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A multicenter prospective study on the management of hepatoblastoma in children:a report from the Chinese children's Cancer Group

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World Journal of Pediatrics 2024年第6期20卷 590-601页

作者： Meng-Jie Tang Xiao-Li Ma Xiang-Ling He Wei-Hua Pan Xiao-Hong Zhang sha-Yi Jiang Ju Gao Fu Lie Wei Yao song Gu Wei-Ling Zhang Qiang Zhao shi-Hao Huang Yong-Jun Fang Wei Liu Hui-Zhong Niu Chun-Mei Wang Li-Rong sun Hui Gao Yun-Peng Dai shun-Gen Huang Zhi-Yong Zhong Xi-Ge Wang Zhong-Rong Li Liang-Chun Yang Ye-Ming Wu Huan-Min Wang Xin sun Xiao-Jun Yuan Department of Pediatric Hematology/Oncology Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of MedicineNo.1665Kongjiang RoadYangpu DistrictShanghai 200092China Medical Oncology Department Pediatric Oncology CenterBeijing Children's HospitalCapital Medical UniversityNational Center for Children's HealthBeijing 100045China Department of Hematology/Oncology Children's Medical CenterHunan Provincial People's HospitalThe First Affiliated Hospital of Hunan Normal UniversityChangsha 41005China Department of Pediatric surgery Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of MedicineShanghai 200092China Department of Hematology/Oncology Guangzhou Women and Children's Medical CenterNational Children's Medical Center for South Central RegionGuangzhou 510623China Department of Hematology Shanghai Children's HospitalShanghai Jiao Tong UniversityShanghai 200062China Department of Pediatrics West China Second University HospitalSichuan UniversityChengdu 610041China Department of Hematology/Oncology Qilu Children's Hospital of Shandong UniversityJinan 250022China Department of Pediatric surgery Children's Hospital of Fudan UniversityShanghai 201102China Department of General surgery Shanghai Children's Medical Center(National Children's Medical Center-Shanghai)Shanghai Jiao Tong University School of MedicineShanghai 200127China Department of Pediatrics Beijing Tongren HospitalCapital Medical UniversityBeijing 100730China Department of Pediatric Oncology Tianjin Medical University Cancer Institute and HospitalTianjin 300060China Department of Hematology/Oncology Children's Hospital of Nanjing Medical UniversityNanjing 210008China Department of Hematology/Oncology Henan Children's HospitalZhengzhou 450018China Department of Pediatric General surgery Hebei Children's Hospital of Hebei Medical UniversityShijiazhuang City 050031China children's Hospital the First Affliated Hospital of Zhengzhou UniversityZhengzhou 450052China Department of Paediatric

Background This study aimed to identify survival risk factors in Chinese children with hepatoblastoma(HB)and assess the effectiveness of the new treatment protocol proposed by the Chinese children's Cancer Group(CCCG)in *** A multicenter,prospective study that included 399 patients with HB from January 2015 to June 2020 was *** demographics,treatment protocols,and other related information were *** regression models and Kaplan-Meier curve methods were *** The 4-year event-free survival(EFs)and overall survival(Os)were 76.9 and 93.5%,*** 4-year EFs rates for the very-low-risk,low-risk,intermediate-risk,and high-risk groups were 100%,91.6%,81.7%,and 51.0%,*** 4-year 0s was 100%,97.3%,94.4%,and 86.8%,*** regression analysis found that age,tumor rupture(R+),and extrahepatic tumor extension(E+)were independent prognostic factors.A total of 299 patients had complete remission,and 19 *** with declining alpha-fetoprotein(AFP)>75%after the first two cycles of neoadjuvant chemotherapy had a better EFs and Os than those≤75%.Conclusions The survival outcome of HB children has dramatically improved since the implementation of CCCG-HB-2016 ***≥8 years,R+,and E+were independent risk factors for *** with a declining AFP>75%after the first two cycles of neoadjuvant chemotherapy had better EFs and Os.

