{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T05:50:03Z","timestamp":1760161803685,"version":"3.37.3"},"reference-count":17,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,5,24]],"date-time":"2020-05-24T00:00:00Z","timestamp":1590278400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2020,5,24]],"date-time":"2020-05-24T00:00:00Z","timestamp":1590278400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["surg case rep"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>We report a rare case of gastrointestinal stromal tumors (GISTs) in Carney\u2019s triad, successfully treated using laparoscopic gastrectomy with lymph node dissection after chemotherapy.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Case presentation<\/jats:title>\n<jats:p>A 21-year-old woman presented to our hospital for treatment of recurrent GISTs. The patient had been admitted for treatment 11\u2009years prior, with black stools being the chief presenting complaint at that time. On examination at that time, multiple submucosal tumors in the pyloric antrum and multiple pulmonary tumors had been observed. She underwent open partial gastrectomy, and the diagnosis of GISTs was confirmed. She was administered tyrosine kinase inhibitors to treat lung metastases from 2\u2009months after surgery. Due to the increasing size of the lung tumors, a right upper lobectomy was performed 9\u2009years after the index gastric surgery. Histopathological examination of the lung specimen, in combination with re-examination of the gastric specimens, was indicative of incomplete-type Carney\u2019s triad. Eleven years after the index gastric surgery, multiple GISTs were observed in her entire stomach. Tumor biopsy revealed a succinate dehydrogenase deficiency, confirming the diagnosis of recurrent GISTs. For treatment, the patient underwent laparoscopic completion gastrectomy, with D1 plus lymph node dissection.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusion<\/jats:title>\n<jats:p>This is a first case report of completion gastrectomy performed laparoscopically for the treatment of GISTs associated with incomplete-type Carney\u2019s triad. The recurrent GISTs developed over a protracted period of 11\u2009years from the index gastric surgery to tumor recurrence.<\/jats:p>\n<\/jats:sec>","DOI":"10.1186\/s40792-020-00877-y","type":"journal-article","created":{"date-parts":[[2020,5,24]],"date-time":"2020-05-24T12:02:40Z","timestamp":1590321760000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Laparoscopic gastrectomy with lymph node dissection for the treatment of remnant stomach gastrointestinal stromal tumors in incomplete-type Carney\u2019s triad: a case report"],"prefix":"10.1186","volume":"6","author":[{"given":"Yuhi","family":"Shimura","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8698-8923","authenticated-orcid":false,"given":"Shingo","family":"Kanaji","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naoki","family":"Urakawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masashi","family":"Yamamoto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masako","family":"Utsumi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gousuke","family":"Takiguchi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Hasegawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiko","family":"Matsuda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kimihiro","family":"Yamashita","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takeru","family":"Matsuda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Taro","family":"Oshikiri","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tetsu","family":"Nakamura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Satoshi","family":"Suzuki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshihiro","family":"Kakeji","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,5,24]]},"reference":[{"key":"877_CR1","doi-asserted-by":"publisher","first-page":"1517","DOI":"10.1056\/NEJM197706302962609","volume":"296","author":"JA Carney","year":"1977","unstructured":"Carney JA, Sheps SG, Go VL, Gordon H. The triad of gastric leiomyosarcoma, functioning extra-adrenal paraganglioma and pulmonary chondroma. N Engl J Med. 1977;296:1517\u20138.","journal-title":"N Engl J Med."