{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,11]],"date-time":"2026-02-11T05:44:29Z","timestamp":1770788669377,"version":"3.50.0"},"reference-count":70,"publisher":"Archives of Pathology and Laboratory Medicine","issue":"11","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2008,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>In this report, we describe a 36-year-old man with the synchronous occurrence of multiple gastric gastrointestinal stromal tumors and multiple intestinal lipomas. Multiple, small, and well-circumscribed gastric gastrointestinal stromal tumors (3 mm to 2 cm) were present within the gastrectomy specimen. The gastrointestinal stromal tumors were composed of epithelioid cells that were strongly positive for CD117 but negative for S100 protein or smooth muscle\u2013specific actin. Also, 17 small submucosal lipomas were identified in the duodenal portion of the gastrectomy specimen. Endoscopic follow-up of the patient revealed more than 20 additional lipomas scattered throughout the intestine. Several large intestinal lipomas were resected endoscopically and histologically confirmed. Although both multicentric gastrointestinal stromal tumor and gastrointestinal lipomatosis have been reported in association with a few genetic syndromes, there has been no report, to our knowledge, of their coexistence. We feel that the coexistence of multiple lesions of these 2 rare diseases in this relatively young patient may represent a novel syndrome.<\/jats:p>","DOI":"10.5858\/132.11.1825","type":"journal-article","created":{"date-parts":[[2021,2,12]],"date-time":"2021-02-12T00:13:07Z","timestamp":1613088787000},"page":"1825-1829","source":"Crossref","is-referenced-by-count":7,"title":["Multiple Gastrointestinal Stromal Tumors and Lipomatosis"],"prefix":"10.5858","volume":"132","author":[{"given":"Xiuli","family":"Liu","sequence":"first","affiliation":[{"name":"From the Division of Anatomic Pathology, Department of Pathology (Dr Liu), and the Division of Gastroenterology and Hepatology, Department of Internal Medicine (Dr Wilcox), University of Alabama, Birmingham; Innovative Pathology Services, Knoxville, Tenn (Dr Nodit); and the Department of Pathology, University of Nebraska, Omaha (Dr Lazenby). Dr Liu is now with the Department of Anatomic Pathology, Cleveland Clinic Foundation, Cleveland, Ohio"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charles Melbern","family":"Wilcox","sequence":"additional","affiliation":[{"name":"From the Division of Anatomic Pathology, Department of Pathology (Dr Liu), and the Division of Gastroenterology and Hepatology, Department of Internal Medicine (Dr Wilcox), University of Alabama, Birmingham; Innovative Pathology Services, Knoxville, Tenn (Dr Nodit); and the Department of Pathology, University of Nebraska, Omaha (Dr Lazenby). Dr Liu is now with the Department of Anatomic Pathology, Cleveland Clinic Foundation, Cleveland, Ohio"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laurentia","family":"Nodit","sequence":"additional","affiliation":[{"name":"From the Division of Anatomic Pathology, Department of Pathology (Dr Liu), and the Division of Gastroenterology and Hepatology, Department of Internal Medicine (Dr Wilcox), University of Alabama, Birmingham; Innovative Pathology Services, Knoxville, Tenn (Dr Nodit); and the Department of Pathology, University of Nebraska, Omaha (Dr Lazenby). Dr Liu is now with the Department of Anatomic Pathology, Cleveland Clinic Foundation, Cleveland, Ohio"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Audrey J.","family":"Lazenby","sequence":"additional","affiliation":[{"name":"From the Division of Anatomic Pathology, Department of Pathology (Dr Liu), and the Division of Gastroenterology and Hepatology, Department of Internal Medicine (Dr Wilcox), University of Alabama, Birmingham; Innovative Pathology Services, Knoxville, Tenn (Dr Nodit); and the Department of Pathology, University of Nebraska, Omaha (Dr Lazenby). Dr Liu is now with the Department of Anatomic Pathology, Cleveland Clinic Foundation, Cleveland, Ohio"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"3906","reference":[{"key":"i1543-2165-132-11-1825-b1","series-title":"Science","first-page":"577","article-title":"Gain-of-function mutations of c-kit in human gastrointestinal stromal tumors.","volume":"279","author":"Hirota, S.","year":"1998","unstructured":"Hirota, S., K.Isozaki, and Y.Moriyama. et al. Gain-of-function mutations of c-kit in human gastrointestinal stromal tumors.Science1998. 