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      Ileal GIST presenting with bacteremia and liver abscess: A case report and review of literature

      case-report

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          Highlights

          • Small intestine gastrointestinal stromal tumors can infrequently present with intra-abdominal abscess, perforation, obstruction or fistula.

          • Tumor-small intestine fistula is a rare phenomenon and occurs as a result of GISTs’ propensity to cause mucosal ulceration.

          • Although liver metastases are common, in the rare case of a GIST complicated by gastrointestinal tract perforation, abscess or fistula, a pyogenic liver abscess is another important entity to consider.

          • Positive blood cultures with members of the S. milleri group should prompt consideration of occult abdominal infection, perforation and gastrointestinal malignancy.

          Abstract

          Introduction

          Small intestine gastrointestinal stromal tumors can infrequently present with intra-abdominal abscess, perforation, obstruction or fistula. Tumor-small intestine fistula is a rare phenomenon and occurs as a result of GISTs’ propensity to cause mucosal ulceration. This allows bacteria from the gut to gain access to the systemic circulation and predisposes the patient to bacteremia and pyogenic liver abscess.

          Presentation of case

          We present a case of a 63-year-old female whose initial symptoms included fever, nausea, vomiting and right upper quadrant pain. Radiologic studies revealed a liver lesion and an intra-abdominal mass containing oral contrast, suggesting involvement of the gastrointestinal tract. She was found to have a liver abscess, Streptococcus anginosus bacteremia and an ileal GIST that formed a fistula between the tumor and small intestine. We performed a surgical resection of the tumor and percutaneous drainage of the liver abscess. Imatinib was initiated post operatively and she experienced no recurrence, as demonstrated by a surveillance computed tomography scan at 12 months.

          Conclusion

          Findings of a liver lesion in association with a small intestine GIST should raise concern for both metastatic disease and a possible infectious complication such as a pyogenic liver abscess. If a member of the Streptococcus milleri group is isolated in blood cultures, a consideration for gastrointestinal malignancy is imperative. This case report reviews a rare presentation of an ileal GIST with tumor-intestinal fistula, complicated by liver abscess and Streptococcus anginosus bacteremia.

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          Most cited references18

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          The SCARE Statement: Consensus-based surgical case report guidelines.

          Case reports have been a long held tradition within the surgical literature. Reporting guidelines can improve transparency and reporting quality. However, recent consensus-based guidelines for case reports (CARE) are not surgically focused. Our objective was to develop surgical case report guidelines.
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            Adjuvant imatinib mesylate after resection of localised, primary gastrointestinal stromal tumour: a randomised, double-blind, placebo-controlled trial.

            Gastrointestinal stromal tumour is the most common sarcoma of the intestinal tract. Imatinib mesylate is a small molecule that inhibits activation of the KIT and platelet-derived growth factor receptor alpha proteins, and is effective in first-line treatment of metastatic gastrointestinal stromal tumour. We postulated that adjuvant treatment with imatinib would improve recurrence-free survival compared with placebo after resection of localised, primary gastrointestinal stromal tumour. We undertook a randomised phase III, double-blind, placebo-controlled, multicentre trial. Eligible patients had complete gross resection of a primary gastrointestinal stromal tumour at least 3 cm in size and positive for the KIT protein by immunohistochemistry. Patients were randomly assigned, by a stratified biased coin design, to imatinib 400 mg (n=359) or to placebo (n=354) daily for 1 year after surgical resection. Patients and investigators were blinded to the treatment group. Patients assigned to placebo were eligible to crossover to imatinib treatment in the event of tumour recurrence. The primary endpoint was recurrence-free survival, and analysis was by intention to treat. Accrual was stopped early because the trial results crossed the interim analysis efficacy boundary for recurrence-free survival. This study is registered with ClinicalTrials.gov, number NCT00041197. All randomised patients were included in the analysis. At median follow-up of 19.7 months (minimum-maximum 0-56.4), 30 (8%) patients in the imatinib group and 70 (20%) in the placebo group had had tumour recurrence or had died. Imatinib significantly improved recurrence-free survival compared with placebo (98% [95% CI 96-100] vs 83% [78-88] at 1 year; hazard ratio [HR] 0.35 [0.22-0.53]; one-sided p<0.0001). Adjuvant imatinib was well tolerated, with the most common serious events being dermatitis (11 [3%] vs 0), abdominal pain (12 [3%] vs six [1%]), and diarrhoea (ten [2%] vs five [1%]) in the imatinib group and hyperglycaemia (two [<1%] vs seven [2%]) in the placebo group. Adjuvant imatinib therapy is safe and seems to improve recurrence-free survival compared with placebo after the resection of primary gastrointestinal stromal tumour. US National Institutes of Health and Novartis Pharmaceuticals.
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              NCCN Task Force report: update on the management of patients with gastrointestinal stromal tumors.

              The standard of care for managing patients with gastrointestinal stromal tumors (GISTs) rapidly changed after the introduction of effective molecularly targeted therapies involving tyrosine kinase inhibitors (TKIs), such as imatinib mesylate and sunitinib malate. A better understanding of the molecular characteristics of GISTs have improved the diagnostic accuracy and led to the discovery of novel immunomarkers and new mechanisms of resistance to TKI therapy, which in turn have resulted in the development of novel treatment strategies. To address these issues, the NCCN organized a task force consisting of a multidisciplinary panel of experts in the fields of medical oncology, surgical oncology, molecular diagnostics, and pathology to discuss the recent advances, identify areas of future research, and recommend an optimal approach to care for patients with GIST at all stages of disease. The task force met for the first time in October 2003 and again in December 2006 and October 2009. This supplement describes the recent developments in the field of GIST as discussed at the October 2009 meeting.
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                Author and article information

                Contributors
                Journal
                Int J Surg Case Rep
                Int J Surg Case Rep
                International Journal of Surgery Case Reports
                Elsevier
                2210-2612
                27 December 2017
                2018
                27 December 2017
                : 42
                : 261-265
                Affiliations
                [0005]Kendall Regional Medical Center, Department of Surgery, 11750 Bird Road, Miami, FL, 33175, United States
                Author notes
                [* ]Corresponding author at: Kendall Regional Medical Center, 11750 Bird Road, Miami, FL, 33175, United States. marina.gorelik@ 123456hcahealthcare.com
                Article
                S2210-2612(17)30685-5
                10.1016/j.ijscr.2017.12.033
                5985260
                29324374
                314d339f-ad34-475e-8960-233c6bdd5fc3
                © 2018 The Authors

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 17 October 2017
                : 11 December 2017
                : 21 December 2017
                Categories
                Article

                gist, gastrointestinal stromal tumor,rfs, recurrence free survival,os, overall survival,ct, computed tomography,cea, carcinoembryonic antigen,ca 19-9, cancer antigen 19-9,afp, alpha-fetoprotein,pdgfr, platelet-derived growth factor receptor alpha,gist,small intestine,liver abscess,streptococcus anginosus,case report,fistula

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