关键词： Alpha-fetoprotein Hepatoblastoma Multicenter Prospective study survival

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Efficacy of cognitive-behavioral therapy combined with habit reversal training on anxiety disorders in children with Tourette’s syndrome

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World Journal of Psychiatry 2024年第7期14卷 999-1008页

作者： Yan-Zhen Wang Xi Zhang Xin-Min Han Department of Rehabilitation Children’s Hospital of ShanxiTaiyuan 030012Shanxi ProvinceChina Department of Pediatrics The Affiliated Hospital of Nanjing University of Chinese MedicineNanjing 210029Jiangsu ProvinceChina

BACKGROUND Cognitive-behavioral therapy(CBT)and habit reversal training(HRT)have shown application potential in addressing tic symptoms and comorbid psychiatric *** their theoretical potential,empirical evidence on their combined efficacy remains *** To evaluate the efficacy of CBT combined with HRT on anxiety disorders in children with Tourette’s syndrome(Ts).METHODs Clinical data of children with Ts admitted to our hospital from January 2022 to June 2023 were collected,and the patients were grouped into the conventional therapy(control)group and the CBT combined with HRT *** charac-teristics,anxiety scores,tic severity scores,treatment adherence,and parental satisfaction were *** analysis was performed using t-tests,chi-square tests,and correlation *** A total of 136 patients,including 65 patients in the control group and 71 patients in the CBT combined with HRT group,were *** CBT combined with HRT group showed remarkable improvements compared with the control ***-intervention assessment revealed a decrease in anxiety scores from 63.52±1.81 to 40.53±1.64(t=2.022,P=0.045),and the Yale Global Tic severity scale total score decreased from 22.14±5.67 to 16.28±4.91(t=2.288,P=0.024).Treatment adherence was significantly higher in the CBT combined with HRT group(85.47±7.62%)compared with the control group(82.32±6.54%;t=2.596,P=0.010).Parental satisfaction scores were also higher in the CBT combined with HRT group(8.69±1.77)compared with the control group(7.87±1.92;t=2.592,P=0.011).CONCLUsION This study demonstrates that CBT combined with HRT significantly reduces anxiety symptoms and tic severity in children with Ts,with higher treatment adherence and parental *** findings support the potential application of this comprehensive therapeutic approach for Ts treatment.

关键词： Tourette's syndrome Cognitive-behavioral therapy Habit reversal training Efficacy evaluation Anxiety disorders

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Development and validation of a novel non-invasive test for diagnosing nonalcoholic fatty liver disease in Chinese children

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World Journal of Pediatrics 2024年第4期20卷 413-421页

作者： Zhe-Wen Qin Qian-Nan Ren Hong-Xi Zhang Ya-Ru Liu Ke Huang Wei Wu Guan-Ping Dong Yan Ni Jun-Fen Fu Division of Endocrinology National Clinical Research Center for Child HealthNational Children’s Regional Medical CenterThe Children’s HospitalDepartment of EndocrinologyNational Clinical Research Center for Child HealthNational Children’s Regional Medical CenterThe Children’s HospitalZhejiang University School of MedicineHangzhou310052China

With the exploding prevalence of obesity,many children are at risk of developing nonalcoholic fatty liver *** anthropometric and laboratory parameters,our study aimed to develop a model to quantitatively evaluate liver fat content(LFC)in children with *** A well-characterized cohort of 181 children between 5 and 16 years of age were recruited to the study in the Endocrinology Department as the derivation *** external validation cohort comprised 77 *** assessment of liver fat content was performed using proton magnetic resonance *** and laboratory metrics were measured in all subjects.B-ultrasound examination was carried out in the external validation *** Kruskal-Wallis test,spearman bivariate correlation analyses,univariable linear regressions and multivariable linear regression were used to build the optimal predictive *** The model was based on indicators including alanine aminotransferase,homeostasis model assessment of insulin resistance,triglycerides,waist circumference and Tanner *** adjusted R^(2)of the model was 0.589,which presented high sensitivity and specificity both in internal[sensitivity of 0.824,specificity of 0.900,area under curve(AUC)of 0.900 with a 95%confidence interval:0.783–1.000]and external validation(sensitivity of 0.918 and specificity of 0.821,AUC of 0.901 with a 95%confidence interval:0.818–0.984).Conclusions Our model based on five clinical indicators was simple,non-invasive,and inexpensive;it had high sensitivity and specificity in predicting LFC in ***,it may be useful for identifying children with obesity who are at risk for developing nonalcoholic fatty liver disease.

关键词： Biochemical markers children Magnetic resonance spectroscopy Nonalcoholic fatty liver disease Pediatric obesity

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Function and dysfunction of GEMIN5:understanding a novel neurodevelopmental disorder

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Neural Regeneration Research 2024年第11期19卷 2377-2386页

作者： Charles H.Nelson Udai B.Pandey Department of Pediatrics Division of Child NeurologyChildren’s Hospital of PittsburghUniversity of Pittsburgh Medical CenterPittsburghPAUSA children’s Neuroscience Institute Children’s Hospital of PittsburghUniversity of Pittsburgh Medical CenterPittsburghPAUSA

