bluuebunny/crossref_metadata_2025_split huggingface dataset:
Effectiveness of pediatric integrative manual therapy in cervical movement limitation in infants with positional plagiocephaly: a randomized controlled trial
Authors: Pastor-Pons Iñaki , Hidalgo-García César , Lucha-López María Orosia , Barrau-Lalmolda Marta , Rodes-Pastor Iñaki , Rodríguez-Fernández Ángel Luis , Tricás-Moreno José Miguel 
Published: February 2021

Abstract
Background
Positional plagiocephaly (PP) is a cranial deformation frequent amongst children and consisting in a flattened and asymmetrical head shape. PP is associated with excessive time in supine and with congenital muscular torticollis (CMT). Few studies have evaluated the efficiency of a manual therapy approach in PP. The purpose of this parallel randomized controlled trial is to compare the effectiveness of adding a manual therapy approach to a caregiver education program focusing on active rotation range of motion (AROM) and neuromotor development in a PP pediatric sample.

Methods
Thirty-four children with PP and less than 28 week-old were randomly distributed into two groups. AROM and neuromotor development with Alberta Infant Motor Scale (AIMS) were measured. The evaluation was performed by an examiner, blinded to the randomization of the subjects. A pediatric integrative manual therapy (PIMT) group received 10-sessions involving manual therapy and a caregiver education program. Manual therapy was addressed to the upper cervical spine to mobilize the occiput, atlas and axis. The caregiver educational program consisted in exercises to reduce the positional preference and to stimulate motor development. The control group received the caregiver education program exclusively. To compare intervention effectiveness across the groups, improvement indexes of AROM and AIMS were calculated using the difference of the final measurement values minus the baseline measurement values. If the distribution was normal, the improvement indexes were compared using the Student t-test for independent samples; if not, the Mann-Whitney U test was used. The effect size of the interventions was calculated using Cohen’s d.

Results
All randomized subjects were analysed. After the intervention, the PIMT group showed a significantly higher increase in rotation (29.68 ± 18.41°) than the control group (6.13 ± 17.69°) (p = 0.001). Both groups improved the neuromotor development but no statistically significant differences were found. No harm was reported during the study.

Conclusion
The PIMT intervention program was more effective in increasing AROM than using only a caregiver education program.
The study has been retrospectively registered at clinicaltrials.gov, with identification number NCT03659032. Registration date: September 1, 2018.
Frailty status among older critically ill patients with severe acute kidney injury
Authors: Beaubien-Souligny William , Yang Alan , Lebovic Gerald , Wald Ron , Bagshaw Sean M. 
Published: February 2021

AbstractBackgroundFrailty status among critically ill patients with acute kidney injury (AKI) is not well described despite its importance for prognostication and informed decision-making on life-sustaining therapies. In this study, we aim to describe the epidemiology of frailty in a cohort of older critically ill patients with severe AKI, the outcomes of patients with pre-existing frailty before AKI and the factors associated with a worsening frailty status among survivors.MethodsThis was a secondary analysis of a prospective multicentre observational study that enrolled older (age > 65 years) critically ill patients with AKI. The clinical frailty scale (CFS) score was captured at baseline, at 6 months and at 12 months among survivors. Frailty was defined as a CFS score of ≥ 5. Demographic, clinical and physiological variables associated with frailty as baseline were described. Multivariable Cox proportional hazard models were constructed to describe the association between frailty and 90-day mortality. Demographic and clinical factors associated with worsening frailty status at 6 months and 12 months were described using multivariable logistic regression analysis and multistate models.ResultsAmong the 462 patients in our cohort, median (IQR) baseline CFS score was 4 (3–5), with 141 (31%) patients considered frail. Pre-existing frailty was associated with greater hazard of 90-day mortality (59% (n = 83) for frail vs. 31% (n = 100) for non-frail; adjusted hazards ratio [HR] 1.49; 95% CI 1.11–2.01,p = 0.008). At 6 months, 68 patients (28% of survivors) were frail. Of these, 57% (n = 39) were not classified as frail at baseline. Between 6 and 12 months of follow-up, 9 (4% of survivors) patients transitioned from a frail to a not frail status while 10 (4% of survivors) patients became frail and 11 (5% of survivors) patients died. In multivariable analysis, age was independently associated with worsening CFS score from baseline to 6 months (adjusted odds ratio [OR] 1.08; 95% CI 1.03–1.13,p = 0.003).ConclusionsPre-existing frailty is an independent risk factor for mortality among older critically ill patients with severe AKI. A substantial proportion of survivors experience declining function and worsened frailty status within one year.
A checklist-based method for improving COPD care for the elderly in general practice: study protocol for a cluster randomized controlled trial using electronic health records
Authors: Kowalczyk Anna , Zakowska Izabela , Andrzejewska Ewa , Kosiek Katarzyna , Godycki-Cwirko Maciek 
Published: February 2021

