scholarly journals Current treatment and outcomes of pediatric gastrointestinal stromal tumors (GIST): a systematic review of published studies

Author(s):  
Arimatias Raitio ◽  
Adeline Salim ◽  
Dhanya Mullassery ◽  
Paul D. Losty

AbstractGastrointestinal stromal tumor (GIST) is a rare cancer of mesenchymal origin mostly seen in adult and elderly populations. Therefore, the prognostic and therapeutic features of pediatric GIST are not clearly defined. Clinical knowledge has been largely extrapolated from case series and adult studies. In this systematic review, we aimed to analyze the health outcome metrics of pediatric GIST. Medline and Embase databases were searched using relevant key terms. The original search retrieved 1,892 titles; 27 studies with 184 patients (68% female) were included for final review. The primary tumors were located in the stomach (165/184, 90%), small bowel (12/184, 7%), and elsewhere (7/184, 4%). Individual patient data were available in 125 cases with a median follow-up of 6.7 years. All patients underwent surgical resection, which varied from wide local excision to total gastrectomy. There were 12 deaths (10%), 65 (52%) patients were alive with no evidence of disease, and 31 cases (25%) were alive with disease. Tumor size > 5 cm, high mitotic index, and spindle morphology were predictive of mortality. Pediatric GIST has a more favorable prognosis and different characteristics versus adult tumors. There is a crucial need for international consensus and specific pediatric guidelines for the treatment of this rare tumor.

2019 ◽  
Vol 24 (5) ◽  
pp. 558-571 ◽  
Author(s):  
Kartik Bhatia ◽  
Hans Kortman ◽  
Christopher Blair ◽  
Geoffrey Parker ◽  
David Brunacci ◽  
...  

OBJECTIVEThe role of mechanical thrombectomy in pediatric acute ischemic stroke is uncertain, despite extensive evidence of benefit in adults. The existing literature consists of several recent small single-arm cohort studies, as well as multiple prior small case series and case reports. Published reports of pediatric cases have increased markedly since 2015, after the publication of the positive trials in adults. The recent AHA/ASA Scientific Statement on this issue was informed predominantly by pre-2015 case reports and identified several knowledge gaps, including how young a child may undergo thrombectomy. A repeat systematic review and meta-analysis is warranted to help guide therapeutic decisions and address gaps in knowledge.METHODSUsing PRISMA-IPD guidelines, the authors performed a systematic review of the literature from 1999 to April 2019 and individual patient data meta-analysis, with 2 independent reviewers. An additional series of 3 cases in adolescent males from one of the authors’ centers was also included. The primary outcomes were the rate of good long-term (mRS score 0–2 at final follow-up) and short-term (reduction in NIHSS score by ≥ 8 points or NIHSS score 0–1 at up to 24 hours post-thrombectomy) neurological outcomes following mechanical thrombectomy for acute ischemic stroke in patients < 18 years of age. The secondary outcome was the rate of successful angiographic recanalization (mTICI score 2b/3).RESULTSThe authors’ review yielded 113 cases of mechanical thrombectomy in 110 pediatric patients. Although complete follow-up data are not available for all patients, 87 of 96 (90.6%) had good long-term neurological outcomes (mRS score 0–2), 55 of 79 (69.6%) had good short-term neurological outcomes, and 86 of 98 (87.8%) had successful angiographic recanalization (mTICI score 2b/3). Death occurred in 2 patients and symptomatic intracranial hemorrhage in 1 patient. Sixteen published thrombectomy cases were identified in children < 5 years of age.CONCLUSIONSMechanical thrombectomy may be considered for acute ischemic stroke due to large vessel occlusion (ICA terminus, M1, basilar artery) in patients aged 1–18 years (Level C evidence; Class IIb recommendation). The existing evidence base is likely affected by selection and publication bias. A prospective multinational registry is recommended as the next investigative step.


2021 ◽  
Vol 22 (1) ◽  
Author(s):  
Matteo Buda ◽  
Riccardo D’Ambrosi ◽  
Enrico Bellato ◽  
Davide Blonna ◽  
Alessandro Cappellari ◽  
...  

