{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,20]],"date-time":"2026-10-20T18:56:06Z","timestamp":1792522566803,"version":"4.5.0"},"reference-count":12,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"C","license":[{"start":{"date-parts":[[2020,1,1]],"date-time":"2020-01-01T00:00:00Z","timestamp":1577836800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2020,1,1]],"date-time":"2020-01-01T00:00:00Z","timestamp":1577836800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2020,9,10]],"date-time":"2020-09-10T00:00:00Z","timestamp":1599696000000},"content-version":"vor","delay-in-days":253,"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":[[2020,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>GIST\u2019s are the most common mesenchymal tumor of the gastrointestinal tract, clinically and radiologically heterogeneous, only a histological study can confirm the diagnosis. The link between NF1 and GISTs have been established but not fully elucidated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>we report 02 cases of NF1 associated GIST, a 60 years old woman with multiples GISTs in the duodenum, proximal jejunum and in the colon presenting an iron deficiency anemia due to chronic bleeding, operated with R0 resection and a low risk of recurrences and a 41 year old male patient with acute abdominal pain with a giant abdominal mass mimicking a hydatid cyst with no relevant medical history, diagnosed at the same time for typical clinical NF1, CT scan showed the cystic mass but did not confirmed its origin, a complete resection of the mass with no capsule fraction was tricky but successful and the histopathology found a high risk of recurrences. The 2 patients received adjuvant imatinib therapy with recurrence free survival at 12 months follow up. Our cases represents a rare entity (multiples GISTs and cystic GIST) within a rarest population (NF1 associated GIST).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>the diagnosis of NF1 is based on typical clinical criterias but the GISTs are known to be the variable, symptomatic or silent, small size or giant. Imaging is based on CT scan with intravenous contrast studying the vascular pattern, the extra intestinal and metastasis localizations. MRI is no superior, but useful in the study of pelvic GISTs and liver metastasis. Histopathology is the only way to confirm the diagnosis with marker staining with CD117 and DOG-1. The emerging imatinib, sunitinib and regorafinib are used as neoadjuvant or adjuvant therapies in GISTs with high or moderate risk of recurrences. No consensual guidelines are yet established for the follow up as the recurrences are more frequent.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>GIST\u2019s association to NF1 is established, but the different aspects of the physiopathological, clinical and the treatment haven\u2019t been established yet with no larger population to study. We believe that the understanding of the development of this type of tumors within the NF type 1 group would allow a better treatment and follow up and may be can lead to screening.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Highlights<\/jats:title>\n                  <\/jats:sec>","DOI":"10.1016\/j.ijscr.2020.09.072","type":"journal-article","created":{"date-parts":[[2020,9,19]],"date-time":"2020-09-19T11:18:30Z","timestamp":1600514310000},"page":"210-216","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":2,"title":["Neurofibromatosis type 1 associated multiple and cystic gastrointestinal tumors"],"prefix":"10.1097","volume":"76","author":[{"given":"Fatima Zahra","family":"Rahou","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Achraf","family":"Miry","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amal","family":"Bennani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohammed","family":"Bouziane","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"R2-50-20260114","doi-asserted-by":"crossref","first-page":"1035","DOI":"10.1016\/j.mcna.2019.07.004","article-title":"The diagnosis and management of neurofibromatosis type 1","volume":"103","author":"Ly","year":"2019","journal-title":"Med. Clin. North Am"},{"key":"R3-50-20260114","doi-asserted-by":"crossref","first-page":"S255","DOI":"10.1016\/j.annder.2017.09.419","article-title":"Tumeurs