AbstractBackgroundThe third most frequent chronic condition, and the fourth most common cause of death, in Poland is chronic obstructive pulmonary disease (COPD). The diagnosis and treatment of COPD is the responsibility of the general practitioner (GP); the GP also serves as gatekeeper, referring patients to the other levels of public health care system when necessary. Undertreatment of COPD can result in a greater frequency of exacerbations and hospitalizations. Elderly patients require special attention due to the increased prevalence of COPD and systemic comorbidities. However, both the occurrence of exacerbations and the quality of life of the patients may be improved by developing and implementing guidelines for practice and ensuring their adherence. This proposal concerns the development of a checklist-based educational program to assist general practitioners in managing COPD patients.MethodsNo less than eighty-four general clinics in the Lodz region, Poland (28 clusters in each of three study arms), will be identified, randomized, and included in the trial. The trial will be based on anonymized data in electronic health records within the national public health care system.The educational intervention program will consist of GPs in two intervention arms being provided with a COPD management checklist: those in the first intervention arm with receive the checklist once at the beginning, while those in the second with receive it twice. The third (control) arm receives standard care without the checklist.The study used the International Code of Diseases (ICD)-10 for COPD. The primary aim is to determine the effect of interventions delivered to general practitioners (GPs) in primary health care. These interventions are aimed at decreasing the hospitalization of elderly patients with medical code J-44 (COPD) as the main reason for hospital admission.DiscussionThe results of this trial will be directly applicable to primary care in Poland and add new data to the growing body of evidence regarding interventions aimed at improving chronic illness care.Trial registrationThis trial has been registered with the Clinical Trials Protocol Registration System. Please see inClinicalTrial.govidentifier (NCT Number):NCT04301505. Registered on 10 March 2020.
ClinSV: clinical grade structural and copy number variant detection from whole genome sequencing data
Authors: Minoche Andre E. , Lundie Ben , Peters Greg B. , Ohnesorg Thomas , Pinese Mark , Thomas David M. , Zankl Andreas , Roscioli Tony , Schonrock Nicole , Kummerfeld Sarah , Burnett Leslie , Dinger Marcel E. , Cowley Mark J. 
Published: February 2021

AbstractWhole genome sequencing (WGS) has the potential to outperform clinical microarrays for the detection of structural variants (SV) including copy number variants (CNVs), but has been challenged by high false positive rates. Here we presentClinSV, a WGS based SV integration, annotation, prioritization, and visualization framework, which identified 99.8% of simulated pathogenic ClinVar CNVs > 10 kb and 11/11 pathogenic variants from matched microarrays. The false positive rate was low (1.5–4.5%) and reproducibility high (95–99%). In clinical practice,ClinSVidentified reportable variants in 22 of 485 patients (4.7%) of which 35–63% were not detectable by current clinical microarray designs.ClinSVis available athttps://github.com/KCCG/ClinSV.
Association of systemic lupus erythematosus autoantibody diversity with breast cancer protection
Authors: Shah Ami A. , Igusa Takeru , Goldman Daniel , Li Jessica , Casciola-Rosen Livia , Rosen Antony , Petri Michelle 
Published: February 2021

Abstract
Background
Epidemiologic data suggest that patients with systemic lupus erythematosus (SLE) have a lower risk of breast cancer than women in the general population. In light of mechanistic studies suggesting that anti-DNA antibodies have anti-cancer effects, we sought to examine breast cancer risk in autoantibody strata in a well-characterized SLE cohort.

