Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report

Abstract Background Peutz–Jeghers syndrome (PJS) is an autosomal dominant disorder characterized by mucocutaneous pigmentation and hamartomatous gastrointestinal polyposis. It is well known that individuals with PJS are at an increased risk of cancer in a variety of organs. Case presentation Here, w...

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Main Authors: Toru Yoshikawa, Tomoyuki Abe, Hironobu Amano, Keiji Hanada, Tomoyuki Minami, Tsuyoshi Kobayashi, Shuji Yonehara, Masahiro Nakahara, Hideki Ohdan, Toshio Noriyuki
Format: Article
Language:English
Published: Japan Surgical Society 2018-08-01
Series:Surgical Case Reports
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Online Access:http://link.springer.com/article/10.1186/s40792-018-0492-6
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author Toru Yoshikawa
Tomoyuki Abe
Hironobu Amano
Keiji Hanada
Tomoyuki Minami
Tsuyoshi Kobayashi
Shuji Yonehara
Masahiro Nakahara
Hideki Ohdan
Toshio Noriyuki
author_facet Toru Yoshikawa
Tomoyuki Abe
Hironobu Amano
Keiji Hanada
Tomoyuki Minami
Tsuyoshi Kobayashi
Shuji Yonehara
Masahiro Nakahara
Hideki Ohdan
Toshio Noriyuki
author_sort Toru Yoshikawa
collection DOAJ
description Abstract Background Peutz–Jeghers syndrome (PJS) is an autosomal dominant disorder characterized by mucocutaneous pigmentation and hamartomatous gastrointestinal polyposis. It is well known that individuals with PJS are at an increased risk of cancer in a variety of organs. Case presentation Here, we present a patient with PJS who achieved long-term survival by undergoing repeat curative surgery for metachronous triple cancer. Her medical history included hilar cholangiocarcinoma and cervical carcinoma; curative surgery was performed for both conditions. On annual follow-up, the level of carcinoembryonic antigen was elevated at 6.9 ng/ml. Enhanced computed tomography revealed a cystic tumor consisting of mural nodules at the pancreatic head; the maximal diameter was 15 mm. Magnetic resonance imaging clearly demonstrated the tumor with low intensity on T1-weighted images and high intensity on T2-weighted images. Endoscopic ultrasound sonography showed a high echoic tumor at the pancreatic head, which was confirmed as adenocarcinoma by fine-needle aspiration biopsy. The preoperative diagnosis was intraductal papillary mucinous carcinoma (IPMC; T1N0M0, stage IA). Subtotal stomach-preserving pancreaticoduodenectomy was performed and the final diagnosis was IPMC, stage 0 (TisN0M0). Conclusions Aggressive surgery for metachronous triple cancer resulted in good long-term prognosis. Continuous and systematic follow-up would allow the detection of malignancy at an early stage and make treatment with curative surgery possible.
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spelling doaj-art-313bfa03ef384572be1487e89f4f1c122025-08-20T02:37:29ZengJapan Surgical SocietySurgical Case Reports2198-77932018-08-01411510.1186/s40792-018-0492-6Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case reportToru Yoshikawa0Tomoyuki Abe1Hironobu Amano2Keiji Hanada3Tomoyuki Minami4Tsuyoshi Kobayashi5Shuji Yonehara6Masahiro Nakahara7Hideki Ohdan8Toshio Noriyuki9Department of Surgery, Onomichi General HospitalDepartment of Surgery, Onomichi General HospitalDepartment of Surgery, Onomichi General HospitalDepartment of Gastroenterology, Onomichi General HospitalDepartment of Gastroenterology, Onomichi General HospitalDepartment of Gastroenterological and Transplant Surgery, Graduate School of Biomedical and Health Sciences, Hiroshima UniversityDepartment of Pathology, Onomichi General HospitalDepartment of Surgery, Onomichi General HospitalDepartment of Gastroenterological and Transplant Surgery, Graduate School of Biomedical and Health Sciences, Hiroshima UniversityDepartment of Surgery, Onomichi General HospitalAbstract Background Peutz–Jeghers syndrome (PJS) is an autosomal dominant disorder characterized by mucocutaneous pigmentation and hamartomatous gastrointestinal polyposis. It is well known that individuals with PJS are at an increased risk of cancer in a variety of organs. Case presentation Here, we present a patient with PJS who achieved long-term survival by undergoing repeat curative surgery for metachronous triple cancer. Her medical history included hilar cholangiocarcinoma and cervical carcinoma; curative surgery was performed for both conditions. On annual follow-up, the level of carcinoembryonic antigen was elevated at 6.9 ng/ml. Enhanced computed tomography revealed a cystic tumor consisting of mural nodules at the pancreatic head; the maximal diameter was 15 mm. Magnetic resonance imaging clearly demonstrated the tumor with low intensity on T1-weighted images and high intensity on T2-weighted images. Endoscopic ultrasound sonography showed a high echoic tumor at the pancreatic head, which was confirmed as adenocarcinoma by fine-needle aspiration biopsy. The preoperative diagnosis was intraductal papillary mucinous carcinoma (IPMC; T1N0M0, stage IA). Subtotal stomach-preserving pancreaticoduodenectomy was performed and the final diagnosis was IPMC, stage 0 (TisN0M0). Conclusions Aggressive surgery for metachronous triple cancer resulted in good long-term prognosis. Continuous and systematic follow-up would allow the detection of malignancy at an early stage and make treatment with curative surgery possible.http://link.springer.com/article/10.1186/s40792-018-0492-6Curative surgeryMetachronous cancerPeutz–Jeghers syndrome
spellingShingle Toru Yoshikawa
Tomoyuki Abe
Hironobu Amano
Keiji Hanada
Tomoyuki Minami
Tsuyoshi Kobayashi
Shuji Yonehara
Masahiro Nakahara
Hideki Ohdan
Toshio Noriyuki
Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
Surgical Case Reports
Curative surgery
Metachronous cancer
Peutz–Jeghers syndrome
title Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
title_full Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
title_fullStr Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
title_full_unstemmed Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
title_short Metachronous triple cancer associated with Peutz–Jeghers syndrome treated with curative surgery: a case report
title_sort metachronous triple cancer associated with peutz jeghers syndrome treated with curative surgery a case report
topic Curative surgery
Metachronous cancer
Peutz–Jeghers syndrome
url http://link.springer.com/article/10.1186/s40792-018-0492-6
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