the resected specimen revealed a papillary tumor that had extensively invaded the liver, cystic duct, bile duct, and pancreas (Figures 4(a) and 4(b)). Permanent histopathological findings indicated that the tumor was papillotubulary and had broad-based growth of columnar cells with mucus production in the gallbladder (Figures 5(a) and 5(b)). In addition, metastasis to the lymph nodes of the hepatoduodenal ligament was observed. Immunohistochemical analysis revealed that the tumor cells were positive for mucin (MUC)1, MUC5AC, and MUC6, but negative for MUC2 and p53 (Figures 5(c), 5(d), and 5(e)). Finally, the diagnosis of gastric-type ICPN was established according to the 2010 WHO classification. The postoperative course was uneventful, and the patient started adjuvant chemotherapy (tegafur/gimeracil/oteracil) 1 month after surgery. At the follow-up after 1 year, the patient had no recurrence. 3. Discussion We report the case of a patient with extensively invasive gallbladder cancer originating in ICPN treated with PPPD and extended cholecystectomy. Adsay et al. reported that invasiveness was observed in 55% of ICPN cases [1]; however, most patients with ICPN are found at an early stage incidentally by imaging studies, as mentioned below, and reports on advanced cases are limited [4–7]. We searched for previous reports on ICPN in PubMed using the keywords “intracholecystic papillary neoplasm” or “intracystic papillary neoplasm” and reviewed 39 cases [4–39], including the present case diagnosed as ICPN histopathologically (Table 1). The mean age of patients with ICPN was 66.3 years, and female patients outnumbered male patients, as previously reported [3]. Approximately half of the ICPNs have invasive components, as found by Adsay et al., however, our patient was the only patient with lymph node metastasis in our review. Moreover, Adsay et al. reported that approximately half of the ICPNs develop in the right upper abdominal region, and the other half are incidentally found by imaging studies [1], which is similar to our findings summarized in Table 1. Conversely, jaundice is an uncommon symptom in ICPN, and there are few previous reports in the literature [4, 5, 25, 27]. Of the four patients, two patients suffered from jaundice resulting from a protruding tumor from the gallbladder to the common bile duct [4, 5]. Interestingly, the other two developed jaundice due to mucus production from the tumor [25, 27]. In the present case, histopathological findings revealed that the tumor had extensively invaded the bile duct; thus, the patient had obstructive jaundice owing to tumor invasion of the common bile duct rather than a protruding tumor from the gallbladder. Protruding or advanced tumors associated with ICPN can cause obstructive jaundice; moreover, mucus production from ICPN can lead to jaundice. Distinguishing between ICPN and other gallbladder tumors using imaging studies is difficult. In our review, only 15.8% of patients with ICPN were diagnosed accurately before surgery. According to previous reports, ICPN is well-defined on enhanced CT and presents high or low T2 signal intensity and high diffusion-weighted imaging signal intensity on MRI [4, 7]. Fluorodeoxyglucose (FDG) accumulation in ICPN has been observed on FDG-positron emission tomography [5, 28]. However, these are non-specific findings that can be observed in other gallbladder tumors. Moreover, histopathological examinations, including cytology and biopsy are not diagnostic in terms of distinguishing between ICPN and other types of gallbladder carcinomas [4–7]. However, endoscopic ultrasound (EUS), including IDUS or peroral cholangioscopy (POCS) has shown the presence of a papillary tumor in most patients diagnosed with ICPN preoperatively [4–6, 25, 27]. EUS and POCS may provide a better definition of ICPN compared with other imaging modalities. In the present case, IDUS revealed a papillary tumor in the common bile duct. Therefore, clinicians should be familiar with ICPN, and make an effort to accurately diagnose it using multiple imaging techniques. The treatment for ICPN is oncological resection; however, the selection of the optimal surgical procedure is often challenging. Simple cholecystectomy is sufficient for ICPN limited to the gallbladder mucosa without invasion. However, approximately half of the ICPN cases have an invasive component [1]. Moreover, some patients have ICPNs suspected of common bile duct invasion due to a protruding tumor from the gallbladder to the common bile duct [4, 5]. In the present case, CT, MRI, and ERCP findings indicated that the tumor was located in the distal bile duct, and IDUS suggested that the tumor had invaded the subserosa of the bile duct; therefore, we decided to perform PPPD. Moreover, intraoperative findings showed a thickened and indurated gallbladder wall, suggesting advanced gallbladder carcinoma; thus, we performed an extended