Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis

Abstract Background Advances in surgical techniques and perioperative care have improved the short- and mid-term postoperative outcomes of patients with Hirschsprung disease (HD). However, the long-term outcomes of these patients (older than 10 years) have not been fully investigated. The aim of thi...

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Main Authors: Ying Dai, Yongfang Deng, Yan Lin, Runxian Ouyang, Le Li
Format: Article
Language:English
Published: BMC 2020-03-01
Series:BMC Gastroenterology
Subjects:
Online Access:http://link.springer.com/article/10.1186/s12876-020-01208-z
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author Ying Dai
Yongfang Deng
Yan Lin
Runxian Ouyang
Le Li
author_facet Ying Dai
Yongfang Deng
Yan Lin
Runxian Ouyang
Le Li
author_sort Ying Dai
collection DOAJ
description Abstract Background Advances in surgical techniques and perioperative care have improved the short- and mid-term postoperative outcomes of patients with Hirschsprung disease (HD). However, the long-term outcomes of these patients (older than 10 years) have not been fully investigated. The aim of this systematic review is to clarify the prevalence of long-term outcomes and the quality of life of these patients. Methods PubMed, AMED, Cochrane Library, CINAHL and PsycINFO databases were searched from inception to October 2018, following the Meta-analysis of Observational Studies in Epidemiology (MOOSE) guideline. Original studies reporting the outcomes of patients older than ten years with HD were selected and reviewed. The overall prevalence of fecal incontinence, constipation, bowel function score, bladder dysfunction symptoms, and patients’ quality of life were extracted from the included studies and pooled through the random-effects meta-analysis model. The heterogeneity and variation in the pooled estimations were evaluated by Cochrane’s Q test and the I2 test. The sensitivity analysis was conducted by the sequential omission of individual studies. Publication bias was evaluated by Egger’s linear regression test. The whole procedure was conducted with Stata (version 14). Results In total, 3406 articles were identified from the literature search, among which twelve studies, including 625 patients, were included for analysis. The pooled prevalences of fecal incontinence, constipation, and bladder dysfunction symptoms and good to excellent bowel function scores were 0.20 (95% CI 0.13–0.28), 0.14 (95% CI 0.06–0.25), 0.07 (95% CI 0.04–0.12), and 0.95 (95% CI: 0.91–0.97), respectively; the pooled mean score of gastrointestinal-related quality of life was 118 (95% CI: 112.56–123.44). Conclusions HD patients older than ten years old have an overall high prevalence of fecal incontinence and a low quality of life. Targeted and evidence-based follow-up procedures and transitional care are essential to meet these patients’ long-term care needs. Prospective and multicenter research that focuses on the attributes and predictors of the long-term prognosis of patients with HD are necessary.
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spelling doaj.art-5202a465809e499482351f020f6a86f52022-12-22T03:38:50ZengBMCBMC Gastroenterology1471-230X2020-03-0120111310.1186/s12876-020-01208-zLong-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysisYing Dai0Yongfang Deng1Yan Lin2Runxian Ouyang3Le Li4Department of Pediatric Surgery, Guangzhou Women and Children’s Medical Center, Guangzhou Medical UniversityDepartment of Obstetrics and Gynecology, Guangzhou Women and Children’s Medical Center, Guangzhou Medical UniversityDepartment of Nursing, Guangzhou Women and Children’s Medical Center, Guangzhou Medical UniversityDepartment of Pediatric Surgery, Guangzhou Women and Children’s Medical Center, Guangzhou Medical UniversityDepartment of Pediatric Surgery, Guangzhou Women and Children’s Medical Center, Guangzhou Medical UniversityAbstract Background Advances in surgical techniques and perioperative care have improved the short- and mid-term postoperative outcomes of patients with Hirschsprung disease (HD). However, the long-term outcomes of these patients (older than 10 years) have not been fully investigated. The aim of this systematic review is to clarify the prevalence of long-term outcomes and the quality of life of these patients. Methods PubMed, AMED, Cochrane Library, CINAHL and PsycINFO databases were searched from inception to October 2018, following the Meta-analysis of Observational Studies in Epidemiology (MOOSE) guideline. Original studies reporting the outcomes of patients older than ten years with HD were selected and reviewed. The overall prevalence of fecal incontinence, constipation, bowel function score, bladder dysfunction symptoms, and patients’ quality of life were extracted from the included studies and pooled through the random-effects meta-analysis model. The heterogeneity and variation in the pooled estimations were evaluated by Cochrane’s Q test and the I2 test. The sensitivity analysis was conducted by the sequential omission of individual studies. Publication bias was evaluated by Egger’s linear regression test. The whole procedure was conducted with Stata (version 14). Results In total, 3406 articles were identified from the literature search, among which twelve studies, including 625 patients, were included for analysis. The pooled prevalences of fecal incontinence, constipation, and bladder dysfunction symptoms and good to excellent bowel function scores were 0.20 (95% CI 0.13–0.28), 0.14 (95% CI 0.06–0.25), 0.07 (95% CI 0.04–0.12), and 0.95 (95% CI: 0.91–0.97), respectively; the pooled mean score of gastrointestinal-related quality of life was 118 (95% CI: 112.56–123.44). Conclusions HD patients older than ten years old have an overall high prevalence of fecal incontinence and a low quality of life. Targeted and evidence-based follow-up procedures and transitional care are essential to meet these patients’ long-term care needs. Prospective and multicenter research that focuses on the attributes and predictors of the long-term prognosis of patients with HD are necessary.http://link.springer.com/article/10.1186/s12876-020-01208-zChildrenConstipationFollow-upFecal incontinenceHirschsprung diseaseLong-term outcome
spellingShingle Ying Dai
Yongfang Deng
Yan Lin
Runxian Ouyang
Le Li
Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
BMC Gastroenterology
Children
Constipation
Follow-up
Fecal incontinence
Hirschsprung disease
Long-term outcome
title Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
title_full Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
title_fullStr Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
title_full_unstemmed Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
title_short Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis
title_sort long term outcomes and quality of life of patients with hirschsprung disease a systematic review and meta analysis
topic Children
Constipation
Follow-up
Fecal incontinence
Hirschsprung disease
Long-term outcome
url http://link.springer.com/article/10.1186/s12876-020-01208-z
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AT yanlin longtermoutcomesandqualityoflifeofpatientswithhirschsprungdiseaseasystematicreviewandmetaanalysis
AT runxianouyang longtermoutcomesandqualityoflifeofpatientswithhirschsprungdiseaseasystematicreviewandmetaanalysis
AT leli longtermoutcomesandqualityoflifeofpatientswithhirschsprungdiseaseasystematicreviewandmetaanalysis