scholarly journals Heterogeneous development of children with Congenital Zika Syndrome-associated microcephaly

PLoS ONE ◽  
2021 ◽  
Vol 16 (9) ◽  
pp. e0256444
Author(s):  
Juan P. Aguilar Ticona ◽  
Nivison Nery ◽  
Simon Doss-Gollin ◽  
Claudia Gambrah ◽  
Millani Lessa ◽  
...  
Keyword(s):  
Motor Development ◽  
Head Circumference ◽  
Multivariable Analysis ◽  
Long Term Outcomes ◽  
Neurodevelopmental Outcomes ◽  
Neurological Exam ◽  
Congenital Zika Syndrome ◽  
Bayley Scales

Objective To describe the neurological and neurodevelopmental outcomes of children with Congenital Zika Syndrome (CZS) associated microcephaly beyond 2 years of age. Method We followed children with CZS-associated microcephaly in an outpatient clinic in Salvador, Brazil. Neurological and neurodevelopmental assessments were performed using the Hammersmith Infant Neurological Examination (HINE) and Bayley Scales of Infant and Toddler Neurodevelopment (Bayley-III) respectively. Results Of the 42 children included, 19 were male (45.2%); median (interquartile range) age at neurological evaluation was 28 (25–32) months, and 36 (85.7%) had severe microcephaly. HINE and Bayley-III results were completed for 35/42 (83.3%) and 33/42 (78.5%) children respectively. Bayley-III identified a severe developmental delay in 32/33 (97.0%) children while 1/33 (3.0%) had only a mild delay. In the multivariable analysis, we found that Bayley-III and HINE scores were correlated. Better HINE scores were associated with higher Bayley-III cognitive raw scores (β = 0.29; CI 95% = 0.02–0.57) and motor raw scores (β = 0.43; CI 95% = 0.04–0.82) after adjusting for head circumference, prematurity, and age at neurodevelopmental evaluation. Furthermore, we found that greater head circumference at follow up was associated with higher cognitive (β = 1.27; CI 95% = 0.01–2.53) and motor raw scores (β = 2.03; CI 95% = 0.25–3.81). Conclusion Children with CZS-associated microcephaly demonstrate severe neurodevelopmental delays and slower growth rates than their peers over time. Still, they have remarkably heterogeneous neurodevelopmental profiles according to neurological exam scores which correlate with their long-term outcomes. We found that HINE scores effectively captured the heterogeneity of neurological capabilities among these children and could be predictive of cognitive and motor development progress.

Antiphospholipid Antibody Profile Stability Over Time: Prospective Results from APS ACTION Clinical Database and Repository

2020 ◽  
pp. jrheum.200513
Author(s):  
Elena Gkrouzman ◽  
Ecem Sevim ◽  
Jackie Finik ◽  
Danieli Andrade ◽  
Vittorio Pengo ◽  
...  
Keyword(s):  
Multivariable Analysis ◽  
Primary Objective ◽  
Antiphospholipid Antibody ◽  
Long Term Outcomes ◽  
Glycoprotein I ◽  
Baseline Characteristics ◽  
Stored Blood ◽  
Over Time

Objective APS ACTION Registry studies long-term outcomes in persistently antiphospholipid antibody (aPL)-positive patients. Our primary objective was to determine whether clinically meaningful aPL profiles at baseline remain stable over time. Our secondary objectives were to determine a) whether baseline characteristics differ between patients with stable and unstable aPL profiles, and b) predictors of unstable aPL profiles over time. Methods Clinically meaningful aPL profile was defined as positive lupus anticoagulant (LA) test and/or anticardiolipin (aCL)/anti-β2 glycoprotein-I (aβ2GPI) IgG/M ≥40 U. Stable aPL profile was defined as a clinically meaningful aPL profile in at least two-thirds of follow-up measurements. Generalized linear mixed models with logit link were used for primary objective analysis. Results Of 472 patients with clinically meaningful aPL profile at baseline (median follow up: 5.1 years), 366/472 (78%) patients had stable aPL profiles over time, 54 (11%) unstable; and 52 (11%) inconclusive. Time did not significantly affect odds of maintaining a clinically meaningful aPL profile at follow-up in univariate (p=0.906) and multivariable analysis (p=0.790). Baseline triple aPL positivity decreased (Odds Ratio [OR] 0.25, 95% Confidence Interval [CI] 0.10-0.64, p=0.004) and isolated LA test positivity increased (OR 3.3, 95% CI 1.53-7.13, p=0.002) the odds of an unstable aPL profile over time. Conclusion Approximately 80% of our international cohort patients with clinically meaningful aPL profile at baseline maintain such at a median follow-up of five years; triple aPL-positivity increase the odds of a stable aPL profile. These results will guide future validation studies of stored blood samples through APS ACTION Core Laboratories.