cholecystectomy in addition to PPPD. Intraoperative frozen-section analysis of the cut end of the hepatic-sided bile duct confirmed no evidence of a tumor. To select the optimal surgical procedure, a comprehensive evaluation that considers preoperative imaging studies and intraoperative findings is essential. Notably, EUS, including IDUS, can be a useful tool for assessing tumor extension of ICPN. Some studies have reported that ICPN with or without invasive carcinoma has a good prognosis, in contrast to other types of gallbladder carcinoma [1, 40, 41]. Adsay et al. reported that the 1-, 3-, and 5-year overall survival rates of non-invasive ICPN were 90%, 90%, and 78%, respectively [1]. In addition, the percentages of invasive ICPN were 69%, 60%, and 60%, respectively [1]. These overall survival rates are much better than those of other types of gallbladder carcinomas, which have an 18–30% 5-year survival rate [1, 42]. In contrast, a recent study reported that in a stage-matching analysis of gallbladder carcinoma, there was no difference between the prognosis of invasive carcinoma and other types of gallbladder carcinoma [43]. In our review, most ICPN patients had a good prognosis. We speculate that this was due to most ICPNs being resected at an early stage. However, our patient had advanced cancer originating from an ICPN with lymph node metastasis. Therefore, our patient was closely followed up with adjuvant chemotherapy. The optimal choice of surgical procedure, including extended cholecystectomy, bile duct resection, and pancreaticoduodenectomy is essential for achieving complete oncological resection of the tumor. In addition, close postoperative follow-up is crucial for patients with ICPN, especially those with advanced cancer arising from the tumor, in accordance with other types of gallbladder carcinoma. 4. Conclusions Accurate preoperative diagnosis of ICPN, including the extent of tumor invasion, is challenging; however, both EUS and POCS are effective tools for resolving these challenges. ICPN has been recognized as a tumor with a better prognosis compared with other types of gallbladder carcinoma; however, a recent study reported that the prognosis of these tumors is equivalent. The optimal choice of surgical procedure and close postoperative follow-up are essential for patients with ICPN, especially those with advanced cancer arising from the tumor. Data Availability Data supporting this research article are available from the corresponding author or first author upon reasonable request. Consent Written informed consent was obtained from the patient for publication of the case details. Conflicts of Interest The author(s) declare(s) that they have no conflicts of interest. Figure 1 Enhanced CT findings. (a) A well-defined tumor from a cystic duct to a distal bile duct was observed (yellow arrow), and a hepatic-sided bile duct seen from the tumor was dilated. (b) Gallbladder mucosa was well-contrasted, and the wall was thickened (yellow arrow). CT: computed tomography. Figure 2 MRI and MRCP findings. (a) The tumor located in the distal bile duct had a low T2 signal (yellow arrow). (b) MRCP showed a filling defect of the distal bile duct and dilation of the hepatic-sided bile duct seen from the tumor (yellow arrow). MRCP: magnetic resonance cholangiopancreatography; MRI: magnetic resonance imaging. Figure 3 ERCP and IDUS findings. (a) ERCP showed a filling defect of the distal bile duct; the cystic duct was invisible. (b) IDUS showed that the tumor had invaded the bile duct subserosa (yellow arrow). (c) The patient's distal bile duct was almost obstructed by the tumor. ERCP: endoscopic retrograde cholangiopancreatography; IDUS: intraductal ultrasonography. Figure 4 Macroscopic findings. (a) Macroscopic findings revealed a papillary tumor that had invaded the common bile duct. The distal bile duct lumen was severely constricted by the tumor invasion (yellow arrow). (b) Cross section of the resected specimen revealed that the white tumor extensively invaded the liver (white arrow), cystic duct (red arrow), common bile duct (yellow arrow), and pancreas (pink arrow). Black arrows point to the papilla of Vater. Figure 5 Histopathological images showed that the tumor was papillotubulary and had broad-based growth of columnar cells with mucus production in the gallbladder; hematoxylin and eosin staining: (a) 20× and (b) 100×. Immunohistochemical analysis showed the tumor cells were positive for mucin (MUC)5AC staining (c), but negative for MUC2 (d) and p53 (e) (all 100×). Table 1 Overview of the recent literature on ICPN. Case First author Age (year) Gender Symptom Imaging findings Preoperative diagnosis Surgical procedure Invasive component Lymph node metastasis Adjuvant therapy Outcome 1 Sato [8] 77 Female Epigastric pain Gallbladder tumor Not mentioned SC Yes No No Alive 10 months 2 Dettoni [9] 62 Female Abdominal pain Gallbladder tumor