bluuebunny/crossref_metadata_2025_split huggingface dataset:
Efficacy of conservative treatment for spastic cerebral palsy children with equinus gait: a systematic review and meta-analysis
Authors: Klaewkasikum Krongkaew , Patathong Tanyaporn , Woratanarat Patarawan , Woratanarat Thira , Thadanipon Kunlawat , Rattanasiri Sasivimol , Thakkinstian Ammarin 
Published: September 2022

AbstractBackgroundComparisons between various conservative managements of spastic equinus deformity in cerebral palsy demonstrated limited evidences, to evaluate the efficacy of conservative treatment among cerebral palsy children with spastic equinus foot regarding gait and ankle motion.MethodsStudies were identified from PubMed and Scopus up to February 2022. Inclusion criteria were randomized controlled trial (RCT), conducted in spastic cerebral palsy children with equinus deformity, aged less than 18 years, compared any conservative treatments (Botulinum toxin A; BoNT-A, casting, physical therapy, and orthosis), and evaluated gait improvement (Physician Rating Scale or Video Gait Analysis), Observational Gait Scale, Clinical Gait Assessment Score, ankle dorsiflexion (ankle dorsiflexion at initial contact, and passive ankle dorsiflexion), or Gross Motor Function Measure. Any study with the participants who recently underwent surgery or received BoNT-A or insufficient data was excluded. Two authors were independently selected and extracted data. Risk of bias was assessed using a revised Cochrane risk-of-bias tool for randomized trials. I2was performed to evaluate heterogeneity. Risk ratio (RR), the unstandardized mean difference (USMD), and the standardized mean difference were used to estimate treatment effects with 95% confidence interval (CI).ResultsFrom 20 included studies (716 children), 15 RCTs were eligible for meta-analysis (35% had low risk of bias). BoNT-A had higher number of gait improvements than placebo (RR 2.64, 95% CI 1.71, 4.07,I2 = 0). Its combination with physical therapy yielded better passive ankle dorsiflexion at knee extension than physical therapy alone (USMD = 4.16 degrees; 95% CI 1.54, 6.78,I2 = 36%). Casting with or without BoNT-A had no different gait improvement and ankle dorsiflexion at knee extension when compared to BoNT-A. Orthosis significantly increased ankle dorsiflexion at initial contact comparing to control (USMD 10.22 degrees, 95 CI% 5.13, 15.31,I2 = 87%).ConclusionBoNT-A and casting contribute to gait improvement and ankle dorsiflexion at knee extension. BoNT-A specifically provided gait improvement over the placebo and additive effect to physical therapy for passive ankle dorsiflexion. Orthosis would be useful for ankle dorsiflexion at initial contact.Trial registrationPROSPERO number CRD42019146373.
Do different multi‐segment foot models detect the same changes in kinematics when wearing foot orthoses?
Authors: Klein Tomas , Chapman Graham J. , Lastovicka Ondrej , Janura Miroslav , Richards Jim 
Published: September 2022

AbstractBackgroundDifferent multi‐segment foot models have been used to explore the effect of foot orthoses. Previous studies have compared the kinematic output of different multi‐segment foot models, however, no study has explored if different multi‐segment foot models detect similar kinematic changes when wearing a foot orthoses. The aim of this study was to compare the ability of two different multi‐segment foot models to detect kinematic changes at the hindfoot and forefoot during the single and double support phases of gait when wearing a foot orthosis.MethodsFoot kinematics were collected during walking from a sample of 32 individuals with and without a foot orthosis with a medial heel bar using an eight‐camera motion capture system. The Oxford Foot Model (OFM) and a multi‐segment foot model using the Calibrated Anatomical System Technique (CAST) were applied simultaneously. Vector field statistical analysis was used to explore the kinematic effects of a medial heel bar using the two models, and the ability of the models to detect any changes in kinematics was compared.ResultsFor the hindfoot, both models showed very good agreement of the effect of the foot orthosis across all three anatomical planes during the single and double support phases. However, for the forefoot, the level of agreement between the models varied with both models showing good agreement of the effect in the coronal plane but poorer agreement in the transverse and sagittal planes.ConclusionsThis study showed that while consistency exists across both models for the hindfoot and forefoot in the coronal plane, the forefoot in the transverse and sagittal planes showed inconsistent responses to the foot orthoses. This should be considered when interpreting the efficacy of different interventions which aim to change foot biomechanics.
Effects of vitrified cryopreservation duration on IVF and neonatal outcomes
Authors: Mao Yuling , Tang Ni , Luo Yanfen , Yin Ping , Li Lei 
Published: September 2022

