{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T18:48:13Z","timestamp":1787683693449,"version":"build-2784847793"},"reference-count":20,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"C","license":[{"start":{"date-parts":[[2015,1,1]],"date-time":"2015-01-01T00:00:00Z","timestamp":1420070400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2015,10,8]],"date-time":"2015-10-08T00:00:00Z","timestamp":1444262400000},"content-version":"vor","delay-in-days":280,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2015,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>INTRODUCTION:<\/jats:title>\n                    <jats:p>Gastrointestinal stromal tumours (GISTs) are thought to derive from or differentiate towards the interstitial cells of Cajal (ICC) as most demonstrate a similar immunoprofile: CD117+, CD34+ and DOG1+. ICC hyperplasia refers to KIT-expressing microscopic spindle cell proliferations involving the myenteric plexus.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CASE REPORT:<\/jats:title>\n                    <jats:p>74 year-old male presented with a 5-year history of heartburn and dysphagia. Imaging revealed a 4 cm GIST in the gastric fundus. Pathology of the resected specimen revealed diffuse segmental ICC hyperplasia harbouring two macroscopic GISTs and a \u2018tumorlet\u2019. A mutation in c-KIT exon 11 was detected in both the solid and the diffuse components.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>DISCUSSION:<\/jats:title>\n                    <jats:p>\n                      ICC hyperplasia can occur either as a sporadic focal lesion or in a syndromic setting, known to predispose to multiple GIST tumours at different sites. The majority of cases of sporadic ICC hyperplasia previously reported were of localised type. The hereditary form is mostly caused by germline mutations in\n                      <jats:italic toggle=\"yes\">c-KIT<\/jats:italic>\n                      and\n                      <jats:italic toggle=\"yes\">PDGFRA<\/jats:italic>\n                      or in patients with NF-1 andpresents as a diffuse hyperplasia, usually with a confluent, nodular or multifocal growth pattern.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CONCLUSION:<\/jats:title>\n                    <jats:p>\n                      We describe a diffuse form of sporadic ICC hyperplasia harbouring multifocal GISTs, mimicking diffuse ICC hyperplasia in hereditary GIST syndromes. Detection of somatic\n                      <jats:italic toggle=\"yes\">c-KIT<\/jats:italic>\n                      exon 11 mutation ruled out a hereditary disorder.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Highlights<\/jats:title>\n                  <\/jats:sec>","DOI":"10.1016\/j.ijscr.2015.10.006","type":"journal-article","created":{"date-parts":[[2015,10,25]],"date-time":"2015-10-25T00:37:40Z","timestamp":1445733460000},"page":"202-205","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":3,"title":["Sporadic diffuse segmental interstitial cell of Cajal hyperplasia harbouring two gastric gastrointestinal stromal tumours (GIST) mimicking hereditary GIST syndromes"],"prefix":"10.1097","volume":"16","author":[{"given":"Mafalda Costa","family":"Neves","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gordon","family":"Stamp","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satvinder","family":"Mudan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"R1-52-20260114","doi-asserted-by":"crossref","first-page":"1466","DOI":"10.5858\/2006-130-1466-GSTROM","article-title":"Gastrointestinal stromal tumors: review on morphology, molecular pathology, prognosis, and differential diagnosis","volume":"130","author":"Miettinen","year":"2006","journal-title":"Arch. Pathol. Lab. Med"},{"key":"R2-52-20260114","doi-asserted-by":"crossref","first-page":"557","DOI":"10.1146\/annurev.pathmechdis.3.121806.151538","article-title":"Molecular pathobiology of gastrointestinal stromal sarcomas","volume":"3","author":"Corless","year":"2008","journal-title":"Annu. Rev. Pathol"},{"key":"R3-52-20260114","doi-asserted-by":"crossref","first-page":"141","DOI":"10.1309\/AJCPPCKGA2QGBJ1R","article-title":"BRAF mutation status in gastrointestinal stromal tumors","volume":"133","author":"Hostein","year":"2010","journal-title":"Am. J. Clin. Pathol"},{"key":"R4-52-20260114","doi-asserted-by":"crossref","first-page":"S1","DOI":"10.1038\/modpathol.2013.173","article-title":"Gastrointestinal stromal tumors: what do we know now?","volume":"27","author":"Corless","year":"2014","journal-title":"Mod. Pathol"},{"key":"R5-52-20260114","doi-asserted-by":"crossref","first-page":"190","DOI":"10.1111\/j.1365-2559.2006.02497.x","article-title":"Interstitial cells of Cajal in health and disease. Part II: ICC and gastrointestinal stromal tumours","volume":"50","author":"Streutker","year":"2007","journal-title":"Histopathology"},{"key":"R6-52-20260114","doi-asserted-by":"crossref","first-page":"326","DOI":"10.1097\/00000478-200002000-00045","article-title":"Cause of familial and multiple gastrointestinal autonomic nerve tumours with hyperplasia of interstitial cells of Cajal is germline mutation of the c-kit gene","volume":"24","author":"Hirota","year":"2000","journal-title":"Am. J. Surg. Pathol"},{"key":"R7-52-20260114","doi-asserted-by":"crossref","first-page":"318","DOI":"10.1053\/j.gastro.2003.10.079","article-title":"PDGFRA germline mutation in a