Outcomes associated with secondary implant removal and arthrolysis in patients with a painful stiff shoulder after proximal humeral fracture fixation

2022 ◽  
Vol 104-B (1) ◽  
pp. 157-167
Author(s):  
Navnit S. Makaram ◽  
Ewan B. Goudie ◽  
C. Michael Robinson
Keyword(s):  
Proximal Humerus ◽  
Plate Fixation ◽  
Multivariable Analysis ◽  
Long Term Results ◽  
Long Term Outcomes ◽  
Patient Reported ◽  
Long Term Follow Up ◽  
Manual Occupation

Aims Open reduction and plate fixation (ORPF) for displaced proximal humerus fractures can achieve reliably good long-term outcomes. However, a minority of patients have persistent pain and stiffness after surgery and may benefit from open arthrolysis, subacromial decompression, and removal of metalwork (ADROM). The long-term results of ADROM remain unknown; we aimed to assess outcomes of patients undergoing this procedure for stiffness following ORPF, and assess predictors of poor outcome. Methods Between 1998 and 2018, 424 consecutive patients were treated with primary ORPF for proximal humerus fracture. ADROM was offered to symptomatic patients with a healed fracture at six months postoperatively. Patients were followed up retrospectively with demographic data, fracture characteristics, and complications recorded. Active range of motion (aROM), Oxford Shoulder Score (OSS), and EuroQol five-dimension three-level questionnaire (EQ-5D-3L) were recorded preoperatively and postoperatively. Results A total of 138 patients underwent ADROM; 111 patients were available for long-term follow-up at a mean of 10.9 years (range 1 to 20). Mean age was 50.8 years (18 to 75);79 (57.2%) were female. Mean time from primary ORPF to ADROM was 11.9 months (6 to 19). Five patients developed superficial wound infection; ten developed symptomatic osteonecrosis/post-traumatic arthrosis (ON/PTA); four underwent revision arthrolysis. Median OSS improved from 17 (interquartile range (IQR) 12.0 to 22.0) preoperatively to 40.0 (IQR 31.5 to 48.0) postoperatively, and 39.0 (IQR 31.5 to 46.5) at long-term follow-up (p < 0.001). Median EQ-5D-3L improved from 0.079 (IQR -0.057 to 0.215) to 0.691 (IQR 0.441 to 0.941) postoperatively, and 0.701 (IQR 0.570 to 0.832) at long-term follow-up (p < 0.001). We found that aROM improved in all planes (p < 0.001). Among the variables assessed on multivariable analysis, a manual occupation, worsening Charlson Comorbidity Index and increasing socioeconomic deprivation were most consistently predictive of worse patient-reported outcome scores. Patients who subsequently developed ON/PTA reported significantly worse one-year and late OSS. Conclusion ADROM in patients with persistent symptomatic stiffness following ORPF can achieve excellent short- and long-term outcomes. More deprived patients, those in a manual occupation, and those with worsening comorbidities have worse outcomes following ADROM. Cite this article: Bone Joint J 2022;104-B(1):157–167.

Active Surveillance for Incidental (cT1a/b) Prostate Cancer: Long-Term Outcomes of the Prospective Noninterventional HAROW Study

2021 ◽  
pp. 1-8
Author(s):  
Jan Herden ◽  
Andreas Schwarte ◽  
Edith A. Boedefeld ◽  
Lothar Weissbach
Keyword(s):  
Prostate Cancer ◽  
Active Surveillance ◽  
Multivariable Analysis ◽  
Low Risk ◽  
Long Term Outcomes ◽  
Invasive Treatment ◽  
Incidental Prostate Cancer ◽  
Free Survival

