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Generic and disease-specific health-related quality of life in patients with hirschsprung disease:A systematic review and meta-analysis

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World Journal of Gastroenterology 2022年第13期28卷 1362-1376页

作者： Veerle Huizer Naveen Wijekoon Daniëlle Roorda Jaap Oosterlaan Marc A Benninga LW Ernest van Heurn Shaman Rajindrajith Joep PM Derikx Department of Pediatric Surgery Emma Children's HospitalAmsterdam UMCUniversity of Amsterdam and Vrije Universiteit AmsterdamAmsterdam Reproduction and DevelopmentAmsterdam 1105 AZNetherlands Department of Surgery University of Colombo and Department of Paediatric SurgeryLady Ridgeway Hospital for ChildrenColombo 00800Sri Lanka Department of Pediatrics Emma Children's HospitalAmsterdam UMCUniversity of AmsterdamEmma Neuroscience GroupAmsterdam Reproduction and DevelopmentAmsterdam 1105 AZNetherlands Department of Pediatrics Lady Ridgeway Hospital for ChildrenColombo 00800Sri Lanka

BACKGROUND Patients with hirschsprung disease(HD)are at risk of persistent constipation,fecal incontinence or recurrent enterocolitis after surgical treatment,which in turn may impact physical and psychosocial *** health-related quality of life(HRQoL)and disease-specific health-related quality of life are relevant outcome measures to assess the impact of HD on the QoL of these *** To summarize all available evidence on HRQoL of patients with HD after surgery and the impact of possible moderating *** Pubmed,Web of Sciences,Psyc Info and Embase were searched with search terms related to’hirschsprung disease’,’Pediatrics’and’Quality of life’.Mean and standard deviation of generic HRQoL overall and domain scores were extracted from each study,as well as data describing potential factors associated with *** effect models were used for meta-analytic aggregation of generic HRQoL ***-regression was used to assess the relationship between patient and clinical characteristics and generic ***-specific HRQoL outcomes of patients with HD were systematically *** Seventeen articles were included in the systematic review(n=1137 patients)and 15 in the quantitative meta-analysis(n=1024 patients).Four studies reported disease-specific ***’s age ranged between 0 and 21 ***-analytic aggregation showed a nonsignificantly impaired generic HRQoL(d=-0.168[95%CI:-0.481;0.145],P=0.293,I^(2)=94.9)in patients with HD compared to healthy ***(d=-0.042[95%CI:-0.419;0.335],P=0.829,I^(2)=95.1),psychosocial(d=-0.159[95%CI:-0.458;0.141],P=0.299,I^(2)=93.6)and social HRQoL(d=-0.092[95%CI:-0.642;0.457],P=0.742,I^(2)=92.3)were also not significantly lower compared to healthy *** was no relation between health-related outcomes and the sex of the patients and whether generic HRQoL was measured by parental proxy or ***-specific complaints of patients with HD impaired physical

关键词： hirschsprung disease Health-related quality of life Meta-analysis Systematic review Pediatrics

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Persistent bowel dysfunction after surgery for hirschsprung’s disease:A neuropathological perspective

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World Journal of Gastrointestinal Surgery 2021年第8期13卷 822-833页

作者： Sanne J Verkuijl Florian Friedmacher Patrick N Harter Udo Rolle Paul MA Broens Department of Pediatric Surgery University Hospital FrankfurtGoethe University FrankfurtFrankfurt 60590Germany Neurological Institute(Edinger-Institute) University Hospital FrankfurtGoethe University FrankfurtFrankfurt 60528Germany Department of Surgery Division of Pediatric SurgeryUniversity of GroningenUniversity Medical Center GroningenGroningen 9700 RBNetherlands

hirschsprung’s disease(HD)is a congenital disorder,characterized by aganglionosis in the distal part of the gastrointestinal *** complete surgical resection of the aganglionic segment,both constipation and fecal incontinence persist in a considerable number of patients with limited treatment *** is growing evidence for structural abnormalities in the ganglionic bowel proximal to the aganglionosis in both humans and animals with HD,which may play a role in persistent bowel *** abnormalities include:(1)Histopathological abnormalities of enteric neural cells;(2)Imbalanced expression of neurotransmitters and neuroproteins;(3)Abnormal expression of enteric pacemaker cells;(4)Abnormalities of smooth muscle cells;and(5)Abnormalities within the extracellular ***,a better understanding of these previously unrecognized neuropathological abnormalities may improve follow-up and treatment in patients with HD suffering from persistent bowel dysfunction following surgical *** the long term,further combination of clinical and neuropathological data will hopefully enable a translational step towards more individual treatment for HD.

关键词： hirschsprung disease Aganglionosis Proximal Ganglionic Constipation Incontinence©The Author(s)2021

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Prevalence and Clinical Characteristics of Chronic Constipation in Senegalese Children

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Open Journal of Pediatrics 2023年第6期13卷 921-928页

作者： Abou Ba Aliou Thiongane Ndeye Fatou Sow Babacar Niang Younoussa Keïta Fatou Ly Ousmane N’diaye Service de pédiatrie centre hospitalier national Dalal Jamm Dakar Senegal Centre hospitalier national d’enfants Albert Royer Dakar Senegal Service de pédiatrie centre hospitalier Abass Ndao Dakar Senegal Service de pédiatrie centre hospitalier national de Pikine Dakar Senegal

Introduction: Constipation is the infrequent emission of hard stools. It is common, most often of functional origin, with a prevalence ranging from 0.7% to 29.6%. The aim of our study was to investigate the prevalence of constipation in children undergoing paediatric gastroenterology. Methodology: This was a bi-centric retrospective study carried out at the Albert Royer Children’s Hospital and the Dalal Jamm Hospital Pediatrics Department. Children followed in the pediatric gastroenterology consultation during the period from January 1, 2013 to February 29, 2020 were included. Results: A total of 337 children (205 boys;60.83%) were included, representing a prevalence of 25%. The median age was 3 years 6 months. The 13 - 24 months age group was the most frequent (27.6%). Rare stools were the main reason for consultation (92.8%). Rare stools were noted in 92.88% of cases. Bristol types 1 and 2 accounted for 6.53% and 54.60% respectively. The main associated signs were abdominal pain (29.4%), followed by abdominal bloating (20.96%). Abdominal pain was functional in 73.58% of cases. Under treatment, 90.19% of children had a favorable outcome. Conclusion: Constipation is common in Senegalese children with a prevalence close to that reported in Western countries. In most case, it is functional with good prognostic.

关键词： Children Chronic Constipation Bristol Scale Dakar hirschsprung disease

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