AbstractBackgroundIn this study, we aimed to evaluate the impact of the duration of cryopreservation storage on embryo viability, implantation competence, pregnancy outcome and neonatal outcomes.MethodsWe retrospectively evaluated the outcomes of patients who underwent IVF with vitrified cryopreserved embryos between January 2004 and August 2019 by following the first frozen embryo transfer cycles within the study period. A total of 31,143 patients met the inclusion criteria and were grouped according to the embryo storage time as follows: Group 1 (n = 20,926),1–90 days; Group 2 (n = 6,472), 91–180 days; Group 3 (n = 2,237), 181–365 days; Group 4 (n = 746), 366–730 days; and Group 5 (n = 762), > 731 days.ResultsThe embryo survival rate decreased significantly with longer durations of cryopreservation. The highest and lowest survival rate was recorded in Group 1 and Group 5, respectively (34853/35338; 98.63% vs. 1281/1801; 71.13%;P < 0.01). The human chorionic gonadotropin (HCG) detection and clinical pregnancy rate was highest in Group 1 (57.85% and 55. 26%, respectively;P < 0.01). Short-term cryopreservation (≤ 3 months) is associated with higher rates of clinical pregnancy. There were no significant differences in neonatal birth weight, neonatal height and congenital anomalies among the groups (P > 0. 05).ConclusionThe prolonged storage time of vitrified embryos negatively affected survival rate and clinical pregnancy rate. It did not have a significant influence on neonatal health. This study provides new findings about the relationship between prolonged storage time of vitrified embryos and clinical outcomes and offers evidence for the safety of using long-stored embryos after vitrification.
Congenital hypopituitarism and multiple midline defects in a newborn with non-familial Cat Eye syndrome
Authors: Serra Gregorio , Giambrone Clara , Antona Vincenzo , Cardella Francesca , Carta Maurizio , Cimador Marcello , Corsello Giovanni , Giuffrè Mario , Insinga Vincenzo , Maggio Maria Cristina , Pensabene Marco , Schierz Ingrid Anne Mandy , Piro Ettore 
Published: September 2022

AbstractBackgroundCat eye syndrome (CES) is a rare chromosomal disease, with estimated incidence of about 1 in 100,000 live newborns. The classic triad of iris coloboma, anorectal malformations, and auricular abnormalities is present in 40% of patients, and other congenital defects may also be observed. The typical associated cytogenetic anomaly relies on an extra chromosome, derived from an inverted duplication of short arm and proximal long arm of chromosome 22, resulting in partial trisomy or tetrasomy of such regions (inv dup 22pter-22q11.2).Case presentationWe report on a full-term newborn, referred to us soon after birth. Physical examination showed facial dysmorphisms, including hypertelorism, down slanted palpebral fissures, and dysplastic ears with tragus hypoplasia and pre-auricular pit. Ophthalmologic evaluation and heart ultrasound identified left chorioretinal and iris coloboma andostium secundumtype atrial septal defect, respectively. Based on the suspicion of cat eye syndrome, a standard karyotype analysis was performed, and detected an extra small marker chromosome confirming the CES diagnosis. The chromosomal abnormality was then defined by array comparative genome hybridization (a-CGH, performed also in the parents), which identified the size of the rearrangement (3 Mb), and its de novo occurrence. Postnatally, our newborn presented with persistent hypoglycemia and cholestatic jaundice. Endocrine tests revealed congenital hypothyroidism, cortisol and growth hormone (GH) deficiencies, which were treated with replacement therapies (levotiroxine and hydrocortisone). Brain magnetic resonance imaging, later performed, showed aplasia of the anterior pituitary gland, agenesis of the stalk and ectopic neurohypophysis, confirming the congenital hypopituitarism diagnosis. She was discharged at 2 months of age, and included in a multidisciplinary follow-up. She currently is 7 months old and shows a severe global growth failure, and developmental delay. She started GH replacement treatment, and continues oral hydrocortisone, along with ursodeoxycholic acid and levothyroxine, allowing an adequate control of glycemic and thyroid profiles as well as of cholestasis.ConclusionsCES phenotypic spectrum is wide and highly variable. Our report highlights how among the possible associated endocrine disorders, congenital hypopituitarism may occur, leading to persistent hypoglycemia and cholestasis. These patients should be promptly assessed for complete hormonal evaluations, in addition to major malformations and midline anomalies. Early recognition of such defects is necessary to decrease fatal events, as well as short and long-term related adverse outcomes.
Scalable, ultra-fast, and low-memory construction of compacted de Bruijn graphs with Cuttlefish 2
Authors: Khan Jamshed , Kokot Marek , Deorowicz Sebastian , Patro Rob 
Published: September 2022

