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Abdominal cocoon in children: A case report and review of literature

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World Journal of Gastroenterology 2021年第37期27卷 6332-6344页

作者： Daniel Keese Andrea Schmedding Kerstin Saalabian Georgy Lakshin Henning Fiegel Udo Rolle Goethe-University Frankfurt Department of Paediatric Surgery and Paediatric UrologyUniversity HospitalFrankfurt 60590Germany

BACKGROUND Abdominal cocoon or“encapsulating peritoneal sclerosis”(EPS)is an uncommon and rare cause of intestinal *** a few cases have been reported in paediatric ***,EPS is described as the primary form in young adolescent girls from tropical and subtropical countries because of viral peritonitis due to retrograde menstruation or a history of peritoneal *** patients are asymptomatic or present with abdominal pain,which is likely to occur secondary to subacute bowel *** at imaging,such as ultrasound,computed tomography,and magnetic resonance imaging,are often *** diagnosed,EPS is characterized by total or partial encasement of the bowel within a thick fibrocollagenous membrane that envelopes the small intestine in the form of a cocoon because of chronic intraabdominal fibroinflammatory *** membrane forms a fibrous tissue sheet that covers,fixes,and finely constricts the gut,compromising its *** SUMMARY We present a case of EPS in a 12-year-old boy 8 wk after primary surgery for resection of symptomatic jejunal *** was no history of peritoneal dialysis or drug *** In this report,we sought to highlight the diagnostic,surgical,and histopathological characteristics and review the current literature on EPS in paediatric patients.

关键词： Abdominal cocoon Peritoneal encapsulation Encapsulating peritoneal sclerosis Intestinal obstruction Children Case report

Three novel homozygous ITGB2 mutations among two patients with leukocyte adhesion defect type-1:Two case reports

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World Journal of Clinical Pediatrics 2022年第5期11卷 429-436页

作者： Yiwa Suksawat Punchama Pacharn Nunthana Siripipattanamongkol Boonchai Boonyawat Division of allergy and immunology Department of PediatricsPhramongkutklao Hospital and Phramongkutklao College of MedicineBangkok 10400Thailand Division of allergy and immunology Department of PediatricsSiriraj HospitalMahidol UniversityBangkok 10700Thailand Division of allergy and immunology Department of PediatricsChiangrai Prachanukroh Regional HospitalChiangrai 57000Thailand Division of Medical Genetics Department of PediatricsPhramongkutklao Hospital and Phramongkutklao College of MedicineBangkok 10400Thailand

BACKGROUND A leukocyte adhesion defect(LAD) is a rare primary immunodeficiency disorder. LAD type 1(LAD-1) is the most common, which is caused by ITGB2 mutation resulting in dysfunction of β2 integrin, which impairs leukocyte adherence to the *** SUMMARY The first two cases of LAD-1 in Thailand presented with recurrent omphalitis, soft tissue infection, marked leukocytosis, and neutrophilia. One patient experienced delayed umbilical cord separation. Mutation analysis was performed by direct DNA sequencing of the ITGB2 gene. The results revealed two novel homozygous missense mutations, c.920C>T(***307Pro) in exon 8 and c.758G>A(***-253His) in exon 7, and one novel homozygous nonsense mutation, c.262C>T(***88Ter) in exon 4, in the genomic DNA of the first and second patients, respectively. Heterozygous mutations were identified in the parents of both patients, suggesting a carrier status. The patients were administered intravenous antibiotics for infections with good clinical responses. Hematopoietic stem cell transplantation could not be performed due to the unavailability of matched donors. However, a significant decline in infections was observed after antibiotic prophylaxis. Several follow-up visits were conducted for both patients. They are currently 6 years *** Molecular analysis is essential for definitive diagnosis, early treatment implementation, and prevention of LAD-1 in future pregnancy.