<b><i>Introduction:</i></b> Optimal treatment for incidental prostate cancer (IPC) after surgical treatment for benign prostate obstruction is still debatable. We report on long-term outcomes of IPC patients managed with active surveillance (AS) in a German multicenter study. <b><i>Methods:</i></b> HAROW (2008–2013) was designed as a noninterventional, prospective, health-service research study for patients with localized prostate cancer (≤cT2), including patients with IPC (cT1a/b). A follow-up examination of all patients treated with AS was carried out. Overall, cancer-specific, and metastasis-free survival and discontinuation rates were determined. <b><i>Results:</i></b> Of 210 IPC patients, 68 opted for AS and were available for evaluation. Fifty-four patients had cT1a category and 14 cT1b category. Median follow-up was 7.7 years (IQR: 5.7–9.1). Eight patients died of which 6 were still under AS or watchful waiting (WW). No PCa-specific death could be observed. One patient developed metastasis. Twenty-three patients (33.8%) discontinued AS changing to invasive treatment: 12 chose radical prostatectomy, 7 radiotherapy, and 4 hormonal treatment. Another 19 patients switched to WW. The Kaplan-Meier estimated 10-year overall, cancer-specific, metastasis-free, and intervention-free survival was 83.8% (95% CI: 72.2–95.3), 100%, 98.4% (95% CI: 95.3–99.9), and 61.0% (95% CI: 47.7–74.3), respectively. In multivariable analysis, age (RR: 0.97; <i>p</i> &#x3c; 0.001), PSA density ≥0.2 ng/mL<sup>2</sup> (RR: 13.23; <i>p</i> &#x3c; 0.001), and PSA ≥1.0 ng/mL after surgery (RR: 5.19; <i>p</i> = 0.016) were significantly predictive for receiving an invasive treatment. <b><i>Conclusion:</i></b> In comparison with other AS series with a general low-risk prostate cancer population, our study confirmed the promising survival outcomes for IPC patients, whereas discontinuation rates seem to be lower for IPC. Thus, IPC patients at low risk of progression may be good candidates for AS.

scholarly journals Clinical outcomes in pediatric intestinal failure: a meta-analysis and meta-regression

2019 ◽  
Vol 110 (2) ◽  
pp. 430-436 ◽  
Author(s):  
Aureliane Chantal Stania Pierret ◽  
James Thomas Wilkinson ◽  
Matthias Zilbauer ◽  
Jake Peter Mann
Keyword(s):  
Small Bowel ◽  
Mortality Rate ◽  
Meta Analysis ◽  
Intestinal Failure ◽  
Long Term Outcomes ◽  
Neurodevelopmental Outcomes ◽  
Small Bowel Length ◽  
Meta Regression

ABSTRACT Background Intestinal failure (IF) is associated with significant morbidity and mortality, yet specific parameters that determine medium- and long-term outcomes remain ill defined. Objective The aim of this study was to determine the long-term outcomes in childhood IF and identify patient characteristics associated with clinical endpoints. Design MEDLINE and EMBASE were searched for cohorts of >10 pediatric-onset IF patients with >12 mo follow-up. Random-effects meta-analysis and meta-regression weighted by follow-up duration were used to calculate clinical outcome rates and patient factors associated with outcomes. Primary outcome was mortality rate; secondary outcomes included neurodevelopmental status, transplantation, IF-associated liver disease (IFALD), enteral autonomy, and sepsis. Results In total, 175 cohorts (9318 patients and 34,549 y follow-up) were included in the meta-analysis. Overall mortality was 5.2% per y (95% CI: 4.3, 6.0) and was associated with sepsis and IFALD on meta-regression. Mortality rate improved with time from 5.9% per y pre-2000 to 4.5% per y post-2005. Sepsis rate was also predictive of IFALD and liver failure. Enteral autonomy was associated with small bowel length but not presence of ileo-cecal valve. There was a relative lack of data on neurodevelopmental outcomes. Conclusions Sepsis is the primary modifiable factor associated with mortality and liver failure, whereas enteral autonomy correlates with small-bowel length. No clear parameters have been identified that accurately predict neurodevelopmental outcomes, and hence further research is needed. Together, our findings are helpful for parental counseling and resource planning, and support targeting reduction in sepsis.

scholarly journals Per cent low attenuation volume and fractal dimension of low attenuation clusters on CT predict different long-term outcomes in COPD