Methods
SLE patients without a cancer diagnosis prior to entry in the Hopkins Lupus Cohort were studied (N = 2431). Overall and site-specific cancer incidence was calculated in racial strata and compared with the US Surveillance, Epidemiology and End Results (SEER) registry. Breast cancer incidence was further examined in autoantibody subsets. Patients were considered positive for an autoantibody if they were ever positive for a specificity during their disease course.

Results
Patients with SLE had a 37% lower risk of breast cancer (SIR 0.63, 95% CI 0.39–0.95). The risk of HPV-associated cancers (SIR 4.39, 95% CI 2.87–6.44) and thyroid cancer (SIR 2.27, 95% CI 1.04–4.30) was increased. Cancer risk varied by race, with breast cancer protection occurring in non-African Americans (SIR 0.29, 95% CI 0.11–0.63) and the increased risk of HPV-associated cancers occurring in African Americans (SIR 7.23, 95% CI 4.35–11.3). Breast cancer risk was decreased in patients ever positive for anti-dsDNA (SIR 0.55, 95% CI 0.29–0.96), anti-La (SIR 0.00, 95% CI 0.00–0.78), and lupus anticoagulant (SIR 0.37, 95% CI 0.10–0.94). Patients who were positive for fewer (0–2) SLE autoantibodies did not have a lower risk of breast cancer (SIR 0.84, 95% CI 0.47–1.39), but patients with 3+ autoantibodies had a 59% decreased risk (SIR 0.41, 95% CI 0.16–0.84).

Conclusions
Positivity for multiple SLE autoantibodies was associated with a lower risk of breast cancer, supporting the hypothesis that a highly diversified immune response may exert an anti-cancer effect against some cancers. Validation of racial differences in cancer risk in SLE is required to determine whether cancer screening strategies should be targeted to racial subgroups.
Increased aperiodic gamma power in young boys with Fragile X Syndrome is associated with better language ability
Authors: Wilkinson Carol L. , Nelson Charles A. 
Published: February 2021

Abstract
Background
The lack of robust and reliable clinical biomarkers in Fragile X Syndrome (FXS), the most common inherited form of intellectual disability, has limited the successful translation of bench-to-bedside therapeutics. While numerous drugs have shown promise in reversing synaptic and behavioral phenotypes in mouse models of FXS, none have demonstrated clinical efficacy in humans. Electroencephalographic (EEG) measures have been identified as candidate biomarkers as EEG recordings of both adults with FXS and mouse models of FXS consistently exhibit alterations in resting state and task-related activity. However, the developmental timing of these EEG differences is not known as thus far EEG studies have not focused on young children with FXS. Further, understanding how EEG differences are associated with core symptoms of FXS is crucial to successful use of EEG as a biomarker, and may improve our understanding of the disorder.

Methods
Resting-state EEG was collected from FXS boys with full mutation of Fmr1 (2.5–7 years old, n = 11) and compared with both age-matched (n = 12) and cognitive-matched (n = 12) typically developing boys. Power spectra (including aperiodic and periodic components) were compared using non-parametric cluster-based permutation testing. Associations between 30 and 50 Hz gamma power and cognitive, language, and behavioral measures were evaluated using Pearson correlation and linear regression with age as a covariate.

Results
FXS participants showed increased power in the beta/gamma range (~ 25–50 Hz) across multiple brain regions. Both a reduction in the aperiodic (1/f) slope and increase in beta/gamma periodic activity contributed to the significant increase in high-frequency power. Increased gamma power, driven by the aperiodic component, was associated with better language ability in the FXS group. No association was observed between gamma power and parent report measures of behavioral challenges, sensory hypersensitivities, or adaptive behaviors.

Limitations
The study sample size was small, although comparable to other human studies in rare-genetic disorders. Findings are also limited to males in the age range studied.