关键词： Leukocyte adhesion defect ITGB2 Omphalitis Bacterial soft tissue infection Molecular investigation Case report

Serum ficolin-2 concentrations are significantly changed in patients with hepatitis B virus infection and liver diseases

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Virologica Sinica 2015年第4期30卷 249-260页

作者： Tielong Chen Yilan Hu Quanquan Ding Jing Yu Fubing Wang Fengling Luo Xiao-Lian Zhang State Key Laboratory of Virology and department of immunology and Hubei Province Key Laboratory of allergy and immunology Wuhan University School of Medicine Zhongnan Hospital of Wuhan University department of immunology Wuhan University of Science and Technology School of Medicine Hubei Cancer Hospital

Human ficolin-2 is an important lectin complement pathway activator that is secreted from liver cells and has been implicated as an anti-infection innate immune molecule. However, the role of ficolin-2 protein and its dynamic changes over the course of and in the prognosis of chronic hepatitis B(CHB) and hepatocellular carcinoma(HCC) remain unclear. In this study, we analyzed ficolin-2 protein expression in a cohort of individuals with CHB infection, HCC and cirrhosis. A sandwich enzyme-linked immunosorbent assay(ELISA) method was used to measure serum ficolin-2 concentrations. Ficolin-2 expression in liver tissues was detected by immunohistochemical staining. Serum ficolin-2 concentrations in CHB patients were significantly higher than in healthy controls and HBV carriers. After 48 weeks of routine amelioration liver function treatment, serum ficolin-2 concentrations decreased and were positively correlated with favorable alanine aminotransferase(ALT), HBV DNA and HBe Ag-seroconversion outcomes. Interestingly, we observed much lower expression of serum and intrahepatic ficolin-2 in HCC and cirrhosis compared with healthy controls. Our findings suggest that serum and intrahepatic ficolin-2 levels may be considered one of the indicators for the response of chronic HBV infection, HCC and cirrhosis.

关键词： ficolin-2 chronic hepatitis B(CHB) hepatocellular

Effect of early atopic sensitization in children aged 0–2 years on the development of asthma symptoms at 9–11 years of age

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World Journal of Pediatrics 2022年第11期18卷 753-760页

作者： Himmet Haluk Akar Ebru Nadir Burçin Beken Yakup Yeşil Division of Pediatric immunology and allergy University of Istanbul Health SciencesKanuni Sultan Suleyman Training and Research HospitalIstanbulTurkey Division of Pediatrics University of Istanbul Health SciencesKanuni Sultan Suleyman Training and Research HospitalIstanbulTurkey department of Pediatric immunology and allergy School of MedicineAcibadem Mehmet Ali Aydinlar UniversityIstanbulTurkey

Background Personal genetic predisposition and early life environmental factors are important for the development of childhood *** aimed to search whether egg,milk and mite sensitizations at 0–2 years old are risk factors for asthma symptoms at 9–11 years *** A total of 210 wheezer children who had specific immunoglobulin(Ig)E in 2010–2012 were included in the study(followed by pediatric allergy).Patients were divided into non-atopic(group 1,n=157)and atopic patients[groups 2–7,n=53(5 patients were in both group 4 and group 5)]based on *** the International Study of Asthma and allergy in Childhood questionnaire,current wheeze(CW,2nd question),exercise wheezing(EW,7th question),and dry cough(DC,8th question)were ***,parental allergies,eczema at 0–2 years,current eosinophil percentage and total IgE were *** Eczema was observed as an important risk factor[CW:odds ratio(OR)=2.83,95%confidence interval(CI)=1.54–5.23,P≤0.001;EW:OR=2.71,95%CI=1.33–5.54,P=0.006;DC:OR=3.03,95%CI=1.47–6.25,P=0.003],whereas having no atopic sensitization at 0–2-year-old(group 1)was found as a significant protective factor for asthma at 9–11 years old(CW:OR=0.32,95%CI=0.15–0.70,P=0.004;EW:OR=0.21,95%CI 0.10–0.44,P≤0.001;DC:OR=0.25,95%CI=0.10–0.59,P=0.002).Conclusion Early personal eczema is a significant risk factor for the development of asthma symptoms at 9–11 years old,whereas not having an allergic sensitization at 0–2 years old(group 1)is an important protective factor.