family with multiple cases of gastrointestinal tumor","volume":"126","author":"Chompret","year":"2004","journal-title":"Gastroenterology"},{"key":"R8-52-20260114","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1097\/01.pas.0000176433.81079.bd","article-title":"Gastrointestinal stromal tumors in patients with neurofibromatosis 1: a clinicopathologic and molecular genetic study of 45 cases","volume":"30","author":"Miettinen","year":"2006","journal-title":"Am. J. Surg. Pathol"},{"key":"R9-52-20260114","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1111\/j.1365-2796.2009.02110.x","article-title":"The triad of paragangliomas, gastric stromal tumours and pulmonary chondromas (Carney triad), and the dyad of paragangliomas and gastric stromal sarcomas (Carney-Stratakis syndrome): molecular genetics and clinical implications","volume":"266","author":"Stratakis","year":"2009","journal-title":"J. Intern. Med"},{"key":"R10-52-20260114","doi-asserted-by":"crossref","first-page":"1568","DOI":"10.1097\/00000478-200011000-00016","article-title":"Congenital interstitial cell of Cajal hyperplasia with neuronal intestinal dysplasia","volume":"24","author":"Jeng","year":"2000","journal-title":"Am. J. Surg. Pathol"},{"key":"R11-52-20260114","doi-asserted-by":"crossref","first-page":"322","DOI":"10.1007\/s00423-005-0005-5","article-title":"Gastrointestinal stromal tumours: a regular origin in the muscularis propria, but an extremely diverse gross presentation. A review of 200 cases to critically re-evaluate the concept of so-called extra-gastrointestinal stromal tumours","volume":"391","author":"Agaimy","year":"2006","journal-title":"Langenbecks Arch. Surg"},{"key":"R12-52-20260114","doi-asserted-by":"crossref","first-page":"288","DOI":"10.1007\/s00428-005-0117-x","article-title":"Sporadic Cajal cell hyperplasia is common in resection specimens for distal oesophageal carcinoma. A retrospective review of 77 consecutive surgical resection specimens","volume":"448","author":"Agaimy","year":"2005","journal-title":"Virchows Arch"},{"key":"R13-52-20260114","doi-asserted-by":"crossref","first-page":"793","DOI":"10.1136\/gut.51.6.793","article-title":"Polyclonal nature of diffuse proliferation of interstitial cells of Cajal in patients with familial and multiple gastrointestinal stromal tumours","volume":"51","author":"Chen","year":"2002","journal-title":"Gut"},{"key":"R14-52-20260114","first-page":"549","article-title":"Sporadic segmental Interstitial cell of Cajal hyperplasia (microscopic GIST) with unusual diffuse longitudinal growth replacing the muscularis propria: differential diagnosis to hereditary GIST syndromes","volume":"3","author":"Agaimy","year":"2010","journal-title":"Int. J. Clin. Exp. Pathol"},{"key":"R15-52-20260114","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1186\/1477-7819-5-50","article-title":"Gastrointestinal stromal tumour in Meckel\u2019s diverticulum","volume":"5","author":"Chandramohan","year":"2007","journal-title":"World J. Surg. Oncol"},{"key":"R16-52-20260114","first-page":"23","article-title":"Small bowel gastrointestinal stromal tumour (GIST) arising in jejunal diverticulum","volume":"92","author":"Schepers","year":"2009","journal-title":"JBR-BTR"},{"key":"R17-52-20260114","doi-asserted-by":"crossref","first-page":"933","DOI":"10.1097\/01.pas.0000213440.78407.27","article-title":"Multicentric sporadic gastrointestinal stromal tumors (GISTs) of the stomach with distinct clonal origin: differential diagnosis to familial and syndromal GIST variants and peritoneal metastasis","volume":"31","author":"Haller","year":"2007","journal-title":"Am. J. Surg. Pathol"},{"key":"R18-52-20260114","doi-asserted-by":"crossref","first-page":"1553","DOI":"10.1097\/PAS.0b013e31817587ea","article-title":"Multiple sporadic gastrointestinal stromal tumors (GISTs) of the proximal stomach are caused by different somatic KIT mutations suggesting a field effect","volume":"32","author":"Agaimy","year":"2008","journal-title":"Am. J. Surg. Pathol"},{"key":"R19-52-20260114","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1097\/01.pas.0000213307.05811.f0","article-title":"Minute gastric sclerosing stromal tumors (GIST tumorlets) are common in adults and frequently show c-KIT mutations","volume":"31","author":"Agaimy","year":"2007","journal-title":"Am. J. Surg. Pathol"},{"key":"R20-52-20260114","doi-asserted-by":"crossref","first-page":"853","DOI":"10.6004\/jnccn.2014.0080","article-title":"Gastrointestinal stromal tumors, version 2","volume":"12","author":"von Mehren","year":"2014","journal-title":"J. Natl. Compr. Cancer Netw"}],"container-title":["International Journal of Surgery Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2210261215004423?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2210261215004423?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.lww.com\/01612930-201516000-00052","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,14]],"date-time":"2026-01-14T13:21:18Z","timestamp":1768396878000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/01612930-201516000-00052"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,1]]},"references-count":20,"journal-issue":{"issue":"C","published-print":{"date-parts":[[2015]]}},"URL":"https:\/\/doi.org\/10.1016\/j.ijscr.2015.10.006","relation":{},"ISSN":["2210-2612"],"issn-type":[{"value":"2210-2612","type":"print"}],"subject":[],"published":{"date-parts":[[2015,1]]}}}