Conclusions
Resting-state EEG measures from this study in young boys with FXS identified similar increases in gamma power previously reported in adults and mouse models. The observed positive association between resting state aperiodic gamma power and language development supports hypotheses that alterations in some EEG measures may reflect ongoing compensatory mechanisms.
3D-surface scan based validated new measurement technique of femoral joint line reconstruction in total knee arthroplasty
Authors: Tuecking Lars-Rene , Ettinger Max , Nebel Dennis , Welke Bastian , Schwarze Michael , Windhagen Henning , Savov Peter 
Published: February 2021

Abstract
Purpose
This study aimed to validate a new joint line measurement technique in total knee arthroplasty for separated assessment of the medial and lateral femoral joint line alteration with 3D-surface scan technology. Separate assessment of the medial and lateral joint line alteration may improve TKA alignment assessment regarding to joint line restoration in kinematic alignment and use of robotic-assisted TKA surgery.

Methods
The medial and lateral joint line difference after TKA implantation on an artificial bone model was analyzed and compared with a 3D-scan and full femoral radiographs pre- and postoperatively. Radiographic analysis included the perpendicular distance between the most distal point of the medial and lateral condyle and the reproduced preoperative lateral distal femoral angle (LDFA). For evaluation of validity and reliability, radiographs were captured initially with true anteroposterior view and subsequently with combined flexion and rotation malpositioning. Reliability of the introduced measurement technique in between three observers was tested with intraclass correlation coefficient (ICC).

Results
Radiographic measurement showed a mean difference of 0.9 mm on the medial side and 0.6 mm on the lateral side when compared to the 3D-surface scan measurement. The reliability of measurement accuracy was ≤ 1 mm in x-rays with < 10° flexion error regardless to malrotation in these images. The ICC test showed very good reliability for the medial joint line evaluation and good reliability for lateral joint line evaluation (ICC 0.92, ICC 0.86 respectively).

Conclusion
The new introduced joint line measurement method showed a sufficient reliability, accuracy and precision. It provides separated information about medial and lateral joint line alteration in TKA surgery in absolute values.

Level of evidence
V - Experimental Study
Lipid profile of childhood cancer survivors and the effects of vitamin D supplementation: a prospective study
Authors: Kandil Shaimaa , Fouda Ashraf , Boujettif Khadijah , Salama Yasmin , Abdelmabood Suzy 
Published: February 2021

Abstract
Background
Childhood cancer survivors (CCS) have high prevalence of obesity vitamin D (VD) deficiency together with dyslipidemia. We aimed to evaluate lipid profile and the effect of VD supplementation in CCS.

Results
VD deficiency was more frequent among obese CCS and their lipid profiles, TC, and LDL-C levels were significantly higher compared to non-obese patients. After VD supplementation trials, VD levels increased among obese and non-obese children albeit it was significantly higher in non-obese subjects while the lipid profile of obese patients significantly decreased. Also, parathyroid hormone levels were persistently elevated among VD-deficient obese patients. Yet, the weight of obese patients remained unchanged.

Conclusion
VD deficiency was more prevalent among obese CCS. VD supplementation helped in normalization of the lipid profile of obese CCS. Regular measurement of PTH and 25OH-VD is recommended for CCS especially obese ones who may need VD supplementation.
Implementation and evaluation of an e-health innovation for personalized care for patients with amyotrophic lateral sclerosis (ALS): protocol for a participatory action research study
Authors:   on behalf of the Study Group ALS Home-monitoring and Coaching, Dontje M. L. , Kruitwagen - van Reenen E. , Visser-Meily J. M. A. , Beelen A. 
Published: February 2021

Abstract
Background
In the absence of a cure for amyotrophic lateral sclerosis (ALS), a progressive neurodegenerative disease, treatment consists of symptomatic management by a multidisciplinary healthcare team and is mainly aimed at optimizing patients’ quality of life. Because the course of the disease is often erratic and varies between patients, it is imperative for patients with ALS to be closely monitored. E-health innovations that can monitor disease progression remotely have great potential to tailor the care to the needs of individual patients with ALS. Therefore, the e-health innovation “ALS Home-monitoring and Coaching” was developed employing a user-centered design process and implemented at the University Medical Center Utrecht, the Netherlands in 2017. Because ALS Home-monitoring and Coaching was shown to be feasible and well received by patients and healthcare professionals at University Medical Centre Utrecht, we aim to implement this e-health innovation nationwide, starting with 10 ALS care teams in different rehabilitation settings spread across the Netherlands.