malignes et neurofibromatose de type 1: \u00e9tude r\u00e9trospective dans un centre de comp\u00e9tence","volume":"144","author":"Cales","year":"2017","journal-title":"Annales de Dermatologie et de V\u00e9n\u00e9r\u00e9ologie"},{"key":"R4-50-20260114","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1038\/modpathol.3800334","article-title":"Multiple gastrointestinal stromal tumors in type I neurofibromatosis: a pathologic and molecular study","volume":"18","author":"Yantiss","year":"2004","journal-title":"Mod. Pathol"},{"key":"R5-50-20260114","doi-asserted-by":"crossref","first-page":"487","DOI":"10.1016\/j.crad.2003.10.018","article-title":"Imaging of gastrointestinal stromal tumour (GIST)","volume":"59","author":"Lau","year":"2004","journal-title":"Clin. Radiol"},{"key":"R6-50-20260114","doi-asserted-by":"crossref","first-page":"1223","DOI":"10.1016\/j.dld.2019.07.006","article-title":"Gastrointestinal stromal tumours (GISTs): French Intergroup clinical practice guidelines for diagnosis, treatments and follow-up (SNFGE, FFCD, GERCOR, UNICANCER, SFCD, SFED, SFRO)","volume":"51","author":"Landi","year":"2019","journal-title":"Dig. Liver Dis"},{"key":"R7-50-20260114","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1016\/j.ctrv.2016.11.011","article-title":"GEIS guidelines for gastrointestinal sarcomas (GIST)","volume":"55","author":"Poveda","year":"2017","journal-title":"Cancer Treat. Rev"},{"key":"R8-50-20260114","doi-asserted-by":"crossref","first-page":"1411","DOI":"10.1016\/j.humpath.2008.06.025","article-title":"Risk stratification of patients diagnosed with gastrointestinal stromal tumour","volume":"39","author":"Joensuu","year":"2008","journal-title":"Hum. Pathol"},{"key":"R9-50-20260114","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1053\/j.semdp.2006.09.001","article-title":"Gastrointestinal stromal tumours: pathology and prognosis at different sites","volume":"23","author":"Miettinen","year":"2006","journal-title":"Semin. Diagn. Pathol"},{"key":"R11-50-20260114","doi-asserted-by":"crossref","first-page":"484","DOI":"10.1016\/j.surg.2005.08.011","article-title":"Operative indications for relatively small (2-5 cm) gastrointestinal stromal tumor of the stomach based on analysis of 60 operated cases","volume":"139","author":"Otani","year":"2006","journal-title":"Surgery"},{"key":"R12-50-20260114","doi-asserted-by":"crossref","first-page":"1959","DOI":"10.1016\/j.ejca.2009.02.011","article-title":"Clinical evaluation of continuous daily dosing of sunitinib malate in patients with advanced gastrointestinal stromal tumour after imatinib failure","volume":"45","author":"George","year":"2009","journal-title":"Eur. J. Cancer"},{"key":"R13-50-20260114","doi-asserted-by":"crossref","first-page":"295","DOI":"10.1016\/S0140-6736(12)61857-1","article-title":"Efficacity and safety of regorafenib for Advanced gastrointestinal stromal tumours after failure of ilatinib and sunitinib. An international, multicentre, randomized, placebo-controlled phase 3 trial","volume":"381","author":"Demetri","year":"2013","journal-title":"Lancet"},{"key":"R14-50-20260114","doi-asserted-by":"crossref","first-page":"1563","DOI":"10.1200\/JCO.2013.51.2046","article-title":"Pathologic and molecular features correlate with long-term outcome after adjuvant therapy of resected primary GI stromal tumour: the ACOSOG Z9001 trial","volume":"32","author":"Corless","year":"2014","journal-title":"J. Clin. Oncol"}],"container-title":["International Journal of Surgery Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2210261220307458?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2210261220307458?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.lww.com\/10.1016\/j.ijscr.2020.09.072","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,14]],"date-time":"2026-01-14T13:33:51Z","timestamp":1768397631000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/10.1016\/j.ijscr.2020.09.072"}},"subtitle":["02 case reports"],"short-title":[],"issued":{"date-parts":[[2020,1]]},"references-count":12,"journal-issue":{"issue":"C","published-print":{"date-parts":[[2020]]}},"URL":"https:\/\/doi.org\/10.1016\/j.ijscr.2020.09.072","relation":{},"ISSN":["2210-2612"],"issn-type":[{"value":"2210-2612","type":"print"}],"subject":[],"published":{"date-parts":[[2020,1]]}}}