Abstract Background Revision surgery after the Latarjet procedure is a rare and challenging surgical problem, and various bony or capsular procedures have been proposed. This systematic review examines clinical and radiographic outcomes of different procedures for treating persistent pain or recurrent instability after a Latarjet procedure. Methods A systematic review of the literature was performed using the Medline, Cochrane, EMBASE, Google Scholar and Ovid databases with the combined keywords “failed”, “failure”, “revision”, “Latarjet”, “shoulder stabilization” and “shoulder instability” to identify articles published in English that deal with failed Latarjet procedures. Results A total of 11 studies (five retrospective and six case series investigations), all published between 2008 and 2020, fulfilled our inclusion criteria. For the study, 253 patients (254 shoulders, 79.8% male) with a mean age of 29.6 years (range: 16–54 years) were reviewed at an average follow-up of 51.5 months (range: 24–208 months). Conclusions Eden–Hybinette and arthroscopic capsuloplasty are the most popular and safe procedures to treat recurrent instability after a failed Latarjet procedure, and yield reasonable clinical outcomes. A bone graft procedure and capsuloplasty were proposed but there was no clear consensus on their efficacy and indication. Level of evidence Level IV Trial registration PROSPERO 2020 CRD42020185090—www.crd.york.ac.uk/prospero/


2021 ◽  
Vol 11 (4) ◽  
pp. 1903
Author(s):  
Adrian Kahn ◽  
Shlomo Matalon ◽  
Rahaf Bassam Salem ◽  
Lazar Kats ◽  
Liat Chaushu ◽  
...  

This study aimed to characterize the demographic and clinical features of underreported surgical ciliated cysts developing after sinus floor augmentation, based on a series of cases from our files and a systematic review of the literature. A series of five cases (four patients) of microscopically confirmed surgical ciliated cysts following sinus floor augmentation procedures from our files are described. A systematic literature search (1991–2020) with strict clinical-, radiological- and microscopic-based exclusion and inclusion criteria was performed to detect additional similar cases. The systematic review revealed only five cases that fulfilled the inclusion criteria. Altogether, surgical ciliated cysts associated with sinus floor augmentation have been rarely reported in the literature, and have not been characterized either demographically or clinically. Graft materials were diverse, implants were placed simultaneously, or up to two years post-augmentation. The associated surgical ciliated cysts developed between 0.5 and 10 years post-augmentation. Although limited in its extent, this study is the first series to characterize possible underreported sequelae of surgical ciliated cysts associated with sinus floor augmentation. It emphasizes the need for long post-operative follow-up and confirmation of lesion by microscopic examination.


2016 ◽  
Vol 8 (1) ◽  
Author(s):  
Andreas Panagopoulos ◽  
Irini Tatani ◽  
Dimitrios Ntourantonis ◽  
Ioannis Seferlis ◽  
Antonis Kouzelis ◽  
...  

The valgus-impacted (VI) 4-part fractures are a subset of fractures of the proximal humerus with a unique anatomic configuration characterized by a relatively lower incidence of avascular necrosis after operative intervention. We systematically reviewed clinical studies assessing the benefits and harms of least possible fixation techniques (LPFT) for this unique fracture type. Such information would be potentially helpful in developing an evidence-based approach in the management of these complex injuries. We performed analytic searches of PubMed, Embase, Web of Science, Google Scholar and the Cochrane Library, restricting it to the years 1991-2014. Included studies had to describe outcomes and complications after primary osteosynthesis with any type of LPFT apart from plate-screws and intramedullary nailing. Eligibility criteria were also included English language, more than 5 cases, minimum follow up of one year and report of clinical outcome using at least one relevant score (Constant, Neer or ASES). Based on 292 database hits we identified 12 eligible studies including 190 four-part valgus impacted fractures in 188 patients. All eligible studies were case series composed of min 8 to max 45 patients per study. The gender distribution was 60% (112) female and 40% (76) male. The average age of the patients at the time of injury was 54.5 years. In 8/12 studies an open reduction was used for fracture fixation using different surgical techniques including KW, cerclage wires, cannulated screws and osteosutures. Closed reduction and percutaneous fixation was used in 4 studies. Mean follow-up time ranged from 24 to 69 months. A good functional outcome (constant score &gt;80) was reported in 9/12 studies. The most common complication was avascular necrosis of the humeral head with an overall incidence of 11% (range, 0-26.3%). Total avascular necrosis (AVN) was found in 15/188 patients (7.9%) and was more common in percutaneous techniques and partial AVN in 6/188 (3.1%) being more common in open techniques. The overall re-operation rate was very low (3.7%). Insufficient study designs cannot provide definite treatment recommendations and quantitative data synthesis of outcome. In general, LPFT for 4-part VI fractures leads to satisfactory outcomes with low incidence of complications. A considerable risk of biases can be attributed to fracture classification, proper radiological control, mean age of patients, mixed types of fixation methods, nonage adjusted clinical scoring and small follow up periods. These factors are discussed in detail. Level of evidence: systematic review of literature (level IV) as most of the studies were level IV.


