Post-Thrombotic Syndrome (PTS) in Children: A Systematic Review of Prevalence, Validity of Outcome Measures, and Prognostic Factors.

Blood ◽  
2009 ◽  
Vol 114 (22) ◽  
pp. 2978-2978 ◽  
Author(s):  
Neil A Goldenberg ◽  
Susan Kahn ◽  
Mark A. Crowther ◽  
Gili Kenet ◽  
Ulrike Nowak-Gottl ◽  
...  

Abstract 2978 Poster Board II-949 BACKGROUND: Post-thrombotic syndrome (PTS) is a condition of chronic venous insufficiency following deep venous thrombosis (DVT) that affects both adults and children. Nevertheless, investigation of PTS in children has lagged behind that in adults. OBJECTIVE: This systematic review was undertaken to summarize evidence from the pediatric literature on the prevalence of PTS, the validity of proposed measures of PTS, and prognostic factors for PTS in children. METHODS: A comprehensive Medline search was performed employing the following terms: “[pediatric OR children] AND [post-thrombotic syndrome OR post-phlebitic syndrome]”. Single case reports, narrative reviews, and commentaries were excluded. RESULTS: Seven case series and cross-sectional studies, nine registries and cohort studies, and one uncontrolled clinical trial were identified that met inclusion criteria; these studies reported on a total of 1316 children with DVT (Table 1). The prevalence of PTS differed substantially between observational and non-observational studies: 15% of 1042 children versus 46% of 274 children, respectively. No pediatric studies have evaluated quality of life (QOL) associated with PTS. Two reports from a single-institutional cohort have identified elevated levels of factor VIII and D-dimer and non-use of thrombolytic therapies as potentially prognostic of PTS in children, particularly among those with veno-occlusive thrombi; these are the only reports employing a PTS outcome measure that has been validated in children (the Manco-Johnson instrument). CONCLUSIONS: Overall, high-quality evidence on pediatric PTS is lacking. Collaborative prospective cohort studies and trials that use validated measures of pediatric PTS are needed to assess the incidence of, prognostic factors for, and QOL impact of PTS in children. Disclosures: No relevant conflicts of interest to declare.

Blood ◽  
2020 ◽  
Vol 136 (Supplement 1) ◽  
pp. 8-9
Author(s):  
Bader Allahyani ◽  
Abdullah Alanazi ◽  
Joshua Feder ◽  
Ewurabena Simpson

Background: Central venous catheters (CVCs) are the most frequent cause of thrombosis in acutely unwell neonates. The natural history CVC-RT remains unclear. The current guideline for symptomatic CVC-related thrombus suggests that anticoagulation likely leads to minimizing complications while the recommendation for pediatric patients with asymptomatic deep vein thrombosis (DVT) is equivocal. Objective: This systematic review was undertaken to summarize evidence from the pediatric literature on the prevalence of asymptomatic CVC-RT in the neonate and whether anticoagulation therapy (ACT) improves the outcomes of asymptomatic CVC-RT in the neonate. Methods: We searched CENTRAL, MEDLINE, Embase, CINAHL, the Web of Science, and clinical trial databases. We considered data from retrospective and prospective cohort studies, case series, and randomized controlled studies evaluating the prevalence and outcomes of CVC-RT following the use or non-use of ACT. Data were analyzed using Fisher exact and Chi-square statistics. Results: In total 762 articles were identified and screened, of which 16 articles included (1 RCT, 4 prospective studies and 8 retrospective cohort studies, and 3 case reports) that met inclusion criteria and contained extracted case data; these studies reported on a total of 1909 neonates with CVC have been screened for thrombosis. All studies were considered at moderate or high risk of bias. Tau2 and I2 suggested a high degree of heterogeneity. Ultimately, 309 cases were identified with the prevalence of asymptomatic CVC-RT was 16%. Notably, only 6.5% of neonates with symptomatic CVC-RT had undergone ultrasound (US) screening. The majority of the cases were premature neonates. 45% of the neonate had Umbilical Vein Catcher (UVC). 114 (41%) of the cases received ACT, of which none complicated with major bleeding. Overall there was no difference following the use or nonuse of ACT in patency of the asymptomatic CVC-RT, recurrence, and thrombosis related mortality. The frequency of each outcome according to therapy is shown in Table1. CONCLOSION: This systematic review has provided additional evidence supporting the favorable outcome of asymptomatic CVC-RT in the neonate regardless of the use of ACT. Further well-designed, prospective, multi-center clinical trials are needed to establish evidence-based treatment recommendations for neonates with asymptomatic CVC-RT. Figure Disclosures No relevant conflicts of interest to declare.