The recent identification of a neurodevelopmental disorder with cerebellar atrophy and motor dysfunction(NEDCAM)has resulted in an increased interest in GEMIN5,a multifunction RNA-binding *** the largest member of the survival motor neuron complex,GEMIN5 plays a key role in the biogenesis of small nuclear ribonucleoproteins while also exhibiting translational regulatory functions as an independent *** many questions remain regarding both the pathogenesis and pathophysiology of this new disorder,considerable progress has been made in the brief time since its *** this review,we examine GEMIN5 within the context of NEDCAM,focusing on the structure,function,and expression of the protein specifically in regard to the disorder ***,we explore the current animal models of NEDCAM,as well as potential molecular pathways for treatment and future directions of *** review provides a comprehensive overview of recent advances in our understanding of this unique member of the survival motor neuron complex.

关键词： cerebellar atrophy GEMIN5 neurodevelopmental disorder neurological disease sMN complex

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Associations of Perchlorate,Nitrate,and Thiocyanate with sex Hormones among children:A Nationally Representative Cross-sectional study in U.s.

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Biomedical and Environmental sciences 2024年第6期37卷 666-671页

作者： Yiwen Wang Guodong Ding Peipei Hu Yongjun Zhang Department of Pediatrics Xinhua HospitalShanghai Jiao Tong University School of MedicineShanghai 200092China MOE-shanghai Key Laboratory of children’s Environmental Health Xinhua HospitalShanghai Jiao Tong University School of MedicineShanghai 200092China

Nitrates,thiocyanates,and perchlorates are ubiquitous in the environment and can be introduced to the public through various dietary and environmental ***,thiocyanates,and perchlorates mainly affect health via the sam... 详细信息

关键词： mechanism sectional perchlorate

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somatic GNA11/GNAQ variants in a cohort of Chinese children with phakomatosis pigmentovascularis

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Pediatric Investigation 2024年第2期8卷 117-125页

作者： Bin Zhang Rui He Riga Wu Zhou Yang Man Hu Nan Zhang Wu Guo Zigang Xu Lin Ma Department of Dermatology Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijingChina Department of Dermatology Zhengzhou UniversityAffiliated Children’s HospitalHenan Children’s HospitalZhengzhou Children’s HospitalZhengzhouChina Department of Dermatology Affiliated Hospital of Inner Mongolia Medical UniversityHohhotChina Department of Ophthalmology Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijingChina Department of Pathology Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijingChina

Importance:Postzygotic mutations in the GNAQ/GNA11 genes,which encode the G-protein nucleotide binding protein alpha subunits,have been identified in patients with phakomatosis pigmentovascularis(PPV).However,little is known about the Chinese ***:To identify pathogenic mutations in pediatric patients with PPV within the Chinese ***:We performed whole-exome sequencing(WEs)using skin lesion tissues from pediatric patients diagnosed with ***,ultradeep-targeted sequencing was conducted to validate the somatic mutations.A genotype-phenotype correlation was analyzed by integrating data from previous reports with the findings of the present ***:Thirteen patients were enrolled,all diagnosed with the cesioflammea type of PPV,except for one patient with an unclassifiable *** identified somatic GNA11 c.547C>T(p.R183C)variant in seven patients and GNAQ c.548G>A(p.R183Q)in four patients,with low allelic fractions ranging from 2.1%to 8.6%through ultradeep ***,a GNAQ c.548G>A(p.R183Q)variant was detected through targeted sequencing in one of two patients who did not exhibit detectable variants via *** genotype-phenotype correlation analysis,involving 15 patients with a GNA11 variant and 10 with a GNAQ variant,revealed that facial capillary malformation(87%vs.50%,P=0.075)and ocular melanocytosis(80%vs.40%,P=0.087)appeared to be more frequent in patients with GNA11 mutation compared to those with GNAQ *** four patients diagnosed with cesiomarmorata type or overlapping cesioflammea and cesiomarmorata type PPV carried the GNA11 ***:Our study demonstrated that the majority of PPV patients in the Chinese population carried a postzygotic variant of GNAQ/GNA11,thus further confirming the pathogenic role of GNAQ/GNA11 mosaicism in the development of PPV cesioflammea type.