Neurosurgery ◽  
2019 ◽  
Vol 85 (6) ◽  
pp. E975-E991 ◽  
Author(s):  
Stephen Shelby Burks ◽  
Ross C Puffer ◽  
Iahn Cajigas ◽  
David Valdivia ◽  
Andrew E Rosenberg ◽  
...  

Abstract BACKGROUND Synovial sarcoma of the nerve is a rare entity with several cases and case series reported in the literature. Despite an improved understanding of the biology, the clinical course is difficult to predict. OBJECTIVE To compile a series of patients with synovial sarcoma of the peripheral nerve (SSPN) and assess clinical and pathological factors and their contribution to survival and recurrence. METHODS Cases from 2 institutions collected in patients undergoing surgical intervention for SSPN. Systematic review including PubMed and Scopus databases were searched for related articles published from 1970 to December 2018. Eligibility criteria: (1) case reports or case series reporting on SSPN, (2) clinical course and/or pathological features of the tumor reported, and (3) articles published in English. RESULTS From patients treated at our institutions (13) the average follow-up period was 3.2 yr. Tumor recurrence was seen in 4 cases and death in 3. Systematic review of the literature yielded 44 additional cases with an average follow-up period of 3.6 yr. From pooled data, there were 10 recurrences and 7 deaths (20% and 14%, respectively). Adjuvant treatment used in 62.5% of cases. Immunohistochemical markers used in diagnosis varied widely; the most common are the following: Epithelial membrane antigen (EMA), cytokeratin, vimentin, cluster of differentiation (CD34), and transducin-like enhancer of split 1 (TLE1). Statistical analysis illustrated tumor size and use of chemotherapy to be negative predictors of survival. No other factors, clinically or from pathologist review, were correlated with recurrence or survival. CONCLUSION By combining cases from our institution with historical data and performing statistical analysis we show correlation between tumor size and death.


2020 ◽  
Vol 33 (10) ◽  
Author(s):  
Madhav Desai ◽  
Venkat Nutalapati ◽  
Sachin Srinivasan ◽  
Jihan Fathallah ◽  
Chandra Dasari ◽  
...  

SUMMARY Published studies have reported variable results on the association between duration of proton pump inhibitor (PPI) use and the risk of dementia. An extensive literature search was performed in PubMed, Embase, Google Scholar, and Cochrane for studies examining the risk of cognitive decline and dementia among PPI users versus non-PPI users in prospective studies. Retrospective database linkage studies, case reports, case series, editorials, uncontrolled cohort studies, cross-sectional studies, and review articles were excluded. Primary outcome was pooled hazard rate (HR) of any dementia among PPI users compared with non-PPI users. Secondary outcomes were pooled HR of Alzheimer’s dementia (AD) and risk with long-term PPI follow-up (more than 5 years) studies. Meta-analysis outcomes, heterogeneity (I2), and meta-regression (for the effect of covariates) were derived by statistical software R and Open meta-analyst. A total of six studies (one RCT and five prospective) with 308249 subjects, average age of 75.8 ± 5.2 years, and follow-up of 5 (range 1.5–11) years were included in the analysis. Pooled HR of any dementia was 1.16 (n = 6, 95% confidence interval (CI) = 0.86–1.47). Results remained unchanged when only studies with long-term PPI use (more than 5 years) were analyzed (n = 4, pooled HR 1.10, 95% CI 0.66–1.53). Finally, the pooled HR for AD was 1.06 (n = 3, 95% CI 0.70–1.41). There was substantial heterogeneity among inclusion studies (I2 = 93%). Meta-regression did not demonstrate a significant role of age at study start (P = 0.1) or duration of PPI use (P = 0.62) to incident dementia. The results of this systematic review and meta-analysis do not show a significant relationship between PPI use and dementia in prospective studies with at least a 5-year follow-up.


2017 ◽  
Vol 4 (4) ◽  
pp. 255-262
Author(s):  
Ryan Rhome ◽  
Isabelle M Germano ◽  
Ren-Dih Sheu ◽  
Sheryl Green