Author(s):  
Giovanna De Michele ◽  
Gianluigi Rosario Palmieri ◽  
Chiara Pane ◽  
Carmen Diletta Paola Dello Iacovo ◽  
Sandra Perillo ◽  
...  

Abstract Introduction Psychosis in Parkinson’s disease (PD) is common and consists of hallucinations, illusions, and delusions. Among the latter, delusional jealousy, also named Othello syndrome (OS), might impair the quality of life of both patients and their partners. We aimed to perform a systematic review and report a series of PD patients presenting with OS. Methods A systematic review research was performed in PubMed database, excluding non-English articles, single case reports, reviews and neuropathology articles, comments, and articles concerning OS associated with deep brain stimulation (DBS) and levodopa-carbidopa intestinal gel infusion. We also described eleven PD patients (9 M and 2 F) with OS, identified in a cohort of consecutive 153 patients, comparing them with eleven matched no OS (nOS) PD subjects taken from the same cohort. Results We included eight articles (four case series and four cross-sectional studies). OS resulted more common among males than females. We did not find higher levodopa dose and levodopa equivalent dose for dopamine agonists and for all anti-parkinsonian drugs in our OS group. In our case series, OS patients showed visual hallucinations (p=0.001) and a trend to have depression (p=0.080) more frequently than nOS ones. Conclusions OS is not a rare disorder in PD, probably due not only to abnormal dopaminergic stimulation but also to serotonergic dysfunction in biologically predisposed subjects. Visual hallucinations and other concomitant psychiatric diseases, in particular depression, might represent a risk factor for the OS development.


Rheumatology ◽  
2021 ◽  
Author(s):  
Vincenzo G Menditto ◽  
Giulia Rossetti ◽  
Diletta Olivari ◽  
Alessia Angeletti ◽  
Marco Rocchi ◽  
...  

Abstract Objective to analyze the available evidence about the use of rituximab (RTX) and other biologic agents in Eosinophilic Granulomatosis with Polyangiitis (EGPA) patients and to provide useful findings to inform the design of future, reliable clinical trials. Methods A systematic review was performed. A systematic search was conducted in PubMed/MEDLINE, Scopus, Web of Science and the Cochrane library databases and an extensive literature search on other biologic agents. Results 45 papers pertinent to our questions were found: 16 retrospective cohort studies, 8 case series, 3 prospective cohort studies and 18 single case reports, for a total of 368 EGPA patients. More than 80% of evaluable patients achieved complete or partial remission with a tendency towards a higher rate of complete response in pANCA positive subgroup. Conclusion Although the majority of the evaluable EGPA patients treated with RTX appears to achieve complete remission, we strongly believe that a number of sources of heterogeneity impairs a clear interpretation of results and limits their transferability in clinical practice. Differences in design, enrollment criteria, outcome definition and measurement make a comparison among data obtained from studies on RTX and other biologic agents unreliable.


2020 ◽  
Vol 19 (1) ◽  
Author(s):  
Daria Piacentino ◽  
Annalisa Maraone ◽  
Valentina Roselli ◽  
Isabella Berardelli ◽  
Massimo Biondi ◽  
...  