279:577\u2013580."},{"key":"i1543-2165-132-11-1825-b2","series-title":"Am J Surg Pathol","first-page":"477","article-title":"Gastrointestinal stromal tumors of the jejunum and ileum: a clinicopathologic, immunohistochemical, and molecular genetic study of 906 cases before imatinib with long-term follow-up.","volume":"30","author":"Miettinen, M.","year":"2006","unstructured":"Miettinen, M., H.Makhlouf, and L. H.Sobin. et al. Gastrointestinal stromal tumors of the jejunum and ileum: a clinicopathologic, immunohistochemical, and molecular genetic study of 906 cases before imatinib with long-term follow-up.Am J Surg Pathol2006. 30:477\u2013489."},{"key":"i1543-2165-132-11-1825-b3","series-title":"Nat Genet","first-page":"323","article-title":"Familial gastrointestinal stromal tumours with germline mutation of the KIT gene.","volume":"19","author":"Nishida, T.","year":"1998","unstructured":"Nishida, T., S.Hirota, and M.Taniguchi. et al. Familial gastrointestinal stromal tumours with germline mutation of the KIT gene.Nat Genet1998. 19:323\u2013324."},{"key":"i1543-2165-132-11-1825-b4","series-title":"Virchows Arch","first-page":"421","article-title":"The reappraisal of gastrointestinal stromal tumors: from Stout to the KIT revolution.","volume":"442","author":"Dei Tos, A. P.","year":"2003","unstructured":"Dei Tos, A. P.\n          \n          The reappraisal of gastrointestinal stromal tumors: from Stout to the KIT revolution.\n          Virchows Arch\n          2003. 442:421\u2013428."},{"key":"i1543-2165-132-11-1825-b5","series-title":"Virchows Arch","first-page":"1","article-title":"Gastrointestinal stromal tumors\u2014definition, clinical, histological, immunohistochemical, and molecular genetic features and differential diagnosis.","volume":"438","author":"Miettinen, M.","year":"2001","unstructured":"Miettinen, M. and J.Lasota. Gastrointestinal stromal tumors\u2014definition, clinical, histological, immunohistochemical, and molecular genetic features and differential diagnosis.Virchows Arch2001. 438:1\u201312."},{"key":"i1543-2165-132-11-1825-b6","series-title":"N Engl J Med","first-page":"472","article-title":"Efficacy and safety of imatinib mesylate in advanced gastrointestinal stromal tumors.","volume":"347","author":"Demetri, G. D.","year":"2002","unstructured":"Demetri, G. D., M.von Mehren, and C. D.Blanke. et al. Efficacy and safety of imatinib mesylate in advanced gastrointestinal stromal tumors.N Engl J Med2002. 347:472\u2013480."},{"key":"i1543-2165-132-11-1825-b7","series-title":"Gastroenterology","first-page":"660","article-title":"Gain-of-function mutations of platelet-derived growth factor receptor alpha gene in gastrointestinal stromal tumors.","volume":"125","author":"Hirota, S.","year":"2003","unstructured":"Hirota, S., A.Ohashi, and T.Nishida. et al. Gain-of-function mutations of platelet-derived growth factor receptor alpha gene in gastrointestinal stromal tumors.Gastroenterology2003. 125:660\u2013667."},{"key":"i1543-2165-132-11-1825-b8","series-title":"Am J Surg Pathol","first-page":"1170","article-title":"NF1-associated gastrointestinal stromal tumors have unique clinical, phenotypic, and genotypic characteristics.","volume":"29","author":"Andersson, J.","year":"2005","unstructured":"Andersson, J., H.Sihto, and J. M.Meis-Kindblom. et al. NF1-associated gastrointestinal stromal tumors have unique clinical, phenotypic, and genotypic characteristics.Am J Surg Pathol2005. 29:1170\u20131176."