关键词： Phakomatosis pigmentovascularis GNA11 GNAQ somatic variant

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Current status of cognition and skin care behavior in adolescent patients with acne:A survey in China

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Chinese Medical Journal 2024年第4期137卷 476-477页

作者： Jing Tian Hong shu Qiufang Qian Zhong shen Chunyu Zhao Li song Ping Li Xiuping Han Hua Qian Jinping Chen Hua Wang Lin Ma Yuan Liang Department of Dermatology Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijing 100045China Department of Dermatology Kunming Children’s HospitalKunmingYunnan 650034China Department of Dermatology Children’s Hospital of ShanghaiChildren’s Hospital Affiliated to Shanghai Jiao Tong University School of MedicineShanghai 200127China Loreal China Co. LtdShanghai 200040China Department of Dermatology Henan Children’s HospitalChildren’s Hospital Affiliated to Zhengzhou UniversityZhengzhou Hospital Beijing Children’s HospitalZhengzhou Children’s HospitalZhengzhouHenan 450052China Department of Dermatology Shenzhen Children’s HospitalShenzhenGuangdong 518026China Department of Dermatology Shengjing Hospital of China Medical UniversityShenyangLiaoning 110004China Department of Dermatology Children’s Hospital of Soochow UniversitySuzhouJiangsu 215025China Department of Dermatology Guangzhou Women and Children's Medical CenterGuangzhouGuangdong 510623China Department of Dermatology Children’s Hospital of Chongqing Medical UniversityChongqing 400014China

To the Editor:Acne vulgaris(AV)is a common inflammatory skin ***-based epidemiological studies have reported associations between dietary habits,lifestyle factors,family history,and the occurrence and development of acne.^([1–4])This study investigated the skincare behavior and cognition of patients with AV regarding predisposing factors using a designed electronic questionnaire.

关键词： patients dietary behavior

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spotlight on eltrombopag concentration in pediatric immune thrombocytopenia:A single-center observational study in China

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Pediatric Investigation 2024年第1期8卷 44-52页

作者： shuyue Dong Zhifa Wang Nan Wang Jingyao Ma Jinxi Meng Yixin sun Xiaoling Cheng Runhui Wu Hematology Oncology Center Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijingChina Department of Pharmacy Beijing Children’s HospitalCapital Medical UniversityNational Center for Children’s HealthBeijingChina

Importance:Eltrombopag has been recommended for pediatric immune thrombocytopenia(ITP).Response and adverse drug reactions(ADRs)varied widely between individuals,even at the same dose of *** appropriate eltrombopag concentration in ITP has not been ***:This study aims to explore the appropriate eltrombopag concentration in pediatric ***:This was a single-center,prospective cohort *** diagnosed with refractory persistent/chronic ITP and platelet countst 2 *** was detected by high-performance liquid chromatography-mass spectrometry at least 2 weeks after *** clinical characteristics-concentration,concentration-response,and concentration-ADRs were ***:A total of 30 patients were enrolled,comprising 13 males and 17 females,with a median age of 72(45-94)*** median dose and concentration were 1.39(1.09-1.56)mg/kg and 2.70(2.25-4.13)mg/L,*** the enrolled patients,14 responded to treatment,whereas 16 did ***,five experienced adverse drug *** linear correlation was observed between eltrombopag concentration and clinical *** concentration was lower in the response group than in the nonresponse group,but there was no significant difference(t=0.755,P=0.457).Patients who experienced ADRs had a higher concentration than those without ADRs(t=2.538,P=0.017).The area under the receiver operating characteristic curve of ADRs was 0.78(95%confidence interval:0.56-1.00).Youden’s index identified the cutoff point as 4.33 mg/L,with a sensitivity of 88%and a specificity of 60%.Logistic regression analysis demonstrated that a higher platelet count before eltrombopag predicted a favorable ***:Eltrombopag proves efficacious and well-tolerated for treating pediatric ***,prolonged and high-dose administration may increase the likelihood of ***,examining

关键词： Concentration Efficacy Eltrombopag Immune thrombocytopenia Pedi-atrics safety

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X-box binding protein 1 caused an imbalance in pyroptosis and mitophagy in immature rats with di-(2-ethylhexyl)phthalate-induced testis toxicity

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Genes & Diseases 2024年第2期11卷 935-951页

作者： Yifan Hong Xiazhu Zhou Qi Li Jing Chen Yuexin Wei Chunlan Long Lianju shen Xiangqin Zheng Dinggang Li Xia Wang Chenjun Yu shengde Wu Guanghui Wei Department of Urology Children's Hospital of Chongqing Medical UniversityChongqing 400014China Pediatric Research Institute Children's Hospital of Chongqing Medical UniversityChongqing 400014China Chongqing Key Laboratory of children Urogenital Development and Tissue Engineering Children's Hospital of Chongqing Medical UniversityChongqing 400014China Chongqing Key Laboratory of Pediatrics Children's Hospital of Chongqing Medical UniversityChongqing 400014China Ministry of Education Key Laboratory of Child Development and Disorders Children's Hospital of Chongqing Medical UniversityChongqing 400014China National Clinical Research Center for Child Health and Disorders Children's Hospital of Chongqing Medical UniversityChongqing 400014China China International science and Technology Cooperation Base of Child Development and Critical Disorders Children's Hospital of Chongqing Medical UniversityChongqing 400014China