Abstract Background Preliminary studies have tested nicotine as a novel treatment for OCD patients who respond partially/incompletely or not at all to first and second-line treatment strategies, with the former represented by SSRIs or clomipramine, and the latter by switching to another SSRI, or augmentation with atypical antipsychotics, and/or combination with/switching to cognitive–behavioural therapy. Some studies found nicotine-induced reduction of obsessive thoughts and/or compulsive behaviour in OCD patients. We aimed to evaluate the efficacy of nicotine administration in OCD patients. Methods We searched the PubMed, ScienceDirect Scopus, CINHAL, Cochrane, PsycINFO/PsycARTICLES, and EMBASE databases from inception to the present for relevant papers. The ‘Preferred Reporting Items for Systematic Review and Meta-Analyses’ (PRISMA) standards were used. We included all studies focusing on the effects of nicotine administration on OCD patients’ obsessions or compulsions. Studies could be open-label, cross-sectional, randomized controlled trials, case series or case reports. Results A total of five studies could be included. Nicotine administration may ameliorate behavioural features and recurrent thoughts of severe, treatment-resistant OCD patients; however, in one study it was not associated with OC symptom improvement or cognitive enhancement across various executive function subdomains. Conclusions Although encouraging, the initial positive response from the use of nicotine in OCD needs testing in large controlled studies. This, however, raises ethical issues related to nicotine administration, due to its addiction potential, which were not addressed in the limited literature we examined. As an alternative, novel treatments with drugs able to mimic only the positive effects of nicotine could be implemented.


2020 ◽  
Vol 14 (1) ◽  
pp. 1-13 ◽  
Author(s):  
Cassandra E.L. Fairhead ◽  
Alexander Hampson ◽  
Louis Dwyer-Hemmings ◽  
Nikhil Vasdev

Background: It is estimated that between 50 and 89% of non-gonococcal urethritis is not caused by Chlamydia trachomatis. Associations between non-chlamydial non-gonococcal urethritis (NCNGU) with balanoposthitis, epididymo-orchitis and reactive arthritis have been suggested, but evidence to support these often-theoretical relationships is sparse and further investigation is called for. Concerns over increasing antimicrobial resistance has rendered the need for clarity over this question ever more pressing in recent years. A review of the current evidence on the complications of NCNGU in men is therefore urgently warranted. Objective: This systematic review summarizes and evaluates the available evidence that NCNGU, whether symptomatic or asymptomatic, causes the significant complications that are already well-recognized to be associated with non-gonococcal urethritis. These significant complications are epididymo-orchitis, balanoposthitis, and sexually-acquired reactive arthritis (Reiter's syndrome) including arthritis or conjunctivitis. Summary: We conducted a systematic review and qualitative synthesis using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis framework. Five databases (PubMed, EMBASE, Cumulative Index to Nursing and Allied Health Literature, PsycINFO, and British Nursing Index) were searched. We included studies that measured clinical outcome after diagnosis of NCNGU in men. Bias was assessed using variations of the Newcastle-Ottawa scale. Data were extracted and entered into a pre-written data abstraction proforma. Seven peer-review studies were included. This included 2 retrospective cohort studies, 1 case series, 2 case reports and 2 cross-sectional studies. The studies described and analyzed 3 types of complication: balanitis, posthitis and/or meatitis; reactive arthritis and/or conjunctivitis; and epididymitis. All studies reported one or more complications. Key Messages: This review identifies an important avenue for future research: while the available evidence suggests that NCNGU has the potential to cause significant complications in men, with the strongest evidence existing for balanitis, posthitis and/or meatitis, the nature and significance of these relationships is far from clear. The findings of this review suggest that prospective, adequately powered research into whether there is a causal link between NCNGU and significant clinical complications in men would be highly worthwhile. The findings of this review raise important questions about the utility of the term NCNGU in research and clinical practice.