},{"key":"i1543-2165-132-11-1825-b9","series-title":"Am J Surg Pathol","first-page":"755","article-title":"Gastrointestinal stromal tumors of neurofibromatosis type I (von Recklinghausen's disease).","volume":"29","author":"Takazawa, Y.","year":"2005","unstructured":"Takazawa, Y., S.Sakurai, and Y.Sakuma. et al. Gastrointestinal stromal tumors of neurofibromatosis type I (von Recklinghausen's disease).Am J Surg Pathol2005. 29:755\u2013763."},{"key":"i1543-2165-132-11-1825-b10","series-title":"Hum Pathol","first-page":"229","article-title":"Solitary solid stromal gastrointestinal tumors in von Recklinghausen's disease with minimal smooth muscle differentiation.","volume":"15","author":"Schaldenbrand, J. D.","year":"1984","unstructured":"Schaldenbrand, J. D. and H. D.Appelman. 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Well differentiated \u201clipoma-like\u201d liposarcoma of the sigmoid mesocolon and multiple lipomatosis of the rectosigmoid colon: report of a case.Hepatogastroenterology1998. 45:2151\u20132156."},{"key":"i1543-2165-132-11-1825-b17","series-title":"Dis Colon Rectum","first-page":"769","article-title":"Diffuse colonic lipomatosis with giant hypertrophy of the epiploic appendices and diverticulosis of the colon: report of a case and review of the literature.","volume":"38","author":"Catania, G.","year":"1995","unstructured":"Catania, G., G. A.Petralia, and M.Migliore. et al. 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Encephalocraniocutaneous lipomatosis with a mutation in the NF1 gene.J Med Genet1995. 32:316\u2013319."},{"key":"i1543-2165-132-11-1825-b20","series-title":"J Med Genet","first-page":"937","article-title":"Germline mutation of the tumour suppressor PTEN in Proteus syndrome.","volume":"39","author":"Smith, J. M.","year":"2002","unstructured":"Smith, J. M., E. P.Kirk, and G.Theodosopoulos. et al. Germline mutation of the tumour suppressor PTEN in Proteus syndrome.J Med Genet2002. 39:937\u2013940."},{"key":"i1543-2165-132-11-1825-b21","series-title":"Hum Mol Genet","first-page":"765","article-title":"Germline and germline mosaic PTEN mutations associated with a Proteus-like syndrome of hemihypertrophy, lower limb asymmetry, arteriovenous malformations and lipomatosis.","volume":"9","author":"Zhou, X-P.","year":"2000","unstructured":"Zhou, X-P., D. J.Marsh, and H.Hampel. et al. Germline and germline mosaic PTEN mutations associated with a Proteus-like syndrome of hemihypertrophy, lower limb asymmetry, arteriovenous malformations and lipomatosis.Hum Mol Genet2000. 9:765\u2013768."},{"key":"i1543-2165-132-11-1825-b22","series-title":"J Assoc Physicians India","first-page":"586","article-title":"Lipomatosis associated with small intestinal leiomyoma.","volume":"42","author":"Joseph, R. C.","year":"1994","unstructured":"Joseph, R. C. and R. B.Deshpande. Lipomatosis associated with small intestinal leiomyoma.J Assoc Physicians India1994. 42:586."},{"key":"i1543-2165-132-11-1825-b23","series-title":"Hum Pathol","first-page":"484","article-title":"Biology and genetic aspects of gastrointestinal stromal tumors: KIT activation and cytogenetic alterations.","volume":"33","author":"Heinrich, M. C.","year":"2002","unstructured":"Heinrich, M. C., B. P.Rubin, and B. J.Longley. et al. Biology and genetic aspects of gastrointestinal stromal tumors: KIT activation and cytogenetic alterations.Hum Pathol2002. 33:484\u2013495."},{"key":"i1543-2165-132-11-1825-b24","series-title":"Am J Pathol","first-page":"15","article-title":"The complexity of KIT gene mutations and chromosome rearrangements and their clinical correlation in gastrointestinal stromal (pacemaker cell) tumors.","volume":"160","author":"Andersson, J.","year":"2002","unstructured":"Andersson, J., H.Sjogren, and J. M.Meis-Kindblom. et al. The complexity of KIT gene mutations and chromosome rearrangements and their clinical correlation in gastrointestinal stromal (pacemaker cell) tumors.Am J Pathol2002. 160:15\u201322."},{"key":"i1543-2165-132-11-1825-b25","series-title":"Am J Pathol","first-page":"791","article-title":"KIT extracellular and kinase domain mutations in gastrointestinal stromal tumors.","volume":"156","author":"Lux, M. L.","year":"2000","unstructured":"Lux, M. L., B. P.Rubin, and T. L.Biase. et al. KIT extracellular and kinase domain mutations in gastrointestinal stromal tumors.Am J Pathol2000. 