},{"key":"877_CR2","doi-asserted-by":"publisher","first-page":"265","DOI":"10.1177\/1066896911415668","volume":"20","author":"F Slevin","year":"2012","unstructured":"Slevin F, Ducan P, Shenjere P, Leahy M, Evans G, Sherlock D. Two out of three required: a case of incomplete Carney triad. Int J Surg Pathol. 2012;20:265\u20138.","journal-title":"Int J Surg Pathol."},{"key":"877_CR3","doi-asserted-by":"publisher","first-page":"543","DOI":"10.4065\/74.6.543","volume":"74","author":"JA Carney","year":"1999","unstructured":"Carney JA. Gastric stromal sarcoma, pulmonary chondroma, and extra-adrenal paraganglioma (Carney Triad): natural history, adrenocortical component, and possible familial occurrence. Mayo Clin Proc. 1999;74:543\u201352.","journal-title":"Mayo Clin Proc."},{"key":"877_CR4","doi-asserted-by":"publisher","first-page":"3656","DOI":"10.1210\/jc.2009-1156","volume":"94","author":"JA Carney","year":"2009","unstructured":"Carney JA. Carney triad: a syndrome featuring paraganglionic, adrenocortical and possibly other endocrine tumors. J Clin Endocrinol Metab. 2009;94:3656\u201362.","journal-title":"J Clin Endocrinol Metab."},{"key":"877_CR5","doi-asserted-by":"publisher","first-page":"3204","DOI":"10.1158\/1078-0432.CCR-07-1984","volume":"14","author":"NP Agaram","year":"2008","unstructured":"Agaram NP, Laquaglia MP, Ustun B, et al. Molecular characterization of pediatric gastrointestinal stromal tumors. Clin Cancer Res. 2008;14:3204\u201315.","journal-title":"Clin Cancer Res."},{"key":"877_CR6","doi-asserted-by":"publisher","first-page":"1171","DOI":"10.1002\/pbc.22123","volume":"53","author":"M Benesch","year":"2009","unstructured":"Benesch M, Wardelman E, Ferrari A, Brennan B, Verschuur A. Gastrointestinal stromal tumors (GIST) in children and adolescents: a comprehensive review of the current literature. Pediatr Blood Cancer. 2009;53:1171\u20139.","journal-title":"Pediatr Blood Cancer."},{"key":"877_CR7","doi-asserted-by":"publisher","first-page":"428","DOI":"10.1016\/S1607-551X(10)70069-3","volume":"26","author":"CF Chen","year":"2010","unstructured":"Chen CF, Chuang CH, Liu MK, Hsu WH, Lin HJ, Hsieh JS. Clinical, radiological and pathologic characteristics of the Carney triad: a case report and literature review. Kaohsiung J Med Sci. 2010;26:428\u201334.","journal-title":"Kaohsiung J Med Sci."},{"key":"877_CR8","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1111\/j.1445-2197.2006.03892.x","volume":"77","author":"CM Byrne","year":"2007","unstructured":"Byrne CM, Daneshjoo R, Ma-Wyatt H, Cox M. Gastrointestinal hemorrhage as a presentation of Carney\u2019s triad. ANZ J Surg. 2007;77:88\u201390.","journal-title":"ANZ J Surg."},{"key":"877_CR9","first-page":"104","volume":"55","author":"Y Samejima","year":"2019","unstructured":"Samejima Y, Fukuzawa H, Maeda K, et al. A case of incomplete-type Carney\u2019s triad with stomach GIST resistant to tyrosine kinase inhibitors. J Japanese Soc Pediatr Surgeons. 2019;55:104\u20139.","journal-title":"J Japanese Soc Pediatr Surgeons."},{"key":"877_CR10","doi-asserted-by":"publisher","first-page":"225","DOI":"10.1016\/0022-3468(91)90919-K","volume":"26","author":"R Hamazoe","year":"1991","unstructured":"Hamazoe R, Shimizu N, Nishidoi H, Maeta M, Koga S. Gastric leiomyoblastoma in childhood. J Pediatr Surg. 1991;26:225\u20137.","journal-title":"J Pediatr Surg."},{"key":"877_CR11","doi-asserted-by":"publisher","first-page":"52","DOI":"10.1186\/1746-1596-6-52","volume":"6","author":"C Otto","year":"2011","unstructured":"Otto C, AgaimyA BA, R\u00e4decke J, Hoeppner J, Illerhaus G, et al. Multifocal gastric gastrointestinal stromal tumors (GISTs) with lymph node metastases in children and young adults: a comparative clinical and histomorphological study of three cases including a new case of Carney triad. Diagnost Pathol. 