Thorax ◽  
2020 ◽  
Vol 75 (2) ◽  
pp. 116-122 ◽  
Author(s):  
Kaoruko Shimizu ◽  
Naoya Tanabe ◽  
Nguyen Van Tho ◽  
Masaru Suzuki ◽  
Hironi Makita ◽  
...  
Keyword(s):  
Fractal Dimension ◽  
Smoking Status ◽  
Multivariable Analysis ◽  
Ct Scans ◽  
Rapid Decline ◽  
Long Term Outcomes ◽  
Poor Clinical Outcome ◽  
Long Term Mortality

BackgroundFractal dimension (D) characterises the size distribution of low attenuation clusters on CT and assesses the spatial heterogeneity of emphysema that per cent low attenuation volume (%LAV) cannot detect. This study tested the hypothesis that %LAV and D have different roles in predicting decline in FEV1, exacerbation and mortality in patients with COPD.MethodsChest inspiratory CT scans in the baseline and longitudinal follow-up records for FEV1, exacerbation and mortality prospectively collected over 10 years in the Hokkaido COPD Cohort Study were examined (n=96). The associations between CT measures and long-term outcomes were replicated in the Kyoto University cohort (n=130).ResultsIn the Hokkaido COPD cohort, higher %LAV, but not D, was associated with a greater decline in FEV1 and 10-year mortality, whereas lower D, but not %LAV, was associated with shorter time to first exacerbation. Multivariable analysis for the Kyoto University cohort confirmed that lower D at baseline was independently associated with shorter time to first exacerbation and that higher LAV% was independently associated with increased mortality after adjusting for age, height, weight, FEV1 and smoking status.ConclusionThese well-established cohorts clarify the different prognostic roles of %LAV and D, whereby lower D is associated with a higher risk of exacerbation and higher %LAV is associated with a rapid decline in lung function and long-term mortality. Combination of %LAV and fractal D may identify COPD subgroups at high risk of a poor clinical outcome more sensitively.

Prediction of ventilator-associated pneumonia outcomes according to the early microbiological response: a retrospective observational study

2021 ◽  
pp. 2100620
Author(s):  
Adrian Ceccato ◽  
Cristina Dominedò ◽  
Miquel Ferrer ◽  
Ignacio Martin-Loeches ◽  
Enric Barbeta ◽  
...  
Keyword(s):  
Retrospective Analysis ◽  
Multivariable Analysis ◽  
Ventilator Associated Pneumonia ◽  
Long Term Outcomes ◽  
Microbiological Criteria ◽  
Short And Long Term ◽  
Current Guidelines

Ventilator-associated pneumonia is a leading infectious cause of morbidity in critically ill patients; yet current guidelines offer no indications for follow-up cultures.We aimed to evaluate the role of follow-up cultures and microbiological response 3 days after diagnosing ventilator-associated pneumonia as predictors of short- and long-term outcomes.We performed a retrospective analysis of a cohort prospectively collected from 2004 to 2017. Ventilator-associated pneumonia was diagnosed based on clinical, radiographic, and microbiological criteria. For microbiological identification, a tracheobronchial aspirate was performed at diagnosis and repeated after 72 h. We defined three groups when comparing the two tracheobronchial aspirate results: persistence, superinfection, and eradication of causative pathogens.One-hundred-fifty-seven patients were enrolled in the study, among whom microbiological persistence, superinfection, and eradication was present in 67 (48%), 25 (16%), and 65 (41%), respectively, after 72hs. Those with superinfection had the highest mortalities in the intensive care unit (p=0.015) and at 90 days (p=0.036), while also having the fewest ventilation-free days (p=0.024). Multivariable analysis revealed shock at VAP diagnosis (odds ratios [OR] 3.43; 95% confidence interval [CI] 1.25 to 9.40), Staphylococcus aureus isolation at VAP diagnosis (OR 2.87; 95%CI 1.06 to 7.75), and hypothermia at VAP diagnosis (OR 0.67; 95%CI 0.48 to 0.95, per +1°C) to be associated with superinfection.Our retrospective analysis suggests that ventilator-associated pneumonia short-term and long-term outcomes may be associated with superinfection in follow-up cultures. Follow-up cultures may help guiding antibiotic therapy and its duration. Further prospective studies are necessary to verify our findings.

scholarly journals Long-Term Outcomes and Responses to Retreatment in Patients With Melanoma Treated With PD-1 Blockade