156:791\u2013795."},{"key":"i1543-2165-132-11-1825-b26","series-title":"Medicine","first-page":"159","article-title":"The triad of gastric epithelioid leiomyosarcoma, pulmonary chondroma, and functioning extra-adrenal paraganglioma: a five-year review.","volume":"62","author":"Carney, J. A.","year":"1983","unstructured":"Carney, J. A.\n          \n          The triad of gastric epithelioid leiomyosarcoma, pulmonary chondroma, and functioning extra-adrenal paraganglioma: a five-year review.\n          Medicine\n          1983. 62:159\u2013169."},{"key":"i1543-2165-132-11-1825-b27","series-title":"Am J Surg Pathol","first-page":"326","article-title":"Cause of familial and multiple gastrointestinal autonomic nerve tumors with hyperplasia of interstitial cells of Cajal is germline mutation of the c-kit gene.","volume":"24","author":"Hirota, S.","year":"2000","unstructured":"Hirota, S., T.Okazaki, and Y.Kitamura. et al. 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NF1-associated gastrointestinal stromal tumors have unique clinical, phenotypic, and genotypic characteristics.Am J Surg Pathol2005. 29:1170\u20131176."},{"key":"i1543-2165-132-11-1825-b9","series-title":"Am J Surg Pathol","first-page":"755","article-title":"Gastrointestinal stromal tumors of neurofibromatosis type I (von Recklinghausen's disease).","volume":"29","author":"Takazawa, Y.","year":"2005","unstructured":"Takazawa, Y., S.Sakurai, and Y.Sakuma. et al. Gastrointestinal stromal tumors of neurofibromatosis type I (von Recklinghausen's disease).Am J Surg Pathol2005. 29:755\u2013763."},{"key":"i1543-2165-132-11-1825-b10","series-title":"Hum Pathol","first-page":"229","article-title":"Solitary solid stromal gastrointestinal tumors in von Recklinghausen's disease with minimal smooth muscle differentiation.","volume":"15","author":"Schaldenbrand, J. D.","year":"1984","unstructured":"Schaldenbrand, J. D. and H. D.Appelman. Solitary solid stromal gastrointestinal tumors in von Recklinghausen's disease with minimal smooth muscle differentiation.Hum Pathol1984. 15:229\u2013232."},{"key":"i1543-2165-132-11-1825-b11","series-title":"Mayo Clin Proc","first-page":"543","article-title":"Gastric stromal sarcoma, pulmonary chondroma, and extra-adrenal paraganglioma (Carney triad): natural history, adrenocortical component, and possible familial occurrence.","volume":"74","author":"Carney, J. A.","year":"1999","unstructured":"Carney, J. A.\n          \n          Gastric stromal sarcoma, pulmonary chondroma, and extra-adrenal paraganglioma (Carney triad): natural history, adrenocortical component, and possible familial occurrence.\n          Mayo Clin Proc\n          1999. 74:543\u2013552."},{"key":"i1543-2165-132-11-1825-b12","series-title":"Am J Surg Pathol","first-page":"52","article-title":"Gastrointestinal stromal tumors of the stomach: a clinicopathologic, immunohistochemical, and molecular genetic study of 1765 cases with long-term follow-up.","volume":"29","author":"Miettinen, M.","year":"2005","unstructured":"Miettinen, M., L. H.Sobin, and J.Lasota. Gastrointestinal stromal tumors of the stomach: a clinicopathologic, immunohistochemical, and molecular genetic study of 1765 cases with long-term follow-up.Am J Surg Pathol2005. 29:52\u201368."},{"key":"i1543-2165-132-11-1825-b13","series-title":"Am J Med Genet","first-page":"132","article-title":"Familial paraganglioma and gastric stromal sarcoma: a new syndrome distinct from the Carney triad.","volume":"108","author":"Carney, J. A.","year":"2002","unstructured":"Carney, J. A. and C. A.Stratakis. Familial paraganglioma and gastric stromal sarcoma: a new syndrome distinct from the Carney triad.Am J Med Genet2002. 108:132\u2013139."},{"key":"i1543-2165-132-11-1825-b14","series-title":"Surgery","first-page":"598","article-title":"Lipoma of the alimentary tract.","volume":"53","author":"Mayo, C. W.","year":"1963","unstructured":"Mayo, C. W., R. J.Pagtalunan, and D. J.Brown. 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