Blood ◽  
2014 ◽  
Vol 124 (21) ◽  
pp. 5415-5415 ◽  
Author(s):  
Shiraj Sen ◽  
Eileen Marley ◽  
Harris V. Naina

Abstract Primary effusion lymphoma (PEL) is a human herpesvirus 8-associated non-Hodgkin lymphoma (NHL) that predominantly develops in serous body cavities leading to recurrent lymphomatous effusions. It is poorly understood and very uncommon, accounting for only 3% of all HIV-related NHL. While outcomes have improved in patients with HIV-associated NHL since the widespread use of HAART, the prognosis of PEL remains very poor and median survival is less than 6 months. This study reports our institution’s experience with PEL over the past decade. Between 2004 and 2014, we treated 8 patients with HIV-associated PEL. All patients were male. The median age was 46.5 years old (range 35-63), three were Caucasian, four were Hispanic, and one was African-American. The primary site of involvement was pericardial in 3 patients, pleural in 3 patients, and extra-cavitary in 2 patients. All patients were Ann Arbor stage IV at the time of diagnosis. The mean LDH and CD4 count at time of diagnosis was 625 U/L and 161 cells/mm3, respectively. Of the eight patients identified with PEL, four have achieved complete remission (CR). Prognostic factors associated with achieving CR include compliance with HAART therapy prior to PEL diagnosis as well as lower serum lactate dehydrogenase (LDH) levels (202±30 versus 1049±290, p=.03). Patients achieving CR also had a higher average CD4 count (228±93 versus 95±35, p=.22) at time of diagnosis. Median survival is nearly 6 years for those in CR whereas it was 32 days in patients that died (one from an acute stroke, one from septic shock, and two from progressive disease). All patients who achieved CR had previously diagnosed HIV whereas two of the four who died were previously undiagnosed with HIV. Interestingly, pericardial sites of involvement were associated with significantly better outcomes as compared to pleural sites in our case series, as well. There was no association between outcome and HIV viral load, duration of HIV infection prior to PEL diagnosis, or presence of AIDS-defining illnesses. The patients achieving CR were treated with HAART in addition to Hyper-CVAD (1a-3b) or 6 cycles of CHOP or EPOCH. In addition, two patients received bortezomib as initial therapy. Given its rarity, our knowledge of PEL relies heavily on case reports and small case series. Here we report our institution’s experience with PEL, identify LDH and CD4 as possible prognostic factors in PEL, and suggest bortezomib-based chemotherapy as an effective treatment option in HIV-associated PEL. Disclosures No relevant conflicts of interest to declare.


Author(s):  
Giulia Orilisi ◽  
Marco Mascitti ◽  
Lucrezia Togni ◽  
Riccardo Monterubbianesi ◽  
Vincenzo Tosco ◽  
...  

Background: COVID-19 disease first appeared in 2019 and quickly spread worldwide, causing a global pandemic. The oral cavity represents a target of SARS-CoV-2, and oral lesions are observed in both non-hospitalized and hospitalized patients. This systematic review aims to investigate the frequency of oral manifestations in COVID-19 hospitalized patients. Methods: An electronic search was conducted in PubMed, Scopus, and Web of Science databases, including articles published up to September 2021. The review protocol was based on PRISMA-P. The risk of bias of the studies was assessed using the Joana Briggs Institute. The certainty of evidence was assessed using the GRADE instrument. Results: Fifty-nine articles were included: 19 case reports, 17 case series, 2 case-control studies, 13 cross-sectional studies, 4 observational studies, and 4 retrospective studies. Oral ulcers, cheilitis, and tongue lesions were more common in patients before hospitalization, while perioral pressure ulcers, macroglossia, blisters, and oral candidiasis were more recurrent in patients during hospitalization. The first could be related directly to COVID-19, while the latter could be caused by medical devices, treatments, prone position, and immunological impairment. Conclusions: An accurate oral examination during the hospital admission of all confirmed COVID-19 cases is encouraged to recognize oral early manifestations and to apply appropriate treatments.


Blood ◽  
2018 ◽  
Vol 132 (Supplement 1) ◽  
pp. 3806-3806 ◽  
Author(s):  
Elissa Engel ◽  
Manuela Albisetti ◽  
Leonardo R. Brandao ◽  
Ernest Amankwah ◽  
Anthony Nguyen ◽  
...  