Methods
This research focuses on the implementation process and the user experiences with ALS Home-monitoring and Coaching of both patients and healthcare professionals. We will use a participatory action research approach, with the stakeholders involved in all stages of the implementation process. The implementation process model of Grol and Wensing was used to structure and support planning, execution and evaluation of the implementation strategy. The expected barriers and facilitators will be explored and identified in focus group settings using the Theoretical Domains Framework. After that, each team will develop their own action plan with strategies for how to resolve each barrier. The teams will include 5-10 ALS patients with whom they will test their implementation plan and provide care with ALS Home-monitoring and Coaching for approximately 3 months. Afterwards, the implementation and the user experiences will be evaluated with digital surveys based on the evaluation framework of Proctor (e.g., acceptability, adoption, appropriateness).

Discussion
Using implementation theories, this study will provide inside in factors influencing implementation outcomes and strategies that can be used to overcome barriers. This will enhance our understanding of how to successfully implement e-health innovations in multidisciplinary care in rehabilitation settings.

Trial registration
Trial NL8542 registered at Netherlands Trial Register (trialregister.nl) on 15th April 2020.
Impact of Risk-Adapted Therapy for Pediatric Hodgkin Lymphoma on Risk of Long-Term Morbidity: A Report From the Childhood Cancer Survivor Study
Authors: Oeffinger Kevin C. , Stratton Kayla L. , Hudson Melissa M. , Leisenring Wendy M. , Henderson Tara O. , Howell Rebecca M. , Wolden Suzanne L. , Constine Louis S. , Diller Lisa R. , Sklar Charles A. , Nathan Paul C. , Castellino Sharon M. , Barnea Dana , Smith Susan A. , Hutchinson Raymond J. , Armstrong Gregory T. , Robison Leslie L. 
Published: February 2021

PURPOSE To determine the incidence of serious chronic health conditions among survivors of pediatric Hodgkin lymphoma (HL), compare by era of therapy and by selected cancer therapies, and provide estimates of risks associated with contemporary therapy. METHODS Assessing 2,996 5-year HL survivors in the Childhood Cancer Survivor Study diagnosed from 1970 to 1999, we examined the cumulative incidence of severe to fatal chronic conditions (grades 3-5) using self-report conditions, medically confirmed subsequent malignant neoplasms, and cause of death based on the National Death Index. We used multivariable regression models to estimate hazard ratios (HRs) per decade and by key treatment exposures. RESULTS HL survivors were of a mean age of 35.6 years (range, 12-58 years). The cumulative incidence of any grade 3-5 condition by 35 years of age was 31.4% (95% CI, 29.2 to 33.5). Females were twice as likely (HR, 2.1; 95% CI, 1.8 to 2.4) to have a grade 3-5 condition compared with males. From the 1970s to the 1990s, there was a 20% reduction (HR, 0.8; 95% CI, 0.7 to 0.9) in decade-specific risk of a grade 3-5 condition ( P trend = .002). In survivors who had a recurrence and/or hematopoietic cell transplant, the risk of a grade 3-5 condition was substantially elevated, similar to that of survivors treated with high-dose, extended-field radiotherapy (HR, 1.2; 95% CI, 0.9 to 1.5). Compared with survivors treated with chest radiotherapy ≥ 35 Gy in combination with an anthracycline or alkylator, a contemporary regimen for low-intermediate risk HL was estimated to lead to a 40% reduction in risk of a grade 3-5 condition (HR, 0.6; 95% CI, 0.4 to 0.8). CONCLUSION This study demonstrates that risk-adapted therapy for pediatric HL has resulted in a significant reduction in serious long-term outcomes.