Intraductal papillary bile duct adenocarcinoma and gastrointestinal stromal tumor in a case of neurofibromatosis type 1

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Abstract

We report our experience with a synchronous case of gastrointestinal stromal tumor (GIST) and intraductal papillary neoplasm of the bile duct (IPNB) in an elderly woman with neurofibromatosis type 1 (NF-1). A 72-year-old woman presented with a 2-mo history of right upper abdominal pain unrelated to diet and indigestion. Fourteen years earlier, she had been diagnosed with NF-1, which manifested as café au lait spots and multiple nodules on the skin. Computed tomography (CT) revealed a multilocular low-density mass with septation, and mural nodules in the right hepatic lobe, as well as a 1.7-cm-sized well-demarcated enhancing mass in the third portion of the duodenum. The patient subsequently underwent right hepatectomy and duodenal wedge resection. We present here the first report of a case involving a synchronous IPNB and GIST in a patient with NF-1. Our findings demonstrate the possibility of various tumors in NF-1 patients and the importance of diagnosis at an early stage.

Original languageEnglish
Pages (from-to)537-542
Number of pages6
JournalWorld Journal of Gastroenterology
Volume24
Issue number4
DOIs
Publication statusPublished - 2018 Jan 28

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Gastrointestinal Stromal Tumors
Neurofibromatosis 1
Bile Ducts
Adenocarcinoma
Bile Duct Neoplasms
Dyspepsia
Hepatectomy
Duodenum
Abdominal Pain
Tomography
Diet
Skin
Liver
Neoplasms

Keywords

  • Gastrointestinal stromal tumor Synchronous
  • Intraductal papillary neoplasm of the bile duct
  • Neurofibromatosis type 1

ASJC Scopus subject areas

  • Gastroenterology

Cite this

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abstract = "We report our experience with a synchronous case of gastrointestinal stromal tumor (GIST) and intraductal papillary neoplasm of the bile duct (IPNB) in an elderly woman with neurofibromatosis type 1 (NF-1). A 72-year-old woman presented with a 2-mo history of right upper abdominal pain unrelated to diet and indigestion. Fourteen years earlier, she had been diagnosed with NF-1, which manifested as caf{\'e} au lait spots and multiple nodules on the skin. Computed tomography (CT) revealed a multilocular low-density mass with septation, and mural nodules in the right hepatic lobe, as well as a 1.7-cm-sized well-demarcated enhancing mass in the third portion of the duodenum. The patient subsequently underwent right hepatectomy and duodenal wedge resection. We present here the first report of a case involving a synchronous IPNB and GIST in a patient with NF-1. Our findings demonstrate the possibility of various tumors in NF-1 patients and the importance of diagnosis at an early stage.",
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author = "Lee, {Jung Min} and Lee, {Jae Min} and Hyun, {Jong Jin} and Choi, {Hyuk Soon} and Eun-Sun Kim and Bora Keum and Jeen, {Yoon Tae} and Hoon-Jai Chun and Lee, {Hong Sik} and Kim, {Chang Duck} and Dong-Sik Kim and Kim, {Joo Young}",
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AU - Kim, Eun-Sun

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AU - Kim, Chang Duck

AU - Kim, Dong-Sik

AU - Kim, Joo Young

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N2 - We report our experience with a synchronous case of gastrointestinal stromal tumor (GIST) and intraductal papillary neoplasm of the bile duct (IPNB) in an elderly woman with neurofibromatosis type 1 (NF-1). A 72-year-old woman presented with a 2-mo history of right upper abdominal pain unrelated to diet and indigestion. Fourteen years earlier, she had been diagnosed with NF-1, which manifested as café au lait spots and multiple nodules on the skin. Computed tomography (CT) revealed a multilocular low-density mass with septation, and mural nodules in the right hepatic lobe, as well as a 1.7-cm-sized well-demarcated enhancing mass in the third portion of the duodenum. The patient subsequently underwent right hepatectomy and duodenal wedge resection. We present here the first report of a case involving a synchronous IPNB and GIST in a patient with NF-1. Our findings demonstrate the possibility of various tumors in NF-1 patients and the importance of diagnosis at an early stage.

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