关键词： Asthma Children Risk factors

Longitudinal Evaluation of Right Ventricle Function after Right Ventricle-Pulmonary Artery Shunt ***-Taussig Shunt

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Congenital Heart Disease 2021年第1期16卷 27-37页

作者： Nunzia Borrelli Jolanda Sabatino Martina Avesani Josefa Paredes Manjit Josen Alain Fraisse Paolo Guccione Guido Michielon Giovanni Di Salvo paediatric Cardiology department Royal Brompton HospitalLondonSW36NPUK paediatric Cardiology department Bambino GesùPaediatric HospitalRome00165Italy Cardiology department “Magna Graecia”UniversityCatanzaro88100Italy paediatric Cardiology department University of PaduaPadua35128Italy

Background:Still little is known about the impact on right ventricle function of the 2 main approaches to Norwood palliation in Hypoplastic left heart syndrome,the right ventricle-pulmonary artery shunt(RVPAS)and the modified Blalock-Taussig shunt(mBTS).Methods:The cohort included 27 patients with Hypoplastic left heart syndrome(10 in the mBTS group,17 in the RVPAS group).Longitudinal strain,tricuspid annulus peak systolic excursion and fractional area change were evaluated before Norwood and in four different breakpoints in the steady-state after Norwood procedure(30-days,90-days,140-days and 200-days after Norwood).Results:No significant differences were found in all standard echocardiographic functional parameters between the two groups at any ***,when we compared right ventricle function before and after Norwood procedure,longitudinal strain significantly improved in mBTS group compared to pre-surgical assessment(after 90-days:mBTS+27,35%±43,47 ***-8,20%±25,25,p=0,03;after 200-days:mBTS+10.19%±36.58 ***-21.64%±30.43,p=0.04).Conclusion:The mBTS group,which did not receive any ventriculotomy,showed a significant increase in right ventricle longitudinal strain during *** data support the use of longitudinal strain in Hypoplastic left heart syndrome evaluation and may suggest potential value in terms of cardiac mechanics in using mBTS,preserving the right ventricle integrity.

关键词： Hypoplastic left heart syndrome speckle tracking echocardiography congenital heart disease

Psychiatric comorbidities in children and adolescents with chronic urticaria

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World Journal of Pediatrics 2023年第4期19卷 315-322页

作者： George N.Konstantinou Gerasimos N.Konstantinou department of allergy and Clinical immunology 424 General Military Training HospitalThessaloníkiGreece department of Psychiatry University of TorontoTorontoONCanada Center for Addiction and Mental Health(CAMH) TorontoONCanada

Background Chronic urticaria(CU)has been shown to impact patients'quality of life negatively and may coexist with psychiatric *** systematically reviewed the published evidence of comorbid psychiatric disorders in children and adolescents with *** A systematic review of studies published until February 2022 in PubMed,Google Scholar,and Scopus was *** a priori set of inclusion criteria was predefined for the studies to be included:(1)clear distinction between urticaria and other allergies;(2)precise distinction between acute and CU;(3)participants younger than 18 years old,exclusively;(4)use of appropriate standardized questionnaires,psychometric tools,and standard diagnostic nomenclature for the mental health and behavioral disorders diagnosis;and(5)manuscripts written or published in the English *** Our search identified 582 potentially relevant *** eight of them satisfied the inclusion ***-tive meta-analysis was not deemed appropriate,given the lack of relevant randomized control trials,the small number of relevant shortlisted,the small sample size of the patients included in each study,and the remarkable heterogeneity of the studies'*** The included studies suggest an increased incidence of psychopathology among children and adolescents with CU as opposed to healthy age-matched individuals,but the data are *** research is required to clarify whether psychopathology is just a comorbid entity,the cause,or the consequence of *** an interdisciplinary collaboration between allergists/dermatologists and psychiatrists is expected to substantially minimize CU burden and improve patients'quality of life.

