Unexpected metastasis of intraductal papillary neoplasm of the bile duct without an invasive component to the brain and lungs: A case report

Nam, Nguyen Hai; Taura, Kojiro; Kanai, Masashi; Fukuyama, Keita; Uza, Norimitsu; Maeda, Hatsuo; Yutaka, Yojiro; Chen‐Yoshikawa, Toyofumi F.; Muto, Manabu; Üemoto, Shinji

doi:10.3748/wjg.v26.i3.366

Cited by 2 publications

(5 citation statements)

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“…The diagnosis in this case was based on hematoxylin and eosin staining and immunohistological staining. To the best of our knowledge, ours is only the third reported case of lung metastasis from IPNB, with the two other cases being described in a single-center survey by Höhn et al [ 6 ] and in another case report [ 7 ]. TTF-1 is useful to distinguish primary lung adenocarcinoma from metastatic adenocarcinoma.…”

Section: Discussionmentioning

confidence: 91%

“…However, we treated our patient using adjuvant chemotherapy after the lung resections, based on his history of recurrence and metastasis. The two other reports did not describe the outcomes after resection of the lung metastases [ 6 , 7 ], although both reports indicated that the diagnosis of metastasis was based on a comparison of the immunostaining results from the primary and lung specimens. Thus, the long-term outcomes of resection remain unclear, although our patient has achieved good survival to this point.…”

Section: Discussionmentioning

confidence: 99%

“…In one of the previous cases, the preoperative suspicion was that the patient had lung cancer because non-invasive IPNB without lymph node metastasis is extremely rare; however, after the genomic analysis of the resected specimen, IPNB metastasis was confirmed. Thus, the authors suggested that the mechanism underlying metastasis is actually more complex than what was previously thought [ 7 ]. Performance status and age may be relevant when considering the resection of IPNB recurrence and/or metastasis; using appropriate surgical technique and careful case selection will not only allow better determination of long-term outcomes but also help to clarify the mechanism underlying metastasis by analysis of surgical specimens.…”

Section: Discussionmentioning

confidence: 99%

“…We are only aware of a few case reports regarding metastatic IPNB because it has a relatively benign course [ 7 , 16 ]. Most of these reports have involved small retrospective studies, although Gordon-Weeks et al reported a systematic review and meta-analysis in 2016 [ 2 ].…”

Section: Discussionmentioning

confidence: 99%

“…You et al reported 74 cases with invasive IPNB [ 5 ] and noted that local recurrence was the most common first recurrence site, with distal metastasis potentially involving the liver, peritoneum, and retroperitoneal lymph nodes. However, we are only aware of two previous cases involving lung metastasis from IPNB, which were reported by Höhn et al in a single-center survey [ 6 ] and another case report [ 7 ]. Thus, because it is unclear what treatment is appropriate for IPNB recurrence and/or metastasis, we report what we believe is the third case of lung metastasis from IPNB.…”

Section: Introductionmentioning

confidence: 99%

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Lung metastases from intraductal papillary neoplasm of the bile duct: a case report

et al. 2020

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Background Intraductal papillary neoplasm of the bile duct (IPNB) is considered a pre-cancerous biliary lesion and/or an early cancer lesion, although its classification remains unclear. The 2019 revised edition of the World Health Organization Classification of Tumors of the Digestive System proposed type 1 and type 2 as new classification categories, and meta-analyses and/or multi-center cohort studies are beginning to be reported. However, treatment for IPNB recurrence and metastasis remains unclear. Case presentation A 60-year-old man who was referred to our hospital after a suspected liver tumor was diagnosed using abdominal ultrasonography. Imaging findings revealed an irregularly shaped tumor in segment 5 (S5) of the liver (size 20 mm). The S5 lesion was suspected as IPNB, and segmentectomy was performed. The pathological findings revealed invasive carcinoma derived from IPNB, and immunohistochemistry revealed positive expression of MUC1, MUC5AC, and MUC6, but negative expression of CDX2 and MUC2. At 9 months after the surgery, computed tomography revealed a tumor in the right bile duct, which was diagnosed as liver recurrence of IPNB, and right hepatectomy was performed. The histopathological findings were the same as for the first resected specimen (i.e., IPNB). At 45 months after the second surgery, computed tomography revealed nodules in both lungs, which were diagnosed as lung metastases from IPNB and resected in two separate procedures. The pathological findings were metastatic carcinoma from IPNB for both lung lesions. The patient is currently alive and undergoing adjuvant chemotherapy (S-1), which was initiated 64 months after the first resection and 12 months after resection of the lung metastases. Conclusion We encountered a rare case of lung metastases from IPNB, which were diagnosed immunohistologically. Because IPNB is generally a slow-growing tumor, resection may be feasible for IPNB recurrence and/or metastasis, which may be detected during long-term follow-up. Thus, even if resection is performed for primary IPNB, additional surgical treatment may be feasible in this setting.

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Section: Discussionmentioning

confidence: 91%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Lung metastases from intraductal papillary neoplasm of the bile duct: a case report

et al. 2020

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Surgical Treatment of Intraductal Papillary Neoplasm of the Bile Duct: A Report of Two Cases and Review of the Literature

Liu

Zhang

et al. 2022

Front. Oncol.

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Intraductal papillary neoplasm of the bile duct (IPNB) is a rare bile duct tumor characterized by intraductal papillary or villous neoplasms covered by neoplastic epithelium with fine fibrovascular stalks in the dilated bile ducts (1). Its true etiology remains unknown. Herein, we report two cases of IPNB that underwent surgical resection. The first case was a 66-year-old male who complained of upper abdominal pain for three years. We found obstruction of the common bile duct and dilation of the intrahepatic and extrahepatic bile ducts after MRCP. Laparoscopic hepatic segmentectomy (S2, S3, S4), resection of the common bile duct, cholecystectomy, and hepaticojejunostomy were performed. The second case was a 67-year-old male with asymptomatic dilation of the intrahepatic duct. The patient underwent robot-assisted laparoscopic hepatic segmentectomy (S5, S6, S7, S8), resection of the common bile duct, hepaticojejunostomy and cholecystectomy.

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Unexpected metastasis of intraductal papillary neoplasm of the bile duct without an invasive component to the brain and lungs: A case report

Cited by 2 publications

References 10 publications

Lung metastases from intraductal papillary neoplasm of the bile duct: a case report

Lung metastases from intraductal papillary neoplasm of the bile duct: a case report

Surgical Treatment of Intraductal Papillary Neoplasm of the Bile Duct: A Report of Two Cases and Review of the Literature

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