2011;6:52.","journal-title":"Diagnost Pathol"},{"key":"877_CR12","doi-asserted-by":"publisher","first-page":"229","DOI":"10.4240\/wjgs.v5.i7.229","volume":"5","author":"SW Hong","year":"2013","unstructured":"Hong SW, Lee WY, Lee HK. Hepatic paraganglioma and multifocal gastrointestinal stromal tumor in a female: incomplete Carney triad. World J Gastrointest Surg. 2013;5:229\u201332.","journal-title":"World J Gastrointest Surg."},{"key":"877_CR13","first-page":"748","volume":"56","author":"H Tsunezuka","year":"2014","unstructured":"Tsunezuka H, Kato D, Shimomura M, Terauchi K, Shimada J. A surgical case of incomplete Carney\u2019s triad with multiple heterochronous pulmonary chondromas and recurrent gastrointestinal stromal tumors. JACS. 2014;56:748\u201353.","journal-title":"JACS."},{"key":"877_CR14","first-page":"416","volume":"13","author":"T Nishida","year":"2008","unstructured":"Nishida T, Hirota S, Yanagisawa A, Sugino Y, Minami M, Yamamura Y, Otani Y, et al. Clinical practice guidelines for gastrointestinal stromal tumor (GIST) in Japan: English version. In J Clin Oncol. 2008;13:416\u201330.","journal-title":"In J Clin Oncol."},{"key":"877_CR15","doi-asserted-by":"publisher","first-page":"229","DOI":"10.1136\/jcp.2009.074062","volume":"63","author":"A Agaimy","year":"2010","unstructured":"Agaimy A, Carney JA. Lymphatics and D2-40\/podoplanin expression in gastrointestinal stromal tumours of the stomach with and without lymph node metastasis: an immunohistrochemical study with special reference to the Carney triad. J Clin Pathol. 2010;63:229\u201334.","journal-title":"J Clin Pathol."},{"key":"877_CR16","doi-asserted-by":"publisher","first-page":"375","DOI":"10.1007\/s00423-008-0449-5","volume":"394","author":"A Agaimy","year":"2009","unstructured":"Agaimy A, Wunsch PH. Lymph node metastasis in gastrointestinal stromal tumors (GIST) occurs preferentially in young patients \u226440 years: an overview based on our case material and the literature. Langenbecks Arch Surg. 2009;394:375\u201381.","journal-title":"Langenbecks Arch Surg."},{"key":"877_CR17","doi-asserted-by":"publisher","first-page":"53","DOI":"10.1097\/PAS.0b013e3181c20f4f","volume":"34","author":"L Zhang","year":"2010","unstructured":"Zhang L, Smyrk TC, Young WF Jr, Stratakis CA, Carney A. Gastric stromal tumors in Carney triad are different clinically, pathologically, and behaviorally from sporadic gastric gastrointestinal stromal tumors: finding in 104 cases. Am J Surg Pathol. 2010;34:53\u201364.","journal-title":"Am J Surg Pathol."}],"container-title":["Surgical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40792-020-00877-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s40792-020-00877-y\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40792-020-00877-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,5,23]],"date-time":"2021-05-23T23:06:41Z","timestamp":1621811201000},"score":1,"resource":{"primary":{"URL":"https:\/\/surgicalcasereports.springeropen.com\/articles\/10.1186\/s40792-020-00877-y"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,5,24]]},"references-count":17,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["877"],"URL":"https:\/\/doi.org\/10.1186\/s40792-020-00877-y","relation":{},"ISSN":["2198-7793"],"issn-type":[{"type":"electronic","value":"2198-7793"}],"subject":[],"published":{"date-parts":[[2020,5,24]]},"assertion":[{"value":"2 March 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"14 May 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"24 May 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The genetic analysis was approved by the ethics committee of Kobe University, and written informed consent was obtained from the patient for treatment.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"When obtaining informed consent for the surgical procedure, general consent for publication and presentation was also obtained from the patient.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors have no competing interests to declare.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"112"}}