Abstract BACKGROUND: Post-thrombotic syndrome (PTS) is the most common long-term complication in pediatric deep venous thrombosis (DVT), affecting approximately 25% of children with an extremity DVT. PTS leads to a high physical, psychological and financial burden in affected patients. Although several risk factors have been associated with the development of pediatric PTS, few of them have been validated in the pediatric literature. A better understanding of the prognostic factors leading to PTS is a vital step for early identification of those children at greatest risk in order to develop risk-stratified interventions aimed at preventing this complication. AIM: To perform a systematic review and meta-analysis of available published evidence from the pediatric literature on prognostic factors for pediatric PTS. METHODS: A systematic search of MEDLINE, EMBASE, and the Cochrane Library from 1960 to December 2017 was performed. MeSH terms and search strategy employed were as follows: "postthrombotic syndrome" OR "postphlebitic syndrome" AND "all child 0-18 years" AND "young adult 19-24 years". A study was eligible for inclusion if it evaluated the development of PTS in pediatric patients (<21 years of age) with a confirmed extremity DVT and reported on at least one prognostic factor for the development of PTS. Single case reports, narrative reviews, and commentaries were excluded. Studies assessing the efficacy/safety of thrombolysis, and studies including patients >21 years of age with outcomes not reported by age group, were also excluded. Two reviewers independently screened all studies and extracted the data of interest. Data were analyzed using STATA v.15 statistical software. Meta-analyses were conducted for risk factors reported in at least three studies. Summary odds ratios (ORs) and 95% confidence intervals (CI) were calculated from the effect estimates from the individual studies using a random effects model. Statistical heterogeneity was quantified by I2 statistic. RESULTS: A total of 12 studies met the final inclusion criteria (Figure 1), nine cohort studies, two cross-sectional studies, and one case-control study. These studies reported a total of 1,160 patients with venous thromboembolism (VTE), of whom 938 (81%) were assessed for PTS (Table 1). Median age across studies ranged from 0.02 - 15.5 years. VTE was considered provoked in nearly 80% of patients. The most common reported risk factor for VTE was the presence of a central venous catheter (CVC, 54%) followed by congenital heart disease (26%). PTS was diagnosed in 46% (n=434) of patients with an extremity DVT. The median time from DVT diagnosis to PTS diagnosis ranged from 12 to 33 months across studies. Among studies reporting this information, mild PTS was most frequently diagnosed, followed by moderate and severe PTS (35%, 5% and 0.6% of patients respectively). Most common prognostic factors associated with PTS in individual studies included patient characteristics: age and gender; and DVT characteristics: recurrent DVT, symptomatic DVT, DVT degree of occlusion, and time between DVT diagnosis and PTS assessment. Three studies investigated the association of elevated factor VIII and d-dimer levels with PTS. Elevated levels of these biomarkers were found to be associated with development of adverse VTE outcomes in one study but this finding was not confirmed in the other studies. Meta-analysis of reported prognostic factors identified the presence of a CVC and occlusive DVT as significant risk factors for the development of pediatric PTS (OR= 1.8, 95%CI=1.08-2.98, and OR=1.89, 95%CI=1.04-3.46 respectively; Figure 2). CONCLUSION: Among 12 studies evaluating prognostic factors for PTS in children and meeting criteria for this meta-analysis, CVC-related DVT and complete occlusion were associated with pediatric PTS. Overall, high-quality evidence on pediatric PTS is lacking. Collaborative prospective cohort studies and trials that use validated pediatric PTS measures and standardized prognostic factor definitions are needed to better understand the risk factors associated with PTS. Disclosures No relevant conflicts of interest to declare.


2021 ◽  
Vol 34 (Supplement_1) ◽  
Author(s):  
Marina Orlandini ◽  
Maria Carolina Serafim ◽  
Letícia Datrino ◽  
Clara Santos ◽  
Luca Tristão ◽  
...  