AbstractThe de Bruijn graph is a key data structure in modern computational genomics, and construction of its compacted variant resides upstream of many genomic analyses. As the quantity of genomic data grows rapidly, this often forms a computational bottleneck. We present Cuttlefish 2, significantly advancing the state-of-the-art for this problem. On a commodity server, it reduces the graph construction time for 661K bacterial genomes, of size 2.58Tbp, from 4.5 days to 17–23 h; and it constructs the graph for 1.52Tbp white spruce reads in approximately 10 h, while the closest competitor requires 54–58 h, using considerably more memory.
Performance of model-based vs. permutation tests in the HEALing (Helping to End Addiction Long-termSM) Communities Study, a covariate-constrained cluster randomized trial
Authors: Tang Xiaoyu , Heeren Timothy , Westgate Philip M. , Feaster Daniel J. , Fernandez Soledad A. , Vandergrift Nathan , Cheng Debbie M. 
Published: September 2022

Abstract
Background
The HEALing (Helping to End Addiction Long-termSM) Communities Study (HCS) is a multi-site parallel group cluster randomized wait-list comparison trial designed to evaluate the effect of the Communities That Heal (CTH) intervention compared to usual care on opioid overdose deaths. Covariate-constrained randomization (CCR) was applied to balance the community-level baseline covariates in the HCS. The purpose of this paper is to evaluate the performance of model-based tests and permutation tests in the HCS setting. We conducted a simulation study to evaluate type I error rates and power for model-based and permutation tests for the multi-site HCS as well as for a subgroup analysis of a single state (Massachusetts). We also investigated whether the maximum degree of imbalance in the CCR design has an impact on the performance of the tests.

Methods
The primary outcome, the number of opioid overdose deaths, is count data assessed at the community level that will be analyzed using a negative binomial regression model. We conducted a simulation study to evaluate the type I error rates and power for 3 tests: (1) Wald-type t-test with small-sample corrected empirical standard error estimates, (2) Wald-type z-test with model-based standard error estimates, and (3) permutation test with test statistics calculated by the difference in average residuals for the two groups.

Results
Our simulation results demonstrated that Wald-type t-tests with small-sample corrected empirical standard error estimates from the negative binomial regression model maintained proper type I error. Wald-type z-tests with model-based standard error estimates were anti-conservative. Permutation tests preserved type I error rates if the constrained space was not too small. For all tests, the power was high to detect the hypothesized 40% reduction in opioid overdose deaths for the intervention vs. comparison group both for the overall HCS and the subgroup analysis of Massachusetts (MA).

Conclusions
Based on the results of our simulation study, the Wald-type t-test with small-sample corrected empirical standard error estimates from a negative binomial regression model is a valid and appropriate approach for analyzing cluster-level count data from the HEALing Communities Study.

Trial registration
ClinicalTrials.gov http://www.clinicaltrials.gov; Identifier: NCT04111939
Empowering the willing: the feasibility of tele-mentored self-performed pleural ultrasound assessment for the surveillance of lung health
Authors: Kirkpatrick Andrew W. , McKee Jessica L. , Ball Chad G. , Ma Irene W. Y. , Melniker Lawrence A. 
Published: September 2022

Abstract
Background
SARS-CoV-2 infection, manifesting as COVID-19 pneumonia, constitutes a global pandemic that is disrupting health-care systems. Most patients who are infected are asymptomatic/pauci-symptomatic can safely self-isolate at home. However, even previously healthy individuals can deteriorate rapidly with life-threatening respiratory failure characterized by disproportionate hypoxemic failure compared to symptoms. Ultrasound findings have been proposed as an early indicator of progression to severe disease. Furthermore, ultrasound is a safe imaging modality that can be performed by novice users remotely guided by experts. We thus examined the feasibility of utilizing common household informatic-technologies to facilitate self-performed lung ultrasound.