2020 ◽  
Vol 38 (15) ◽  
pp. 1655-1663 ◽  
Author(s):  
Allison Betof Warner ◽  
Jessica S. Palmer ◽  
Alexander N. Shoushtari ◽  
Debra A. Goldman ◽  
Katherine S. Panageas ◽  
...  
Keyword(s):  
Treatment Failure ◽  
Single Agent ◽  
Multivariable Analysis ◽  
Complete Response ◽  
Median Number ◽  
Long Term Outcomes ◽  
Duration Of Treatment ◽  
Unresectable Stage

PURPOSE To analyze long-term outcomes after treatment discontinuation of anti–programmed death-1 (anti–PD-1) therapy in a cohort of patients with melanoma with the longest follow-up yet available to our knowledge, including a majority of patients treated outside of a clinical trial. We also assessed efficacy of retreatment with anti–PD-1 therapy with or without ipilimumab in relapsing patients. METHODS We retrospectively analyzed all patients with nonuveal, unresectable stage III/IV melanoma treated with single-agent anti–PD-1 therapy at Memorial Sloan Kettering from 2009-2018 who had discontinued treatment and had at least 3 months of follow-up after discontinuation (n = 396). Overall survival for patients with complete response (CR) was calculated from time of CR. Time to treatment failure for patients with CR was time from CR to the next melanoma treatment or death. RESULTS CRs were seen in 102 of 396 patients (25.8%). The median number of months of treatment after CR was zero (range, stopped before CR to 26 months after CR). With a median follow-up of 21.1 months from time of CR in patients who did not relapse, the probability of being alive and not needing additional melanoma therapy at 3 years was 72.1%. There was no significant association between treatment duration and relapse risk. In multivariable analysis, CR was associated with M1b disease and cutaneous versus mucosal or acral primaries. Among the 78 patients (of 396) retreated after disease progression, response was seen in 5 of 34 retreated patients with single-agent anti–PD-1 therapy and 11 of 44 patients escalated to anti–PD-1 plus ipilimumab. CONCLUSION In our cohort, most patients discontinued treatment at the time of CR. Most CRs were durable but the probability of treatment failure was 27% at 3 years. Responses to retreatment were infrequent. The optimal duration of treatment after CR is not yet established.

Periprocedural risk and long-term outcome of intracranial angioplasty based on a single-centre experience

VASA ◽  
2013 ◽  
Vol 42 (4) ◽  
pp. 264-274
Author(s):  
Dagmar Krajíčková ◽  
Antonín Krajina ◽  
Miroslav Lojík ◽  
Martina Mulačová ◽  
Martin Vališ
Keyword(s):  
Death Rate ◽  
Long Term Outcomes ◽  
Single Centre ◽  
Intracranial Atherosclerotic Stenosis ◽  
Atherosclerotic Stenosis ◽  
Angioplasty And Stenting ◽  
Aggressive Medical Management ◽  
Stroke Death

Background: Intracranial atherosclerotic stenosis is a major cause of stroke and yet there are currently no proven effective treatments for it. The SAMMPRIS trial, comparing aggressive medical management alone with aggressive medical management combined with intracranial angioplasty and stenting, was prematurely halted when an unexpectedly high rate of periprocedural events was found in the endovascular arm. The goal of our study is to report the immediate and long-term outcomes of patients with ≥ 70 % symptomatic intracranial atherosclerotic stenosis treated with balloon angioplasty and stent placement in a single centre. Patients and methods: This is a retrospective review of 37 consecutive patients with 42 procedures of ballon angioplasty and stenting for intracranial atherosclerotic stenosis (≥ 70 % stenosis) treated between 1999 and 2012. Technical success (residual stenosis ≤ 50 %), periprocedural success (no vascular complications within 72 hours), and long-term outcomes are reported. Results: Technical and periprocedural success was achieved in 90.5 % of patients. The within 72 hours periprocedural stroke/death rate was 7.1 % (4.8 % intracranial haemorrhage), and the 30-day stroke/death rate was 9.5 %. Thirty patients (81 %) had clinical follow-up at ≥ 6 months. During follow-up, 5 patients developed 6 ischemic events; 5 of them (17 %) were ipsilateral. The restenosis rate was 27 %, and the retreatment rate was 12 %. Conclusions: Our outcomes of the balloon angioplasty/stent placement for intracranial atherosclerotic stenosis are better than those in the SAMMPRIS study and compare favourably with those in large registries and observational studies.

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