Abstract Background Growth hormone (GH)-secreting pituitary adenomas represent an uncommon subset of pituitary neoplasms. Stereotactic radiosurgery (SRS) and fractionated stereotactic radiotherapy (FSRT) have been used as primary or adjuvant treatment. The purpose of this study is to report the long-term tumor control and toxicity from our institution and to perform a systematic literature review of acromegaly patients treated with FSRT. Methods We retrospectively reviewed all patients treated with FSRT (median dose 50.4 Gray [Gy], range 50.4–54 Gy) between 2005 and 2012 who had: 1) GH-secreting adenoma with persistently elevated insulin growth factor-1 (IGF-1) despite medical therapy and 2) clinical follow up >3 years after FSRT. Patients were treated with modern FSRT planning techniques. Biochemical control was defined as IGF-1 normalization. Systematic review of the literature was performed for FSRT in acromegaly. Results With a median follow-up of 80 months, radiographic control was achieved in all 11 patients and overall survival was 100%. Long-term biochemical control was achieved in 10 patients (90.9%) with either FSRT alone (36.4%) or FSRT with continued medical management (45.5%). No patient experienced new hypopituitarism, cranial nerve dysfunctions, or visual deficits. Our systematic review found published rates of biochemical control and hypopituitarism vary, with uniformly good radiographic control and low incidence of visual changes. Conclusions Adjuvant FSRT offered effective long-term biochemical control and radiographic control, and there was a lower rate of complications in this current series. Review of the literature shows variations in published rates of biochemical control after FSRT for acromegaly, but low incidence of serious toxicities.


2020 ◽  
Author(s):  
Tung Hoang

AbstractBackgroundPrevious studies reported the recurrence of coronavirus disease 2019 (COVID-19) among discharge patients. This study aimed to examine the characteristic of COVID-19 recurrence cases by performing a systematic review and meta-analysis.MethodsA systematic search was performed in PubMed and Embase and gray literature up to September 17, 2020. A random-effects model was applied to obtain the pooled prevalence of disease recurrence among recovered patients and the prevalence of subjects underlying comorbidity among recurrence cases. The other characteristics were calculated based on the summary data of individual studies.ResultsA total of 41 studies were included in the final analysis, we have described the epidemiological characteristics of COVID-19 recurrence cases. Of 3,644 patients recovering from COVID-19 and being discharged, an estimate of 15% (95% CI, 12% to 19%) patients was re-positive with SARS-CoV-2 during the follow-up. This proportion was 14% (95% CI, 11% to 17%) for China and 31% (95% CI, 26% to 37%) for Korea. Among recurrence cases, it was estimated 39% (95% CI, 31% to 48%) subjects underlying at least one comorbidity. The estimates for times from disease onset to admission, from admission to discharge, and from discharge to RNA positive conversion were 4.8, 16.4, and 10.4 days, respectively.ConclusionThis study summarized up-to-date evidence from case reports, case series, and observational studies for the characteristic of COVID-19 recurrence cases after discharge. It is recommended to pay attention to follow-up patients after discharge, even if they have been in quarantine.


2021 ◽  
Vol 12 ◽  
Author(s):  
Max J. Dullaart ◽  
Marijn Kip ◽  
Adriana L. Smit ◽  
Inge Stegeman

Objectives: To systematically review studies on the effect of treatment of subjective tinnitus in children.Data Sources: We searched for studies in MEDLINE and EMBASE databases, after which additional studies were hand searched using Scopus databases. The methods are described in the study protocol, which has been registered in the PROSPERO register. PRISMA guidelines were followed in the reporting of this study.Eligibility Criteria: We considered for inclusion randomized controlled trials (RCTs), observational studies, case reports, and case series, with tinnitus as primary outcome in children (0–18 years old) with acute or chronic subjective tinnitus. We excluded studies in which both children and adults participated but outcomes were not specifically reported for children, as well as animal studies, studies with a non-original study design and studies about children with pulsatile or objective tinnitus.Data Selection: Two reviewers independently assessed studies for eligibility and quality, collected and extracted data. Statistical analyses were performed in case of homogeneous outcomes.Results: The search yielded a total of 4,447 studies. Of these, 147 eligible studies were selected. One case report and five observational studies met the eligibility criteria. Three studies applied counseling and (simplified-)TRT and reported improvement in tinnitus outcome in 68 out of 82 children after 3–6 months of treatment. Two studies used pharmacological treatments and reported improvement in 74 out of 86 patients after 10 days to 3 months of treatment. One study reported the outcome of biofeedback therapy, describing an improvement in tinnitus loudness and annoyance after 2 months of treatment.Conclusion: Due to the high risk of bias of the included studies, we cannot determine the effectiveness of the treatment of subjective tinnitus in children. Also, owing to brief follow-up periods, it is not possible to draw conclusions regarding long-term effects. Randomized controlled trials with longer follow-up periods are necessary to provide substantial evidence of the effects of therapies for children affected by tinnitus. https://www.crd.york.ac.uk/prospero/Systematic Review Registration:https://www.crd.york.ac.uk/prospero/, identifier [CRD42020178134].


Sign in / Sign up

Export Citation Format

Share Document