Methods
A lung ultrasound expert remotely mentored and guided participants to image their own chests with a hand-held ultrasound transducer. The results were evaluated in real time by the mentor, and independently scored by three independent experts [planned a priori]. The primary outcomes were feasibility in obtaining good-quality interpretable images from each anatomic location recommended for COVID-19 diagnosis.

Results
Twenty-seven adults volunteered. All could be guided to obtain images of the pleura of the 8 anterior and lateral lung zones (216/216 attempts). These images were rated as interpretable by the 3 experts in 99.8% (647/648) of reviews. Fully imaging one’s posterior region was harder; only 108/162 (66%) of image acquisitions was possible. Of these, 99.3% of images were interpretable in blinded evaluations. However, 52/54 (96%) of participants could image their lower posterior lung bases, where COVID-19 is most common, with 99.3% rated as interpretable.

Conclusions
Ultrasound-novice adults at risk for COVID-19 deterioration can be successfully mentored using freely available software and low-cost ultrasound devices to provide meaningful lung ultrasound surveillance of themselves that could potentially stratify asymptomatic/paucisymptomatic patients with early risk factors for serious disease. Further studies examining practical logistics should be conducted.
Trial Registration: ID ISRCTN/77929274 on 07/03/2015.
Discovery and characterization of heterogeneous and multipotent fibroblast populations isolated from excised cleft lip tissue
Authors: Parisi Ludovica , Rihs Silvia , La Scala Giorgio C. , Schnyder Isabelle , Katsaros Christos , Degen Martin 
Published: September 2022

Abstract
Background
Regularly discarded lip tissue obtained from corrective surgeries to close the cleft lip represents an easily accessible and rich source for the isolation of primary fibroblasts. Primary fibroblasts have been described to show compelling similarities to mesenchymal stem cells (MSCs). Hence, cleft lip and palate (CLP) lip-derived fibroblasts could be thought as an intriguing cell source for personalized regenerative therapies in CLP-affected patients.

Methods
Initially, we thoroughly characterized the fibroblastic nature of the lip-derived mesenchymal outgrowths by molecular and functional assays. Next, we compared their phenotype and genotype to that of bone marrow-mesenchymal stem cells (BM-MSCs) and of human lung-derived fibroblasts WI38, by assessing their morphology, surface marker expression, trilineage differentiation potential, colony-forming (CFU) capacity, and immunomodulation property. Finally, to better decipher the heterogeneity of our CLP cultures, we performed a single cell clonal analysis and tested expanded clones for surface marker expression, as well as osteogenic and CFU potential.

Results
We identified intriguingly similar phenotypic and genotypic properties between CLP lip fibroblasts and BM-MSCs, which makes them distinct from WI38. Furthermore, our own data in combination with the complex anatomy of the lip tissue indicated heterogeneity in our CLP cultures. Using a clonal analysis, we discovered single cell-derived clones with increased levels of the MSC markers CD106 and CD146 and clones with variabilities in their commitment to differentiate into bone-forming cells and in their potential to form single cell-derived colonies. However, we were not able to gain clones possessing superior MSC-like capacities when compared to the heterogeneous parental CLP population. Additionally, all clones could still generate contractile forces and retained robust levels of the fibroblast specific marker FSP1, which was not detectable in BM-MSCs.