Gallbladder cancer EC Yes No No Alive 29 months 3 Meguro [10] 54 Female Epigastric pain Papillary tumor Gallbladder tumor EC, extrahepatic bile duct resection, and choledochojejunostomy Yes (complicated with mixed adenoneuroendocrine carcinoma) No No Alive 24 months 4 Hashimoto [4] 58 Female Jaundice Papillary tumor Gallbladder cancer EC, PD Yes No No Recurrence 16 months after surgery 5 Michalinos [11] 28 Female Epigastric pain Dilation of common bile duct Choledochal cyst SC, extrahepatic bile duct resection, and choledochojejunostomy No No No Not mentioned 6 Sato [12] 64 Male Epigastric pain Cystic tumor Not mentioned SC Yes No No Alive 8 months 7 Páez Cumpa [13] 39 Male Epigastric pain Gallbladder tumor Not mentioned SC No No No Alive 6 months 8 Unno [14] 74 Male No Papillary tumor Gallbladder cancer EC Yes No No Alive 3 months 9 Mizobuchi [15] 74 Female No Papillary tumor Gallbladder cancer EC No No No Not mentioned 10 Mizobuchi [15] 61 Female No Papillary tumor Gallbladder cancer EC Not mentioned No No Not mentioned 11 Mizobuchi [15] 83 Male No Papillary tumor Not mentioned EC Not mentioned No No Not mentioned 12 Sakai [5] 74 Male Jaundice Papillary tumor Gallbladder cancer EC, extrahepatic bile duct resection, and choledochojejunostomy Yes No No Not mentioned 13 Muranushi [16] 70 Male No Gallbladder tumor Gallbladder cancer EC No No No Alive 42 months 14 Hara [17] 71 Male No Papillary tumor ICPN SC No No No Not mentioned 15 Fujii [6] 59 Male Fatigue Papillary tumor ICPN SSPPD after SC Yes No Tegafur/gimeracil/oteracil Alive 2 months 16 Yokode [18] 58 Female Fever Papillary tumor ICPN EC and SSPPD Yes No No Alive 6 months 17 Sarıtaş [19] 52 Female Right upper quadrant pain Papillary tumor Gallbladder tumor SC No No No Not mentioned 18 Park [20] 78 Female Epigastric pain Gallbladder tumor Not mentioned SC Yes (complicated with angiosarcoma) No No Alive 3 months 19 Fraga [21] 85 Female Epigastric pain Gallbladder tumor Not mentioned EC Yes (complicated with NEC) No No Death 3 months after surgery 20 Sciarra [22] 66 Female Abdominal pain Papillary tumor Not mentioned EC Yes (complicated with MiNEN) No No Alive 5 months 21 Oh [23] 79 Female Abdominal pain Papillary tumor ICPN SC Yes No No Alive 36 months 22 Iwasaki [24] 52 Female No Papillary tumor Gallbladder cancer EC, extrahepatic bile duct resection, and choledochojejunostomy No No No Alive 5 months 23 Oba [25] 78 Female Jaundice and epigastric pain Papillary tumor Gallbladder tumor SC Yes No No Alive 12 months 24 Logrado [26] 71 Female Epigastric pain Wall thickness of the gallbladder Not mentioned SC No No No Alive 30 months 25 Iseki [7] 83 Male No Papillary tumor Distal bile duct cancer SSPPD Yes N0 No Alive 20 months 26 Kuniyoshi [27] 86 Female Jaundice Papillary tumor ICPN SC No No No Alive 12 months 27 Ismail [28] 48 Female No Gallbladder tumor Gallbladder tumor SC No No No Not mentioned 28 Aida [29] 65 Female No Papillary tumor Gallbladder cancer EC No (complicated with xanthogranulomatous cholecystitis) No No Alive 3 months 29 Dörr [30] 77 Female No Wall thickness of the gallbladder Chronic cholecystitis SC Yes No No Not mentioned 30 Shimada [31] 69 Male No Papillary tumor Gallbladder cancer EC Yes No No Alive 12 months 31 Wong [32] 49 Male No Gallbladder tumor Not mentioned SC No No No Not mentioned 32 Trisal [33] 48 Male Epigastric pain Wall thickness of the gallbladder Not mentioned SC No (complicated with xanthogranulomatous cholecystitis) No No Alive 6 months 33 Watanabe [34] 79 Female No Gallbladder tumor Gallbladder cancer EC after subtotal cholecystectomy Yes No No Alive 8 months 34 Oishi [35] 60s Female No Papillary tumor ICPN EC and resection of the extrahepatic bile duct No No No Alive 24 months 35 Limaiem [36] 76 Male Right upper quadrant pain Papillary tumor Not mentioned EC Yes No No Not mentioned 36 Scarola [37] 80 Male Fatigue Gallbladder tumor Not mentioned EC Yes No No Alive 6 months 37 Mruthyunjayappa [38] 70s Male Right upper quadrant pain Gallbladder tumor Gallbladder cancer EC Yes (complicated with NEC) No No Death 50 months after surgery 38 Koike [39] 44 Male No Gallbladder tumor Cholesterol polyp or pyloric-type adenoma SC No No No Not mentioned 39 Our case 75 Male Jaundice Papillary tumor Distal bile duct cancer PPPD and EC Yes Yes Tegafur/gimeracil/oteracil Alive 12 months Summary Age Male No symptom Papillary tumor ICPN SC Yes Yes Tegafur/gimeracil/oteracil Follow-up duration, 66.3 17 17 (43.6) 22 (56.4) 6 (15.4) 14 (35.9) 22 1 2 Months (28–86) Female Abdominal pain Gallbladder tumor Gallbladder cancer EC No No No 14.2 (2–50) 22 16 (41.0) 12 (30.8) 13 (33.3) 21 (53.8) 17 38 37 Alive Jaundice Wall thickness of the gallbladder Gallbladder tumor Resection of the extrahepatic bile duct 24 5 (12.8) 3 (7.8) 4 (10.3) Recurrence 5 (12.8) 3 PD Death 5 (12.8) 2 Data are expressed as the mean (range) or number (%). 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