As a widely used plasticizer,di-(2-ethylhexyl)phthalate(DEHP)is known to induce significant testicular ***,the potential mechanism and effects of pubertal exposure to DEHP on testis development remain *** vivo,postnatal day(PND)21 male rats were gavaged with 0,250,and 500 mg/kg DEHP for ten *** to the seminiferous epithelium and disturbed spermatogenesis were observed after DEHP ***,oxidative stress-induced injury and pyroptosis were *** endoplasmic reticulum(ER)stress and mitophagy were involved in this *** phthalate(MEHP)was used as the biometabolite of DEHP in *** GC-1 and GC-2 cell lines were exposed to 0,100μM,200μM,and 400μM MEHP for 24 *** oxygen species(ROs)generation,oxidative stress damage,ER stress,mitophagy,and pyroptosis were significantly increased after MEHP *** ultrastructure of the ER and mitochondria was destroyed.X-box binding protein 1(XBP1)was observed to be activated and translocated into the *** generation was inhibited by *** levels of antioxidative stress,ER stress,mitophagy,and pyroptosis were decreased as *** the administration of the ER stress inhibitor 4-phenyl-butyric acid,both mitophagy and pyroptosis were ***-induced XBP1 down-regulation decreased the levels of mitophagy and *** equilibrium between pyroptosis and mitophagy was disturbed by XBP1 *** summary,our findings confirmed that DEHP induced a ROs-mediated imbalance in pyroptosis and mitophagy in immature rat testes via ***,XBP1 might be the key target in DEHP-related testis dysfunction.

关键词： DEHP Mitophagy Pyroptosis Testicular development XBP1

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Proteomic profiling of cerebrospinal fluid in pediatric myelin oligodendrocyte glycoprotein antibody-associated disease

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World Journal of Pediatrics 2024年第3期20卷 259-271页

作者： Yi-Long Wang Meng-Ying Zhu Zhe-Feng Yuan Xiao-Yan Ren Xiao-Tong Guo Yi Hua Lu Xu Cong-Ying Zhao Li-Hua Jiang Xin Zhang Guo-Xia sheng Pei-Fang Jiang Zheng-Yan Zhao Feng Gao Department of Neurology Children's HospitalZhejiang University School of MedicineHangzhou 310052China children's Hospital Zhejiang University School of MedicineHangzhou 310052China children's Hospital National Clinical Research Center for Child HealthZhejiang University School of MedicineHangzhou 310052China

Background Myelin oligodendrocyte glycoprotein(MOG)antibody-associated disease(MOGAD)is an autoimmune demy-elinating disorder of the central nervous *** Extracted proteins from 34 cerebrospinal fluid(CsF)samples[patients with MOGAD(MOG group,n=12);healthy controls(HC group,n=12);patients with MOG seronegative and metagenomics next-generation sequencing-negative inflammatory neurological diseases(IND group,n=10)]were processed and subjected to label-free quantitative *** partial least squares-discriminant analysis(PLs-DA)and orthogonal PLs-DA(O-PLs-DA)models were also performed based on proteomics *** analysis of differentially expressed proteins(DEPs)was performed using Gene Ontology,InterPro,and Kyoto Encyclopedia Genes and *** enzyme-linked immunosorbent assay was used to determine the complement levels in serum from patients with *** Four hundred and twenty-nine DEPs(149 upregulated and 280 downregulated proteins)were identified in the MOG group compared to the HC group according to the P value and fold change(FC).Using the O-PLs-DA model,872 differentially abundant proteins were identified with variable importance projection(VIP)scores>*** proteins(gamma-glutamyl hydrolase,cathepsin F,interalpha-trypsin inhibitor heavy chain 5,latent transforming growth factor beta-binding protein 4 and leukocyte-associated immunoglobulin-like receptor 1)overlapping between the top 30 DEPs with top-ranked P value and FC and top 30 proteins in PLs-DA VIP lists were *** analysis revealed that the dysregulated proteins in the MOG group were primarily involved in complement and coagulation cascades,cell adhesion,axon guidance,and glycosphingolipid biosynthesis compared to the HC *** The proteomic alterations in CsF samples from children with MOGAD identified in the current study might provide opportunities for developing novel biomarker candidates.

关键词： Acute disseminated encephalomyelitis Cerebrospinal fluid Complement cascades Myelin oligodendrocyte glycoprotein-associated disease Proteomics

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