Conclusions
Our results suggest that we isolate heterogeneous populations of fibroblasts from discarded CLP lip tissue, which show a prominently multipotent character in their entirety avoiding the need for elaborate subpopulation selections in vitro. These findings suggest that CLP lip fibroblasts might be a novel potential cell source for personalized regenerative medicine of clinical benefit for CLP patients.
Therapeutic efficacy of human adipose mesenchymal stem cells in Crohn’s colon fibrosis is improved by IFN-γ and kynurenic acid priming through indoleamine 2,3-dioxygenase-1 signaling
Authors: Ye Yixin , Zhang Xiaomei , Su Dongsheng , Ren Yushuang , Cheng Fuyi , Yao Yunqi , Shi Gang , Ji Yanhong , Chen Shuang , Shi Pengyi , Dai Lei , Su Xiaolan , Deng Hongxin 
Published: September 2022

Abstract
Background
Inflammatory bowel diseases (IBD) are chronic relapsing–remitting inflammatory diseases of the gastrointestinal tract that are typically categorized into two subtypes: Crohn's disease (CD) and ulcerative colitis (UC). Although MSCs therapy has achieved encouraging outcomes in IBD therapy, objective responses are limited in colon fibrosis stenosis owing to the complicated microenvironment of CD and MSCs heterogeneity of quality. Here, we chose IFN-γ and kynurenic acid (KYNA) to overcome the low response and heterogeneity of human adipose-derived MSCs (hADSCs) to treat IBD and expand the therapeutic effects based on the excellent ability of IFN-γ and KYNA to promote indoleamine 2,3-dioxygenase-1 (IDO-1) signaling, providing a potential protocol to treat IBD and fibrosis disease.

Methods
hADSCs were isolated, cultured, and identified from human abdominal adipose tissue. The CD pathology-like acute colitis and chronic colon fibrosis rat model was induced by 2,4,6-trinitrobenzen sulfonic acid (TNBS). hADSCs were pretreated in vitro with IFN-γ and KYNA and then were transplanted intravenously at day 1 and 3 of TNBS administration in colitis along with at day 1, 15, and 29 of TNBS administration in chronic colonic fibrosis. Therapeutic efficacy was evaluated by body weights, disease activity index, pathological staining, real-time PCR, Western blot, and flow cytometry. For knockout of IDO-1, hADSCs were transfected with IDO-1-targeting small gRNA carried on a CRISPR-Cas9-lentivirus vector.

Results
hADSCs treated with IFN-γ and KYNA significantly upregulated the expression and secretion of IDO-1, which has effectively ameliorated CD pathology-like colitis injury and fibrosis. Notably, the ability of hADSCs with IDO-1 knockout to treat colitis was significantly impaired and diminished the protective effects of the primed hADSCs with IFN-γ and KYNA.

Conclusion
Inflammatory cytokines IFN-γ- and KYNA-treated hADSCs more effectively alleviate TNBS-induced colitis and colonic fibrosis through an IDO-1-dependent manner. Primed hADSCs are a promising new strategy to improve the therapeutic efficacy of MSCs and worth further research.
YAP and TAZ play a crucial role in human erythrocyte maturation and enucleation
Authors: Damkham Nattaya , Lorthongpanich Chanchao , Klaihmon Phatchanat , Lueangamornnara Usaneeporn , Kheolamai Pakpoom , Trakarnsanga Kongtana , Issaragrisil Surapol 
Published: September 2022

Abstract
Background
Yes-associated protein (YAP) and WW domain-containing transcription regulator protein 1 (WWTR1, also known as TAZ) are two key transcription co-activators of the Hippo pathway. Both were originally characterized as organ size and cell proliferation regulators. Later studies demonstrated that the Hippo pathway may play a role in Drosophila and mammal hematopoiesis. However, the role of the Hippo pathway in human erythropoiesis has not yet been fully elucidated.

Methods
The role of YAP and TAZ was studied in human erythropoiesis and hematopoietic stem cell (HSC) lineage determination by using mobilized peripheral blood (PB) and cord blood (CB)-derived HSC as a model. HSCs were isolated and cultured in an erythroid differentiation medium for erythroid differentiation and culture in methylcellulose assay for HSC lineage determination study.

Results
YAP and TAZ were barely detectable in human HSCs, but became highly expressed in pro-erythroblasts and erythroblasts. Depletion or knockdown of YAP and/or TAZ did not affect the ability of HSC lineage specification to erythroid lineage in either methylcellulose assay or liquid culture. However, depletion of YAP and TAZ did impair erythroblast terminal differentiation to erythrocytes and their enucleation. Moreover, ectopic expression of YAP and TAZ in pro-erythroblasts did not exert an apparent effect on erythroid differentiation, expansion, or morphology.

Conclusions
This study demonstrated that YAP/TAZ plays important role in erythroid maturation and enucleation but is dispensable for lineage determination of human HSCs.