关键词： Adolescents Children Chronic urticaria Psychiatry Psychopathology Quality of life

New therapeutic perspectives in Type 1 Diabetes: dietary interventions prevent β cell-autoimmunity by modifying the gut metabolic environment

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Cellular & Molecular immunology 2017年第12期14卷 951-953页

作者： Chiara Sorini Ilaria Cosorich Marika Falcone immunology and allergy Unit Department of MedicineKarolinska InstitutetStockholmSweden Experimental Diabetes Unit-DRI Division of ImmunologyTransplantation and Infectious DiseasesIRCCS San Raffaele Scientific InstituteMilanItaly

The gut microbiota plays a key role in the pathogenesis of autoimmune diseases such as Type 1 Diabetes(T1D),but the mechanism underlying this modulation is yet to be *** paper highlights the crucial role of the microbiota-induced gut metabolic profile,rather than the presence of single bacterial strains,in T1D *** importantly,it provides the first evidence that dietary interventions can modulate the autoimmune pathogenesis of T1D by altering the gut metabolic environment.

关键词： Diabetes autoimmune pathogenesis

Laparoscopic excision of Meckel's diverticulum in children:What is the current evidence?

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World Journal of Gastroenterology 2014年第41期20卷 15158-15163页

作者： Kin Wai Edwin Chan Kim Hung Lee Hei Yi Vicky Wong Siu Yan Bess Tsui Yuen Shan Wong Kit Yi Kristine Pang Jennifer Wai Cheung Mou Yuk Him Tam Division of paediatric Surgery and paediatric Urology Department of SurgeryThe Prince of Wales HospitalThe Chinese University of Hong Kong

Complications aroused from Meckel’s diverticulum tend to developed in children. Children presented with abdominal pain, intestinal obstruction, intussusception or gastrointestinal bleeding may actually suffered from complicated Meckel’s diverticulum. With the advancement of minimally invasive surgery (MIS) in children, the use of laparoscopy in the diagnosis and subsequent laparoscopic excision of Meckel’s diverticulum has gained popularity. Recently, single incision laparoscopic surgery (SILS) has emerged as a new technique in minimally invasive surgery. This review offers the overview in the development of MIS in the management of children suffered from Meckel’s diverticulum. The current evidence in different laparoscopic techniques, including conventional laparoscopy, SILS, the use of special laparoscopic instruments, intracorporeal diverticulectomy and extracorporeal diverticulectomy in the management of Meckel’s diverticulum in children were revealed.

关键词： Laparoscopy Meckel’ s diverticulum Children

Possible therapeutic role of Ig E blockade in irritable bowel syndrome

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World Journal of Gastroenterology 2016年第43期22卷 9451-9456页

作者： Eli Magen Tinatin Chikovani Medicine C department Allergy and Clinical Immunology Unit Barzilai Medical Center Ben Gurion University of Negev department of Microbiology and Im-munology Tbilisi State Medical University

Omalizumab is a humanized monoclonal antibody that binds to the high-affinity type-I Ig E Fc receptors on mast cells(MCs) and basophils, inhibiting the Ig E immune pathway. Irritable bowel syndrome(IBS) is the most common functional gastrointestinal disorder, and dysregulation of the immune system likely contributes to its etiology and/or symptomatology. Colonic biopsies from patients with IBS demonstrate considerable increase in the number of degranulating MCs releasing histamine in proximity to nerves, and this event may underlie the common IBS symptom of abdominal pain. Pharmacologic control of MC activation and mediator release is a current area of active interest in the field of IBS research. Recently, we and Pearson et al described 2 cases of patients with IBS with diarrhea(IBS-D) showing positive clinical response to omalizumab. In both cases, the female patients had severe, long-lasting IBS-D and achieved an almost complete resolution of IBS symptoms. Both patients were also able to discontinue all IBS medications after commencing the anti-Ig E therapy. For both patients, the omalizumab treatment showed a relatively rapid onset of action, resembling the efficacy observed in and previously reported for patients with chronic spontaneous urticaria. In this Editorial, we discuss the possible biological mechanisms that may underlie the clinical efficacy of omalizumab in IBS. We suggest that there is a need for a well-designed prospective study to investigate the therapeutic effects of anti-Ig E in IBS.

关键词： IgE Omalizumab Irritable bowel syndrome Anti-inflammatory Irritable bowel syndrome with diarrhea