Abstract   Megaesophagus progress to sigmoid megaesophagus (SM) in 10–15% of patients, presenting tortuosity and sigmoid colon aspect. Esophagectomy is the choice treatment but is associated with high complications and mortality rates. To avoid the esophagectomy inherent morbidity, several authors recommend Heller myotomy (HM) with pull-down technique for SM, mainly for patients with comorbidities and the elderly. This systematic review and meta-analysis is the first to analyze the effectiveness of HM for treating SM. Methods A systematic review was conducted in PubMed, Embase, Cochrane Library Central, Lilacs (BVS), and manual search of references. Inclusion criteria were: a) clinical trials, cohort studies, case series; b) patients with SM and esophageal diameter ≥ 6 cm; and c) patients undergoing primary myotomy. The exclusion criteria were: a) reviews, case reports, cross-sectional studies, editorials, letters, congress abstracts, full-text unavailability; b) animal studies, c) previous surgical treatment for achalasia; and d) pediatric studies. There were no restrictions on language or date of publication, and no filters were applied for the selection process. Random model and a 95% confidence interval (CI) were used. Results Sixteen articles were selected, encompassing 231 patients. The mean age ranged from 36 to 61 years old, and the mean follow-up ranged from 16 to 109 months. The analyzed outcomes include mortality, complications (pneumonia, pneumothorax, gastroesophageal reflux), need for reintervention (remyotomy, dilation and esophagectomy), and results classified as ‘good’ and ‘excellent’. Mortality rate was 0.035 (CI: 0.017–0.07; p &lt; 0.01). Complications rate was 0.08 (CI: 0.04–0.153; p = 0.01). Need for retreatment rate was 0.161 (CI: 0.053–0.399; p &lt; 0.01). Probability of good or excellent outcomes after myotomy was 0.762 (CI: 0.693–0.819; p &lt; 0,01). Conclusion Heller myotomy is an option for avoiding esophagectomy in achalasia, with a low morbimortality rate and good results. It is effective for most patients but will fail in a minority of patients and demand retreatment, be it a remyotomy, endoscopic treatment or esophagectomy.


2019 ◽  
Vol 8 (1) ◽  
Author(s):  
Berta Grau-Pujol ◽  
Marilia Massangaie ◽  
Jorge Cano ◽  
Carmen Maroto ◽  
Alcino Ndeve ◽  
...  

Abstract Background Neglected tropical diseases (NTDs) affect more than one billion people living in vulnerable conditions. In spite of initiatives recently contributing to fill NTDs gaps on national and local prevalence and distribution, more epidemiological data are still needed for effective control and elimination interventions. Main text Mozambique is considered one of the countries with highest NTDs burden although available data is scarce. This study aims to conduct a systematic review on published available data about the burden and distribution of the different NTDs across Mozambique since January 1950 until December 2018. We identified manuscripts from electronic databases (Pubmed, EmBase and Global Health) and paper publications and grey literature from Mozambique Ministry of Health. Manuscripts fulfilling inclusion criteria were: cross-sectional studies, ecological studies, cohorts, reports, systematic reviews, and narrative reviews capturing epidemiological information of endemic NTDs in Mozambique. Case-control studies, letters to editor, case reports and case series of imported cases were excluded. A total of 466 manuscripts were initially identified and 98 were finally included after the revision following PRISMA guidelines. Eleven NTDs were reported in Mozambique during the study span. Northern provinces (Nampula, Cabo Delgado, Niassa, Tete and Zambezia) and Maputo province had the higher number of NTDs detected. Every disease had their own report profile: while schistosomiasis have been continuously reported since 1952 until nowadays, onchocerciasis and cysticercosis last available data is from 2007 and Echinococcosis have never been evaluated in the country. Thus, both space and time gaps on NTDs epidemiology have been identified. Conclusions This review assembles NTDs burden and distribution in Mozambique. Thus, contributes to the understanding of NTDs epidemiology in Mozambique and highlights knowledge gaps. Hence, the study provides key elements to progress towards the control and interruption of transmission of these diseases in the country.


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